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Sample records for extremely rare case

  1. Papilliferous Keratoameloblastoma: An Extremely Rare Case Report

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    Neeta Mohanty

    2013-01-01

    Full Text Available Odontogenic tumors develop in the jaw bones from the odontogenic tissue-oral epithelium in tooth germ, enamel organ, dental papilla, reduced enamel epithelium, remnants of Hertwig’s root sheath or dental lamina, and so forth. Hence, a bewildering variety of tumors are encountered in the maxilla and mandible. Ameloblastoma is the second most common odontogenic neoplasm after odontomes, and it has numerous clinical and histologic variants. We report a very rare histologic variant: the papilliferous keratoameloblastoma which is the fifth reported case in the English literature.

  2. Malignant Hidradenocarcinoma in the Lower Extremity: A Case Report of a Rare Tumor.

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    Kane, Brendan; Adler, Evan; Bhandari, Tarun; Rose, Michael; DiGuglielmo, Nicola; Sun, Xiu

    Malignant hidradenocarcinomas are rare soft tissue tumors of sweat gland origin. We present the case of a soft tissue, fungating tumor of 15 years' duration of the medial ankle in an 85-year-old male that exhibited malignant features clinically and radiographically. Subsequent punch biopsy revealed a diagnosis of malignant hidradenocarcinoma. Given the risk of recurrence and the poor radiation and chemotherapy options, the patient initially decided to leave the lesion untreated. However, he soon developed lower extremity cellulitis from the exposed lesion and decided to have the tumor excised, eliminating the source of the infection. In the present case study, we discuss the etiology, clinical and radiographic characteristics, and treatment options for this rare lesion. At the 18-month follow-up visit, he had had no recurrence of the lesion. Copyright © 2017 The American College of Foot and Ankle Surgeons. Published by Elsevier Inc. All rights reserved.

  3. Cryptococcoma mimicking a brain tumor in an immunocompetent patient: case report of an extremely rare presentation.

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    Paiva, Aline Lariessy Campos; Aguiar, Guilherme Brasileiro de; Lovato, Renan Maximilian; Zanetti, Arthus Vilar Deolindo; Panagopoulos, Alexandros Theodoros; Veiga, José Carlos Esteves

    2017-11-06

    Central nervous system (CNS) infectious diseases have high prevalence in developing countries and their proper diagnosis and treatment are very important for public health planning. Cryptococcus neoformans is a fungus that may cause several CNS manifestations, especially in immunocompromised patients. Cryptococcal meningitis is the most common type of involvement. Mass-effect lesions are uncommon: they are described as cryptococcomas and their prevalence is even lower among immunocompetent patients. The aim here was to report an extremely rare case of cryptococcoma causing a mass effect and mimicking a brain tumor in an immunocompetent patient. The literature on CNS cryptococcal infections was reviewed with emphasis on cryptococcomas. Clinical, surgical and radiological data on a female patient with this rare presentation of cryptococcoma mimicking a brain tumor are described. A 54-year-old female patient presented to the emergency department with a rapid-onset progressive history of confusion and completely dependency for basic activities. Neuroimaging showed a left occipital lesion and neurosurgical treatment was proposed. From histopathological evaluation, a diagnosis of cryptococcoma was established. She received clinical support with antifungals, but despite optimal clinical treatment, her condition evolved to death. Cryptococcal infections have several forms of presentation and, in immunocompetent patients, their manifestation may be even more different. Cryptococcoma is an extremely rare presentation in which proper surgical and clinical treatment should be instituted as quickly as possible, but even so, there is a high mortality rate.

  4. An extremely rare neoplasm, histiocytic sarcoma: A report of two cases with an aggressive clinical course

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    Erkan Kayikcioglu

    2017-07-01

    Conclusions: HS is an extremely rare malignant neoplasm of the monocytic/macrophage lineage, with no standardized chemotherapy regimen for multisystemic disease. Metastatic patients have a more aggressive clinical course than those with unifocal disease.

  5. Thyroid Gland Metastasis from Cancer of the Uterine Cervix: An Extremely Rare Case Report.

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    Celik, Suleyman Utku; Besli, Dilara; Sak, Serpil Dizbay; Genc, Volkan

    The thyroid gland is a relatively uncommon site for a metastatic disease, although it is richly supplied with blood. The metastases may originate from various primary sites, mainly kidney, lung, head and neck, and breast. Thyroid metastasis from cervical carcinomas is extremely rare; and only a few cases have been previously reported in the literature. In patient with thyroid nodules and an oncological history, the possibility of thyroid metastasis should be seriously considered. Despite the rarity of the metastasis of cervical carcinoma to the thyroid, it is difficult to say appropriate treatment approach for these lesions. When managing such patients, decision-making should balance the possibility of gaining long-term survival against estimation of the aggressiveness of the disease and its possible complications. Here, a case of thyroid metastasis from a squamous cell carcinoma of the uterine cervix presenting with cervical mass and difficulty in swallowing and its treatment is reported.

  6. An Extremely Rare Case of Advanced Metastatic Small Cell Neuroendocrine Carcinoma of Sinonasal Tract

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    Yu Yu Thar

    2016-01-01

    Full Text Available Small cell neuroendocrine carcinoma (SNEC is a rare form of malignancy. It mainly presents as bronchogenic neoplasm, and the extrapulmonary form accounts for only 0.1% to 0.4% of all cancers. These extrapulmonary tumors have been described most frequently in the urinary bladder, prostate, esophagus, stomach, colon and rectum, gall bladder, head and neck, cervix, and skin. Primary SNEC of the sinonasal tract is extremely rare with only less than 100 cases reported in the literature. Because of extreme rarity and aggressiveness of the tumor, the management for this entity varies considerably mandating multimodality approach. In this paper, we report a patient presented with left-sided facial swelling, and the histopathologic examination confirmed primary SNEC of left sinonasal tract. The tumor involved multiple paranasal sinuses with invasion into the left orbit and left infratemporal fossa and metastasized to cervical lymph nodes and bone. The patient encountered devastating outcome in spite of optimal medical management and treatment with palliative chemotherapy highlighting the necessity for further research of primary SNEC of head and neck.

  7. Carcinoma ex basal cell adenoma of the parotid gland: A report of an extremely rare case.

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    Kusafuka, Kimihide; Kawasaki, Takuya; Nakajima, Takashi; Sugino, Takashi

    2017-07-01

    Malignant non-basaloid tumors that arise from basal cell adenoma (BCA) are extremely rare. The patient was a 72-year-old Japanese male, who had noticed swelling of the left parotid region 21 years ago. A superficial lobectomy was performed. About 60% of the tumor was made up of cribriform and trabecular tissue composed of basaloid cells, which exhibited mild atypia and nuclear expression of β-catenin. This portion of the tumor was considered to be a BCA. In the other part of the tumor, the proliferation of large eosinophilic atypical cells, most of which formed intraductal structures, was observed. These tumor cells displayed cellular atypia, and some of them formed Roman bridge structures or contributed to intracapsular invasion. Immunohistochemically, these cells were positive for cytokeratin 7, gross cystic disease fluid proten-15 (GCDFP-15), androgen receptor (AR), and mammaglobin (MMG) and exhibited a high Ki-67 labeling index. So, this portion of the tumor was considered to be a salivary duct carcinoma (SDC). The tumor's final diagnosis was SDC ex BCA (intracapsular type), which is extremely rare. GCDFP-15, AR, MMG, and Ki-67 are useful immunohistochemical markers for diagnosing SDC ex BCA. © 2017 The Authors. Pathology International Published by Japanese Society of Pathology and John Wiley & Sons Australia, Ltd.

  8. [The hidradenocarcinoma of the wrist - an extremely rare malignant carcinoma: case presentation and literature review].

    Science.gov (United States)

    Arsalan-Werner, A; Mentzel, T; Kempf, B; Sauerbier, M

    2013-10-01

    Hidradenocarcinomas are rare, yet highly malignant tumors of eccrine sweat gland origin. Due to its locally aggressive growth and likelihood for metastasis it should be considered as a differential diagnosis especially in case of suspicious intraoperative findings. We report the case of a 73-year-old female patient presenting with a hidradenocarcinoma of the wrist. Treatment of hidradenocarcinomas is similar to the treatment of sarcomas: The first step is an incisional biopsy and obtaining an expert second opinion on the histopathological diagnosis as well as staging. The second step is a clear margin resection of the tumor and the plastic-surgical reconstruction. A long-term follow-up is mandatory to detect potential recurrence or metastasis. © Georg Thieme Verlag KG Stuttgart · New York.

  9. An Extremely Rare and Unusual Case of Retroperitoneal and Pelvic Metastasis from Squamous Cell Carcinoma of Vallecula

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    Abhishek Purkayastha

    2016-06-01

    Full Text Available We report an extremely rare and unusual case of retroperitoneal and pelvic metastasis from primary squamous cell carcinoma of vallecula. Generally carcinoma oropharynx metastasizes to lungs, liver and bone while retroperitoneal and pelvic metastasis is rarely heard of. To the best of our knowledge this case is one of the scantly reported cases ever of this kind in the world.  A 60-year-old male presented with dysphagia and hoarseness of voice of four month duration.  Computed tomography (CT scan face and neck showed growth right vallecula. Biopsy of lesion showed squamous cell carcinoma. Metastatic work up was negative. He received definitive chemo-radiation. Patient during follow up presented with dyspepsia, abdominal discomfort and weight loss. Whole body positron emission tomography (WB PET scan revealed retroperitoneal and pelvic lymph node deposits which were confirmed as metastasis of squamous cell carcinoma by CT guided fine needle aspiration cytology (FNAC. Patient was exhibited palliative chemotherapy but his general condition deteriorated and he finally succumbed to his metastatic illness. This case is being reported to highlight its extreme rarity, the diagnostic and therapeutic challenges it presented and its overall dismal prognosis.

  10. Probability estimation of rare extreme events in the case of small samples: Technique and examples of analysis of earthquake catalogs

    Science.gov (United States)

    Pisarenko, V. F.; Rodkin, M. V.; Rukavishnikova, T. A.

    2017-11-01

    The most general approach to studying the recurrence law in the area of the rare largest events is associated with the use of limit law theorems of the theory of extreme values. In this paper, we use the Generalized Pareto Distribution (GPD). The unknown GPD parameters are typically determined by the method of maximal likelihood (ML). However, the ML estimation is only optimal for the case of fairly large samples (>200-300), whereas in many practical important cases, there are only dozens of large events. It is shown that in the case of a small number of events, the highest accuracy in the case of using the GPD is provided by the method of quantiles (MQs). In order to illustrate the obtained methodical results, we have formed the compiled data sets characterizing the tails of the distributions for typical subduction zones, regions of intracontinental seismicity, and for the zones of midoceanic (MO) ridges. This approach paves the way for designing a new method for seismic risk assessment. Here, instead of the unstable characteristics—the uppermost possible magnitude M max—it is recommended to use the quantiles of the distribution of random maxima for a future time interval. The results of calculating such quantiles are presented.

  11. Hyperthyroidism secondary to hysterosalpingography: an extremely rare complication: A case report.

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    Ma, Guotao; Mao, Rui; Zhai, Haixin

    2016-12-01

    Hysterosalpingography (HSG), a standard procedure for the evaluation of women with infertility and repetitive pregnancy loss, is associated with complications such as uterine perforation, infection, allergic reactions, syncope, hemorrhage and shock, and pulmonary or retinal embolus. However, hyperthyroidism has not been reported as one of its complications. We report the case of a 33-year-old euthyroid woman who presented to our hospital with palpitation, hand tremor, fatigue, and excessive sweating after HSG. Thyroid function tests revealed a thyroid stimulating hormone (TSH) level of 0.012 μIU/mL (range 0.38-4.34 μIU/mL), free T4 of 2.886 ng/dL (range 0.81-1.89 ng/dL), and free T3 levels of 9.4 pg/mL (range 1.80-4.10 pg/mL), and antithyroglobulin antibody of 31.78 IU/mL (range hyperthyroidism (IIH), but was not treated with antithyroid drugs. She has spontaneously recovered and is pregnant currently. This is the first reported case of overt IIH caused by HSG in a euthyroid patient without risk factors. It suggests that HSG also leads to excessive iodine absorption, which induces secondary hyperthyroidism.

  12. Mining with Rare Cases

    Science.gov (United States)

    Weiss, Gary M.

    Rare cases are often the most interesting cases. For example, in medical diagnosis one is typically interested in identifying relatively rare diseases, such as cancer, rather than more frequently occurring ones, such as the common cold. In this chapter we discuss the role of rare cases in Data Mining. Specific problems associated with mining rare cases are discussed, followed by a description of methods for addressing these problems.

  13. Testicular calculus: A rare case.

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    Sen, Volkan; Bozkurt, Ozan; Demır, Omer; Tuna, Burcin; Yorukoglu, Kutsal; Esen, Adil

    2015-01-01

    Testicular calculus is an extremely rare case with unknown etiology and pathogenesis. To our knowledge, here we report the third case of testicular calculus. A 31-year-old man was admitted to our clinic with painful solid mass in left testis. After diagnostic work-up for a possible testicular tumour, he underwent inguinal orchiectomy and histopathologic examination showed a testicular calculus. Case hypothesis: Solid testicular lesions in young adults generally correspond to testicular cancer. Differential diagnosis should be done carefully. Future implications: In young adults with painful and solid testicular mass with hyperechogenic appearance on scrotal ultrasonography, testicular calculus must be kept in mind in differential diagnosis. Further reports on this topic may let us do more clear recommendations about the etiology and treatment of this rare disease.

  14. A rare case of extremely high counts of circulating tumor cells detected in a patient with an oral squamous cell carcinoma

    International Nuclear Information System (INIS)

    Wu, Xianglei; Mastronicola, Romina; Tu, Qian; Faure, Gilbert Charles; De Carvalho Bittencourt, Marcelo; Dolivet, Gilles

    2016-01-01

    Despite aggressive regimens, the clinical outcome of head and neck squamous cell carcinoma remains poor. The detection of circulating tumor cells could potentially improve the management of patients with disseminated cancer, including diagnosis, treatment strategies, and surveillance. Currently, CellSearch ® is the most widely used and the only Food and Drug Administration-cleared system for circulating tumor cells detection in patients with metastatic breast, colorectal, or prostate cancer. In most cases of head and neck squamous cell carcinoma, only low counts of circulating tumor cells have been reported. A 56-year-old white male with no particular medical history, was diagnosed with a squamous cell carcinoma of oral cavity. According to the imaging results (computed tomography and 18 F-fluorodeoxyglucose positron emission tomography / computed tomography) and panendoscopy, the TNM staging was classified as T4N2M0. A non-interruptive pelvimandibulectomy was conducted according to the multidisciplinary meeting advices and the postoperative observations were normal. The patient complained of a painful cervical edema and a trismus 6 weeks after the surgery. A relapse was found by computed tomography and the patient died two weeks later. The search for circulating tumor cells in peripheral venous blood by using the CellSearch ® system revealed a very high count compared with published reports at three time points (pre-operative: 400; intra-operative: 150 and post-operative day 7: 1400 circulating tumor cells). Of note, all detected circulating tumor cells were epidermal growth factor receptor negative. We report here for the first time a rare case of oral squamous cell carcinoma with extremely high circulating tumor cells counts using the CellSearch ® system. The absolute number of circulating tumor cells might predict a particular phase of cancer development as well as a poor survival, potentially contributing to a personalized healthcare

  15. Glomus tumor of penis- A rare case

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    Madhuri S. Kate

    2013-01-01

    Full Text Available Glomus tumors are rare, painful, and benign soft tissue tumors of the skin arising from the glomus body, an arteriovenous thermoregulatory structure. This lesion is usually found on the extremities. Glomus tumor involving the external genital organs including penis is extremely rare. Until now, only few cases have been reported in the available literature. A 22 year old male patient presented with a painful nodule over the penis. The pathologic diagnosis was glomus tumor of the glans penis. We report herewith a case of glomus tumor of penis diagnosed on histopathologic examination. Glomus tumor, being a benign neoplasm, complete extirpation of the glomus tumor is the treatment of choice.

  16. A rare case of Charlin's syndrome

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    Mohit P Singh

    2017-01-01

    Full Text Available Charlin's syndrome is an extremely rare condition characterized by pain in the nasal and paranasal areas, which is precipitated by touching the lateral aspect of the ipsilateral nostril. We are presenting one such case of a 42-year-old man who was admitted to Dr. Chaudhary Hospital and Medical Research Centre.

  17. A Rare Seen Case Report: Sirenomelia

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    Nilay Piskinpasa

    2016-01-01

    Full Text Available Sirenomelia is a rare congenital anomaly characterized by abnormal development of the caudal body structures.The disease can be diagnosed antenatally by the careful ultrasound examination. In this paper we aimed to discuss a case which had one lower extremity,spinal angulations and anhydramnios image detected by ultrasound examination.We diagnosed sirenomelia after termination.

  18. Two rare cases of laryngeal intralymphatic histiocytosis

    DEFF Research Database (Denmark)

    Reznitsky, Martin; Daugaard, Søren; Charabi, Birgitte Wittenborg

    2016-01-01

    We report two rare cases of intralymphatic histiocytosis causing, respectively, recurrent and persistent episodes of upper airway swelling and breathing difficulties. Case 1 was a 39-year-old man who was referred with recurrent upper airway swelling causing difficulty in breathing. A direct....... Extensive investigations were performed but discovered no abnormal findings. He received CO2 laser treatment twice and the swelling decreased. Intralymphatic histiocytosis is extremely rare in upper airway pathology. It is an important differential diagnosis in patients with recurrent and chronic laryngeal...

  19. Spontaneous Rupture of Splenic Artery Aneurysm during the First Trimester of Pregnancy: Report of an Extremely Rare Case and Review of the Literature

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    Theodoros Pavlis

    2012-01-01

    Full Text Available Splenic artery aneurysm (SAA occurs predominantly in women and the majority of them are asymptomatic until rupture. In cases of spontaneous rupture of an SAA, maternal and fetal mortality rates remain extremely high. Furthermore, the spontaneous ruptures of SAAs predominantly appear during the third trimester of pregnancy. We present the third known case of spontaneous SAA rupture during the first trimester of pregnancy, which manifested as sudden hypovolemic collapse and was successfully confronted with combined aggressive resuscitation and emergency surgical operation.

  20. Dioctophymiasis: A Rare Case Report.

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    Chauhan, Sapna; Kaval, Sunil; Tewari, Swati

    2016-02-01

    Dioctophyma renale commonly known as "giant kidney worm' is found in the kidney of carnivorous mammals. Human infestation is rare, but results in destruction of the kidneys. Very few cases have been reported worldwide. We are here reporting a case of Dioctophymiasis in a 35-year-old male patient who presented with retention of urine and subsequent passage of worm and blood in urine. The worm was confirmed as Dioctophyma renale based on its morphology and clinical presentation. This is a very rare case report and to best of our knowledge only two cases have been reported from India.

  1. Laws of small numbers extremes and rare events

    CERN Document Server

    Falk, Michael; Hüsler, Jürg

    2004-01-01

    Since the publication of the first edition of this seminar book in 1994, the theory and applications of extremes and rare events have enjoyed an enormous and still increasing interest. The intention of the book is to give a mathematically oriented development of the theory of rare events underlying various applications. This characteristic of the book was strengthened in the second edition by incorporating various new results on about 130 additional pages. Part II, which has been added in the second edition, discusses recent developments in multivariate extreme value theory. Particularly notable is a new spectral decomposition of multivariate distributions in univariate ones which makes multivariate questions more accessible in theory and practice. One of the most innovative and fruitful topics during the last decades was the introduction of generalized Pareto distributions in the univariate extreme value theory. Such a statistical modelling of extremes is now systematically developed in the multivariate fram...

  2. Laws of small numbers extremes and rare events

    CERN Document Server

    Falk, Michael; Reiss, Rolf-Dieter

    2011-01-01

    Since the publication of the first edition of this seminar book in 1994, the theory and applications of extremes and rare events have enjoyed an enormous and still increasing interest. The intention of the book is to give a mathematically oriented development of the theory of rare events underlying various applications. This characteristic of the book was strengthened in the second edition by incorporating various new results. In this third edition, the dramatic change of focus of extreme value theory has been taken into account: from concentrating on maxima of observations it has shifted to l

  3. Ainhum - A Rare Case Report.

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    Prabhu, Ravi; Kannan, Narayanasamy Subbaraju; Vinoth, Sundaresan; Praveen, Chinnappan Balasubramanian

    2016-04-01

    The term 'AINHUM' is derived from the African word meaning 'to saw or cut'. True ainhum otherwise called dactylolysis spontanea is a condition involving soft tissue or digits with constricting rings commonly presenting in fifth toes, usually bilateral. It is to be differentiated from Pseudo-ainhum that occurs secondary to some hereditary and nonhereditary diseases that lead to annular constriction of digits. We report a rare case of true ainhum involving the left fourth toe only. It is a very rare case and a very few were reported worldwide. The highest incidence of ainhum has been reported in South Africa and South America. It is rarely reported in India. Ainhum when diagnosed and treated in early stages can be prevented from progressing to mutilating deformities.

  4. Rare case of acute dengue encephalitis with correlated MRI findings

    International Nuclear Information System (INIS)

    Mathew, Rishi Philip; Basti, Ram Shenoy; Hegde, Pavan; Devdas, Jaidev M.; Khan, Habeeb Ullah; Bukelo, Mario Joseph

    2014-01-01

    Dengue encephalitis is extremely rare, with most patients showing no significant abnormality on neuroimaging (CT/MRI). We report one of the very few documented cases of dengue encephalitis, with abnormal signal intensities on all major sequences on brain MRI.

  5. An integrated wave modelling framework for extreme and rare events for climate change in coastal areas – the case of Rethymno, Crete

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    Vasiliki K. Tsoukala

    2016-04-01

    Full Text Available Coastal floods are regarded as among the most dangerous and harmful of all natural disasters affecting urban areas adjacent to the shorelines. Rapid urbanization combined with climate change and poor governance often results in significant increase of flood risk, especially for coastal communities. Wave overtopping and wave run-up are the key mechanisms for monitoring the results of coastal flooding and as such, significant efforts are currently focusing on their predicting. In this paper, an integrated methodology is proposed, accounting for wave overtopping and wave run-up under extreme wave scenarios caused by storm surges. By taking advantage of past and future climatic projections of wind data, a downscaling approach is proposed, utilizing a number of appropriate numerical models than can simulate the wave propagation from offshore up to the swash zone. The coastal zone of Rethymno in Greece is selected as a case study area and simulations of wave characteristics with the model SWAN for the period 1960–2100 in the offshore region are presented. These data are given as boundary conditions to further numerical models (MIKE21 PMS and HD in order to investigate the spatial evolution of the wave and the hydrodynamic field in intermediate and shallow waters. Finally, the calculated wave height serves as input to empirical formulas and time dependent wave propagation models (MIKE21 BW to estimate the wave run-up and wave overtopping (EurOtop. It is suggested that the proposed procedure is generic enough to be applicable to any similar region.

  6. Microfibrillar cardiomyopathy: A rare case

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    Narender Kumar

    2011-01-01

    Full Text Available Microfibrillar cardiomyopathy is a very rare cause of restrictive cardiomyopathy (RCM. The index case was a male patient who presented with shortness of breath and pedal edema. Further clinical investigations favored a clinical diagnosis of RCM. An endomyocardial biopsy revealed subendocardial and interstitial hyaline eosinophillic material resembling amyloid that did not stain with Congo red. An electron microscopic examination showed that this material was composed of twisted linear and bundles of tangled microfibrils. The etiology of the microfibrillar deposition is currently unknown. The pathologists should entertain the diagnosis of microfibrillar cardiomyopathy in suspected cases of amyloidosis that are negative for Congo red.

  7. Ergotamine-induced upper extremity ischemia: a case report

    Energy Technology Data Exchange (ETDEWEB)

    Kim, Man Deuk; Lee, Gun [Bundang CHA General Hospital, Pochon (China); Shin, Sung Wook [Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul (Korea, Republic of)

    2005-06-15

    Ergotamine-induced limb ischemia is an extremely rare case. We present a case of a 64-year-old man, who developed ischemia on the right upper extremity due to long-term use of Ergot for migraine headache. Angiography revealed diffused, smooth, and tapered narrowing of the brachial artery. The patient was successfully treated with intravenous nitroprusside.

  8. Progeria in siblings: A rare case report

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    R Sowmiya

    2011-01-01

    Full Text Available Progeria, also known as Hutchinson-Gilford syndrome, is an extremely rare, severe genetic condition wherein symptoms resembling aspects of aging are manifested at an early age. It is an autosomal dominant disorder. It is not seen in siblings of affected children although there are very few case reports of progeria affecting more than one child in a family. Here we are presenting two siblings, a 14-year-old male and a 13-year-old female with features of progeria, suggesting a possible autosomal recessive inheritance.

  9. Nodules on the Hair: A Rare Case of Mixed Piedra

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    Khatu, Swapna S; Poojary, Shital Amin; Nagpur, Niranjan G

    2013-01-01

    Piedra is a superficial fungal infection of the hair shaft characterized by nodules along the hair shaft. Black piedra affects the scalp hair more frequently than white piedra. Occurrence of both types of piedra simultaneously in a patient is extremely rare. We describe here a rare case of mixed piedra of scalp hair.

  10. Nodules on the hair: a rare case of mixed piedra.

    Science.gov (United States)

    Khatu, Swapna S; Poojary, Shital Amin; Nagpur, Niranjan G

    2013-10-01

    Piedra is a superficial fungal infection of the hair shaft characterized by nodules along the hair shaft. Black piedra affects the scalp hair more frequently than white piedra. Occurrence of both types of piedra simultaneously in a patient is extremely rare. We describe here a rare case of mixed piedra of scalp hair.

  11. A rare disease in an atypical location - Kimura's Disease of the upper extremity

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    Lam, Alan Cheuk Si; Lau, Vince Wing Hang [Queen Mary Hospital, Department of Radiology, Hong Kong (China); Au Yeung, Rex Kwok Him [University of Hong Kong, Department of Pathology, Li Ka Shing Faculty of Medicine, Hong Kong (China)

    2015-12-15

    Kimura's disease is a rare chronic inflammatory disorder predominantly affecting young Asian male patients, occurring mainly in the head and neck regions. Kimura's disease of the upper extremity is extremely rare, and previous case reports in the literature show similar imaging characteristics with consistent location at the medial epitrochlear region, predominantly with unilateral involvement. We present the first reported case of Kimura's disease affecting the anterolateral aspect of the upper arm, sparing the medial epitrochlear region, illustrating that with typical MR appearance and serology, the involvement of this rare disease in an atypical location still warrants consideration of this diagnosis. There was also bilateral asymmetrical involvement in our patient, suggesting the possibility of a propensity for Kimura's disease affecting the upper extremities to have bilateral involvement, which may necessitate imaging of the clinically asymptomatic contralateral limb in these patients for early lesion identification and treatment. (orig.)

  12. A rare disease in an atypical location - Kimura's Disease of the upper extremity

    International Nuclear Information System (INIS)

    Lam, Alan Cheuk Si; Lau, Vince Wing Hang; Au Yeung, Rex Kwok Him

    2015-01-01

    Kimura's disease is a rare chronic inflammatory disorder predominantly affecting young Asian male patients, occurring mainly in the head and neck regions. Kimura's disease of the upper extremity is extremely rare, and previous case reports in the literature show similar imaging characteristics with consistent location at the medial epitrochlear region, predominantly with unilateral involvement. We present the first reported case of Kimura's disease affecting the anterolateral aspect of the upper arm, sparing the medial epitrochlear region, illustrating that with typical MR appearance and serology, the involvement of this rare disease in an atypical location still warrants consideration of this diagnosis. There was also bilateral asymmetrical involvement in our patient, suggesting the possibility of a propensity for Kimura's disease affecting the upper extremities to have bilateral involvement, which may necessitate imaging of the clinically asymptomatic contralateral limb in these patients for early lesion identification and treatment. (orig.)

  13. Retroperitoneal Schwannoma: A Rare Case

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    Murat Kalaycı

    2011-01-01

    Case Presentation. A 38-year-old woman was admitted to the emergency service with the complaints of progressive abdominal pain and nausea for the last 24 hours. Abdominal examination was compatible with acute abdomen. Acute appendicitis was diagnosed by CT. During CT evaluation, a round shaped soft-tissue mass at the retroperitoneal area inferior to the right kidney was detected, The mass was resected and histology revealed schwannoma. Conclusion. Rare tumoral lesions with benign course such as schwannoma can be detected incidentally.

  14. Rare case of gastrointestinal stromal tumor of the anal canal

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    Madhu Kumar

    2013-01-01

    Full Text Available Gastrointestinal stromal tumor (GIST is a rare mesenchymal neoplasm of the gastrointestinal tract. GIST of anal canal is very rare representing only 3% of all anorectal mesenchymal tumors. We report an extremely rare case of GIST of the anal canal in 60-years-old man with history of irregular bowel habits with dark colored stool mixed with blood and constipation from 6 month. Diagnosis was made on the basis of histomorphological and immunohistochemical examination.

  15. Waardenburg syndrome: A rare case

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    Shivlal M Rawlani

    2018-01-01

    Full Text Available Waardenburg Syndrome is a rare disorder of neural crest cell development. It is genetically inherited. Varying in prevalence from 1:42000 to 1:50,000, it compromises approximately 2-5% of congenital deaf children. The syndrome is not expressed in its complete form, in about 20% cases, which adds for its heterogenisity . Even among people affected in the same family,the features do vary. Unilateral heterochromia that manifests as lighter pigmentation of one iris is associated with Waardenburg syndrome and Parry-Romberg syndrome and less commonly with Hirschsprung disease. A case of ten yrs. old boy with a typical facial profile and hearing loss is reported.

  16. A rare case of benign omentum teratoma

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    Sforza Marcos

    2012-01-01

    Full Text Available Introduction. Mature teratomas (benign cystic teratomas or dermoid cysts are among the most common ovarian tumours; however, teratomas of the omentum and mesentery are extremely rare. Teratoma in the intraperitoneal cavity is uncommon and atypical, and it is even more uncommon in adulthood. Case Outline. An 82-year-old female was admitted to our department with clinical signs of abdominal tumour. The ultrasound scan and preoperative laboratory tests were done. Explorative laparotomy revealed tumour with torsion on its pedicle at the greater omentum. After removal of the mass and the incision a tooth and hair were found, characteristics of teratoma. Conclusion. The excision was very effective and also definitive treatment for this case. The patient recovered well and was discharged 3 days later. The patient probably carried the tumour all her life asymptomatically until admission.

  17. Intraoral hydatid cyst: A rare case report

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    Ravi Kiran Alaparthi

    2015-01-01

    Full Text Available "Hydatid" is a Greek word which means "a drop of water." Hydatid cysts occur in hydatid disease or echinococcosis, which is one of the most geographically prevalent zoonosis. This zoonotic infection in humans is mainly caused by infection by the larval stage of the dog tapeworm Echinococcus granulosus. This tapeworm infestation is widespread in sheep, goats, cattle, and dogs. This chronic disease is present worldwide among herding populations who live in close proximity to dogs and herd animals. It is a serious and potentially fatal condition and symptoms may occur a long time after the early infection. The most frequently affected organs are liver, lungs, followed by bones and brain, and extremely unusual occurrence has been found in the oral cavity, which was noticed in the present case. So hereby, we reveal a very rare case of intraoral hydatid cyst in a 20-year-old female patient.

  18. Extreme Quantum Memory Advantage for Rare-Event Sampling

    Science.gov (United States)

    Aghamohammadi, Cina; Loomis, Samuel P.; Mahoney, John R.; Crutchfield, James P.

    2018-02-01

    We introduce a quantum algorithm for memory-efficient biased sampling of rare events generated by classical memoryful stochastic processes. Two efficiency metrics are used to compare quantum and classical resources for rare-event sampling. For a fixed stochastic process, the first is the classical-to-quantum ratio of required memory. We show for two example processes that there exists an infinite number of rare-event classes for which the memory ratio for sampling is larger than r , for any large real number r . Then, for a sequence of processes each labeled by an integer size N , we compare how the classical and quantum required memories scale with N . In this setting, since both memories can diverge as N →∞ , the efficiency metric tracks how fast they diverge. An extreme quantum memory advantage exists when the classical memory diverges in the limit N →∞ , but the quantum memory has a finite bound. We then show that finite-state Markov processes and spin chains exhibit memory advantage for sampling of almost all of their rare-event classes.

  19. Extreme Quantum Memory Advantage for Rare-Event Sampling

    Directory of Open Access Journals (Sweden)

    Cina Aghamohammadi

    2018-02-01

    Full Text Available We introduce a quantum algorithm for memory-efficient biased sampling of rare events generated by classical memoryful stochastic processes. Two efficiency metrics are used to compare quantum and classical resources for rare-event sampling. For a fixed stochastic process, the first is the classical-to-quantum ratio of required memory. We show for two example processes that there exists an infinite number of rare-event classes for which the memory ratio for sampling is larger than r, for any large real number r. Then, for a sequence of processes each labeled by an integer size N, we compare how the classical and quantum required memories scale with N. In this setting, since both memories can diverge as N→∞, the efficiency metric tracks how fast they diverge. An extreme quantum memory advantage exists when the classical memory diverges in the limit N→∞, but the quantum memory has a finite bound. We then show that finite-state Markov processes and spin chains exhibit memory advantage for sampling of almost all of their rare-event classes.

  20. Cronkhite-Canada Syndrome (CCS)-A Rare Case Report.

    Science.gov (United States)

    Chakrabarti, Subrata

    2015-03-01

    Cronkhite-Canada syndrome (CCS) is an extremely rare non-inherited condition characterized by gastrointestinal hamartomatous polyposis, alopecia, onychodystrophy, hyperpigmentation, weight loss and diarrhoea. The aetiology is probably autoimmune and diagnosis is based on history, physical examination, endoscopic findings of gastrointestinal polyposis, and histology. The disease is very rare; approximately 450 cases of CCS have been reported worldwide. The author reports a case of CCS in an elderly Indian male.

  1. Hiccup: An Extremely Rare Presentation of Thyrotoxicosis of Graves’ Disease

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    Irshad Parray

    2011-03-01

    Full Text Available Persistent hiccup is a rare but potentially severe condition that can be symptomatic of a variety of diseases or idiopathic. Most episodes last only a few minutes and are self-limited, but hiccup can get persistent and become a real problem for physician and patient alike. The center of hiccup may be activated by a great variety of stimuli travelling along different nerve pathways and bring different effecter responses. We report a case of persistent hiccup as a presentation of impending thyroid storm of Graves’ disease. Though the condition is rare, clinicians should remain alert to the possibility of this diagnosis.

  2. Primary laryngeal leishmaniasis: A rare case report

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    Kumar Bipin

    2009-01-01

    Full Text Available Laryngeal leishmaniasis is extremely rare. We report a case of primary laryngeal leishmaniasis in a 70-year-old male who was admitted with complaints of gradual progressive hoarseness of the voice, dyspnea, cough for the past 3 months and noisy breathing for the past 5 days. An X-ray of the soft tissue of the neck showed a prevertebral soft tissue mass causing narrowing of the airway at the C6-C7 vertebral level. A computerized tomography (CT scan showed a soft tissue mass in the subglottic region causing significant narrowing of the airway. A direct laryngoscopy showed a pinkish-white, friable mass involving the subglottic region and the anterior half of the vocal cords. With the clinical suspicion of malignancy, an endoscopic biopsy was done. A histopathological examination showed diffuse mixed inflammatory cell infiltrate in subepithelium with numerous Leishmania donovani bodies in the cytoplasm of histiocytes.

  3. Cutaneous collagenous vasculopathy: A rare case report

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    Kinjal Deepak Rambhia

    2016-01-01

    Full Text Available Cutaneous collagenous vasculopathy (CCV is a distinct, rare, and underdiagnosed condition. We report a case of CCV in a 50-year-old woman presenting as asymptomatic, erythematous to hyperpigmented nonblanchable macules over both the lower extremities. The clinical differential diagnosis of the lesions was pigmented purpuric dermatoses (Schamberg's purpura and cutaneous small vessel vasculitis. Histology of the lesions revealed dilated superficial dermal vessels with abundant pink hyaline material in the vessel wall, which stained with periodic acid Schiff stain. The patient was diagnosed as CCV. This condition remains largely underdiagnosed and is commonly mistaken for pigmented purpuric dermatosis or generalized essential telangiectasia. Emphasis on the differentiation of CCV from its clinical and histological mimicks is made.

  4. Scapulothoracic Dissociation: A Rare Variant: A Case Report

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    Rajat Jangir

    2014-07-01

    Full Text Available Scapulothoracic dissociation is a rare injury involving separation of scapula from the thorax along with the upper extremity. Majority of the patients have concomitant neurovascular injury and the prognosis is uniformly poor in such cases. We present a case of scapulothoracic dissociation with comminuted fracture of scapula and acromioclavicular joint disruption without neurovascular deficit. There were associated avulsion fractures of the spinous processes of vertebrae (T3-T5. Such presentation is rare in an already rare scapulothoracic dissociation injury. A discussion regarding the probable mechanism of injury, management and prognosis is presented.

  5. Extreme magnetoresistance in magnetic rare-earth monopnictides

    Science.gov (United States)

    Ye, Linda; Suzuki, Takehito; Wicker, Christina R.; Checkelsky, Joseph G.

    2018-02-01

    The acute sensitivity of the electrical resistance of certain systems to magnetic fields known as extreme magnetoresistance (XMR) has recently been explored in a new materials context with topological semimetals. Exemplified by WTe2 and rare-earth monopnictide La(Sb,Bi), these systems tend to be nonmagnetic, nearly compensated semimetals and represent a platform for large magnetoresistance driven by intrinsic electronic structure. Here we explore electronic transport in magnetic members of the latter family of semimetals and find that XMR is strongly modulated by magnetic order. In particular, CeSb exhibits XMR in excess of 1.6 ×106% at fields of 9 T whereas the magnetoresistance itself is nonmonotonic across the various magnetic phases and shows a transition from negative magnetoresistance to XMR with fields above magnetic ordering temperature TN. The magnitude of the XMR is larger than in other rare-earth monopnictides including the nonmagnetic members and follows a nonsaturating power law to fields above 30 T. We show that the overall response can be understood as the modulation of conductivity by the Ce orbital state and for intermediate temperatures can be characterized by an effective medium model. Comparison to the orbitally quenched compound GdBi supports the correlation of XMR with the onset of magnetic ordering and compensation and highlights the unique combination of orbital inversion and type-I magnetic ordering in CeSb in determining its large response. These findings suggest a paradigm for magneto-orbital control of XMR and are relevant to the understanding of rare-earth-based correlated topological materials.

  6. A genome-wide study reveals rare CNVs exclusive to extreme phenotypes of Alzheimer disease.

    Science.gov (United States)

    Rovelet-Lecrux, Anne; Legallic, Solenn; Wallon, David; Flaman, Jean-Michel; Martinaud, Olivier; Bombois, Stéphanie; Rollin-Sillaire, Adeline; Michon, Agnès; Le Ber, Isabelle; Pariente, Jérémie; Puel, Michèle; Paquet, Claire; Croisile, Bernard; Thomas-Antérion, Catherine; Vercelletto, Martine; Lévy, Richard; Frébourg, Thierry; Hannequin, Didier; Campion, Dominique

    2012-06-01

    Studying rare extreme forms of Alzheimer disease (AD) may prove to be a useful strategy in identifying new genes involved in monogenic determinism of AD. Amyloid precursor protein (APP), PSEN1, and PSEN2 mutations account for only 85% of autosomal dominant early-onset AD (ADEOAD) families. We hypothesised that rare copy number variants (CNVs) could be involved in ADEOAD families without mutations in known genes, as well as in rare sporadic young-onset AD cases. Using high-resolution array comparative genomic hybridisation, we assessed the presence of rare CNVs in 21 unrelated ADEOAD cases, having no alteration on known genes, and 12 sporadic AD cases, with an age of onset younger than 55 years. The analysis revealed the presence of 7 singleton CNVs (4 in ADEOAD and 3 in sporadic cases) absent in 1078 controls and 912 late-onset AD cases. Strikingly, 4 out of 7 rearrangements target genes (KLK6, SLC30A3, MEOX2, and FPR2) encoding proteins that are tightly related to amyloid-β peptide metabolism or signalling. Although these variants are individually rare and restricted to particular subgroups of patients, these findings support the causal role, in human pathology, of a set of genes coding for molecules suspected for a long time to modify Aβ metabolism or signalling, and for which animal or cellular models have already been developed.

  7. Computer tomography for rare soft tissue tumours of the extremities

    International Nuclear Information System (INIS)

    Boettger, E.; Semerak, M.; Stoltze, D.; Rossak, K.

    1979-01-01

    Five patients with undiagnosed soft tissue masses in the extremities were examined and in two a pathological diagnosis could be made. One was an extensive, invasive fibroma (desmoid) 22 cm long which could be followed from the thigh almost into the pelvis. It was sharply demarkated form the surrounding muscles and of higher density. The second case was a 12 cm long cavernous haemangioma in the semi-membranosus muscle. This was originally hypo-dense, but showed marked increase in its density after the administration of contrast. (orig.) [de

  8. Lipofibromatous Hamartoma of the Plantar Nerve An Extremely Rare Localization.

    Science.gov (United States)

    Mert, Murat; Hacısalihoglu, Payam

    2018-03-01

    Lipofibromatous hamartoma (LFH) is a rare, benign, tumor-like soft-tissue lesion that affects the peripheral nerves and forms a palpable neurogenic mass. Lipofibromatous hamartoma is associated with pain and sensory and/or motor deficits in the area of innervation of the affected nerve. This report describes a rare case of LFH of the plantar nerve. A 48-year-old woman presented to our outpatient orthopedic clinic with pain and a burning sensation on her left foot. The patient had a history of Morton's neuroma and had undergone a tarsal tunnel operation 2 years earlier at another center. None of her symptoms was alleviated by two previous operations. Magnetic resonance imaging with contrast revealed tenosynovitis of the flexor hallucis longus tendon and signal changes at deep tissue planes of the foot at the levels of the second and third toes, on the dorsal site and subcutaneous soft-tissue planes, suggesting edema and Morton's neuroma. The lesion was excised under spinal anesthesia, and histopathologic examination of the specimen revealed a diagnosis of LFH. The patient was discharged without any symptoms and her foot was normal at 8-month outpatient follow-up, with no indications of postoperative complications and/or recurrence.

  9. A rare case of spinal dural arteriovenous fistula

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    Mariya Apostolova

    2012-12-01

    Full Text Available Spinal dural arteriovenous fistula (SDAVF is a rare vascular malformation of the spine. Only a limited number of cases of SDAVF have been reported in the current literature. We describe the case of a 74 year old male who presented with gradually progressive bilateral lower extremity weakness and bladder dysfunction and was subsequently diagnosed with SDAVF affecting both the thoracic and lumbar spine. The patient later underwent embolization with some improvement in his neurologic symptoms.

  10. Facial nerve hemangioma: a rare case involving the vertical segment.

    Science.gov (United States)

    Ahmadi, Neda; Newkirk, Kenneth; Kim, H Jeffrey

    2013-02-01

    This case report and literature review reports on a rare case of facial nerve hemangioma (FNH) involving the vertical facial nerve (FN) segment, and discusses the clinical presentation, imaging, pathogenesis, and management of these rare lesions. A 53-year-old male presented with a 10-year history of right hemifacial twitching and progressive facial paresis (House-Brackmann grading score V/VI). The computed tomography and magnetic resonance imaging studies confirmed an expansile lesion along the vertical FN segment. Excision and histopathologic examination demonstrated FNH. FNHs involving the vertical FN segment are extremely rare. Despite being rare lesions, we believe that familiarity with the presentation and management of FNHs are imperative. Laryngoscope, 2012. Copyright © 2012 The American Laryngological, Rhinological, and Otological Society, Inc.

  11. Synovial sarcoma of primary bone origin: a rare case in a rare site with atypical features

    International Nuclear Information System (INIS)

    Jung, Seung Chai; Choi, Jung-Ah; Lee, Joon Woo; Kang, Heung Sik; Chung, Jin-Haeng; Oh, Joo Han

    2007-01-01

    Synovial sarcoma of bone origin is extremely rare and difficult to diagnose. We present a case in which the lesion arose in the cortex of the distal tibia. It showed heterogeneous intermediate signal intensity on T1-weighted images and heterogeneous intermediate to low signal intensity on T2-weighted images with heterogeneous contrast enhancement on MRI. The lesion was confirmed as synovial sarcoma using a combination of histological and molecular genetic studies. (orig.)

  12. Ambras syndrome: A rare case report

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    A Ishita

    2016-01-01

    Full Text Available Congenital generalized hypertrichosis associated with gingival hyperplasia are rare cases published in literature. The frequency incidence of generalized congenital hypertrichosis is about one to billions of people. Hypertrichosis and gingival hyperplasia are termed as Ambras syndrome (AS, which can be noticed at birth or soon after. Here, is a rare case report of 4-year-old male child who presented with generalized hypertrichosis with gingival fibromatosis and dysmorphic facial features.

  13. Rhinoentomophthoromycosis: A rare case report

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    S Agrawal

    2013-01-01

    Full Text Available Entomophthoromycosis is chronic granulomatous fungal infection with varied presentation as subcutaneous,mucocutaneous and visceral infections. The majority of the subcutaneous infection caused by entomophthoralean fungi involves Basidiobolus spp, C. coronatus, or C. incongruous. A case of rhinoentomophthoromycosis in an immunocompetent male involving maxillary sinus and nose is presented. The patient was clinically diagnosed as malignancy of nose but microscopy and histopathology of the aspirate clinched the diagnosis. The patient responded to antifungal therepy.

  14. A RARE CASE OF NEUROBRUCELLOSIS

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    Sreedevi T

    2017-03-01

    Full Text Available BACKGROUND One of the most common causes of fever with back pain in India is brucellosis along with tuberculosis infection of the spinal cord and vertebral column. Brucellosis is a multisystem infection with a broad-spectrum of clinical presentations. Its nervous system involvement is known as neurobrucellosis is complication of brucellosis occurring in 0 to 25% and can present as meningitis, encephalitis, myelitis-radiculoneuritis, brain abscess, peripheral neuropathy and psychosis. MATERIALS AND METHODS In this case study, we present a 54-year-old male patient a cattle worker with unexplained back pain. Appropriate blood investigations, CSF analysis and MRI scan were taken towards the cause of the diagnosis. RESULTS CSF analysis showed lymphocyte pleocytosis with agglutination in Rose Bengal test. MRI diagnostic of neurobrucellosis revealed anterosuperior osteophyte inflammations, namely Pedro Pons’ sign or Pom’s sign. CONCLUSION Through this case report, we would like to notify that neurobrucellosis should be considered as one of the differential diagnosis and not to be missed as it is treatable. In such conditions, patient has to be treated with two or more antimicrobials such as rifampicin, co-trimoxazole and doxycycline in order to achieve complete cure rate and to prevent relapse.

  15. Rare manifestations of sirenomelia syndrome: a report of five cases.

    Science.gov (United States)

    Drossou-Agakidou, Vasiliki; Xatzisevastou-Loukidou, Chariklia; Soubasi, Vasiliki; Kostopoulou, Eyanthia; Laporda, Alexandra; Pantzaki, Afroditi; Agelidou, Stamatia; Kremenopoulos, Georgios

    2004-10-01

    Five cases of sirenomelia presented with rare manifestations are discussed. Three neonates were born alive and died within 2 to 12 hours after birth. One case was the offspring of a triple in vitro fertilization pregnancy with history of early intrauterine death of one of the triplets. The main features included fusion of lower extremities (five of five), renal agenesis (three of five), polycystic renal dysplasia (two of five), anal atresia with large bowel hypoplasia (three of five), pulmonary hypoplasia (four of five), and single umbilical artery (five of five). Other features that have only rarely been associated with sirenomelia included concurrence of congenital heart disease and neuroblastoma, gallbladder agenesis, and upper extremity defects.

  16. Synovial Sarcoma of the Buccal Mucosa: A Rare Case Report

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    Kumar T. S. Mahesh

    2013-01-01

    Full Text Available Synovial sarcoma (SS is a rare malignant neoplasm that arises most commonly in joint capsules and articular tendons, but its relationship to the synovium is not always obvious. Synovial sarcoma is a malignant soft tissue tumor representing 5.6% to 10% of all soft tissue sarcomas. They are termed SS because of their histologic resemblance to the synovium, but they rarely involve a synovial structure and are thought to arise from pluripotential mesenchymal cells. The tumor usually occurs in close association with tendon sheaths, bursae, and joint capsules, primarily in the para-articular regions of the extremities, with approximately 9% occurring in the head and neck region. Synovial sarcoma has been reported rarely in the oral cavity. We report a very rare case of Synovial sarcoma of the buccal mucosa in a 24-year-old male patient.

  17. Chondroma of Falx: Case Report of a Rare Condition

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    Shahryar Shahriarian

    2012-03-01

    Full Text Available Chondroma is a benign tumor which mostly occurs in extremities but also sometimes in brain. Most intracranial chondromas arise from skull base, but chondroma of falx origin is a rare circumstance. Indeed, the intracranial chondromas rise from falx is mostly in relation with syndromic disorders such as Mafuccis syndrome or Olliers syndrome. Here, we reported a rare case of falxian intracranial chondroma in a young man who has normal physical examination and no signs of any syndromic disorder. The goal of this paper was to raise awareness about chondromas and suggest that chondroma be ruled out in any patient with masses arising from falx.

  18. Arterial tortuosity syndrome: An extremely rare disease presenting as a mimic of pulmonary sling

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    Amy Farkas, MD

    2018-02-01

    Full Text Available Pulmonary sling is the anatomic variant defined by the aberrant origin of the left pulmonary artery from the right pulmonary artery. This patient presented with a mimic of pulmonary sling as a result of an extremely rare condition, arterial tortuosity syndrome (ATS. The patient was first diagnosed with pulmonary sling on prenatal echocardiogram performed by cardiology. Computed tomography angiography of the chest obtained at birth to evaluate respiratory depression demonstrated ATS. The early detection of ATS has been demonstrated to improve patient outcome. This case provides an overview of the typical imaging features of ATS to aid radiologists in making this uncommon diagnosis.

  19. Higher order magnetic modulation structures in rare earth metal, alloys and compounds under extreme conditions

    International Nuclear Information System (INIS)

    Kawano, S.

    2003-01-01

    Magnetic materials consisting of rare earth ions form modulation structures such as a helical or sinusoidal structure caused by the oscillating magnetic interaction between rare earth ions due to RKKY magnetic interaction. These modulation structures, in some cases, develop further to higher order modulation structures by additional modulations caused by higher order crystalline electric field, magnetic interactions such as spin-lattice interaction, external magnetic field and pressure. The higher order modulation structures are observed in a spin-slip structure or a helifan structure in Ho, and a tilt helix structure in a TbEr alloy. Paramagnetic ions originated from frustration generate many magnetic phases under applied external magnetic field. KUR neutron diffraction groups have performed the development and adjustment of high-pressure instruments and external magnetic fields for neutron diffraction spectrometers. The studies of 'neutron diffraction under extreme conditions' by the seven groups are described in this report. (Y. Kazumata)

  20. A RARE CASE OF ALKAPTONURIA: CASE REPORT

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    Dharmambal

    2016-01-01

    Full Text Available INTRODUCTION Alkaptonuria is a rare metabolic autosomal recessive disorder due to Homogentisic acid oxidase deficiency. There is generalized deposition of oxidized HGA in tissues mainly in fibrous and cartilaginous tissues. The disease is generally characterized by dark urine, ochronosis, and arthritis. A 50-year-old female attended the hospital with severe Joint Pain, Itching, Parasitophobia, Difficulty in hearing, Lack of clear vision and Discoloration of urine for different durations ranging from 6 months to 10 years. Alkaptonuria was suspected and accordingly detailed clinical examination and investigations were carried out. Biochemical analysis was positive for Alkaptonuria, X-ray was supportive for the diagnosis, and Histopathological examination revealed alkaptonuric pathology. The treatment was initiated with Ascorbic acid and protein restricted diet with symptomatic management and being followed up.

  1. A rare case of post-splenectomy gastric volvulus managed by laparoscopic anterior gastropexy

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    Rahul Amreesh Gupta

    2017-01-01

    Full Text Available We report an extremely rare case of recurrent gastric volvulus after open splenectomy for hereditary spherocytosis. The initial episode was managed by endoscopic derotation. Later, for recurrent symptoms, she was successfully managed by laparoscopic anterior gastropexy.

  2. Intussusception due to a cecal duplication cyst: a rare cause of acute abdomen. Case report.

    Science.gov (United States)

    Corroppolo, M; Zampieri, N; Erculiani, E; Cecchetto, M; Camoglio, F S

    2007-01-01

    Duplications of the alimentary tract are rare congenital anomalies. The ileum is the most common site, whereas rectal, duodenal, gastric and cecal duplications are extremely rare. Duplication cysts of the cecum, in a neonate, are even rarer, with only 19 cases reported in medical literature to date. We report a case of intestinal intussusception due to a cecal duplication cyst.

  3. Focal dermal hypoplasia: A rare case report

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    Sahana M Srinivas

    2015-01-01

    Full Text Available Focal dermal hypoplasia (Goltz syndrome is a rare genetic multisystem disorder primarily involving the skin, skeletal system, eyes, and face. We report the case of an eight-month-old female child who presented with multiple hypopigmented atrophic macules along the lines of blaschko, skeletal anomalies, umbilical hernia, developmental delay, hypoplastic nails, syndactyly, and lobster claw deformity characteristic of Goltz syndrome.

  4. Pediatric gliosarcoma with fibrosarcomatous differentiation: Report of a rare case

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    Shantha Ravisankar

    2012-01-01

    Full Text Available Gliosarcoma is a rare variant of glioblastoma with a biphasic pattern showing glial and mesenchymal differentiation. It is seen in adults during their fifth to sixth decades of life and is extremely rare in children. We report a case of primary gliosarcoma with fibrosarcomatous differentiation in an 11-year-old boy presenting with headache and vomiting. Imaging showed a contrast-enhancing isodense space-occupying lesion with areas of calcification in the right temporoparietal cortex. A total excision was done and, on histopathologic examination, a differential diagnostic consideration of gliosarcoma and teratoma with malignant transformation was made. After immunohistochemical analysis, a final diagnosis of gliosarcoma with fibrosarcomatous differentiation was then made. Primary gliosarcoma is a very rare tumor in children with a poor prognosis.

  5. Odontoameloblastoma: A rare case with unusual presentation

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    Supreet Jain

    2016-01-01

    Full Text Available The odontoameloblastoma (OA, also known as ameloblastic odontoma, is a very rare odontogenic tumor that is similar to ameloblastoma in its locally aggressive behavior. OA includes odontogenic ectomesenchyme in addition to odontogenic epithelium that resembles an ameloblastoma both in structure and in behavior. Its clinical presentation, however, often mimics the more innocuous odontoma, and hence, the recognition of its aggressive nature is commonly only ascertained after its histopathologic diagnosis following enucleation. This paper presents a rare case of OA with unusual clinical and radiological features.

  6. Myxofibrosarcoma of maxilla: A case report of rare entity

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    Venkateswarlu Nallapu

    2015-01-01

    Full Text Available Myxofibrosarcoma is one of the most common sarcomas in the extremities of elderly patients that rarely affect the oral cavity. In this article we describe a case of primary myxofibrosarcoma of maxilla in a 19-year-old boy. Microscopically the tumor showed loosely arranged spindle-shaped tumor cells with mild pleomorphic spindle-shaped nuclei, some with minute nucleoli in a fibromyxoid stroma. The cells were strongly immunoreactive for vimentin and negative for CD-68, S-100 protein and desmin. Based on clinical, histological and immunohistochemical findings, a final diagnosis of myxofibrosarcoma was established. It is an unusual aggressive variant that frequently recurs and metastasizes, reinforcing the importance of its correct diagnosis. We report a rare case of myxofibrosarcoma in maxilla, with emphasis on immunohistochemical findings.

  7. Intraosseous adenoid cystic carcinoma of maxilla: A rare case report

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    Prasannasrinivas Suresh Deshpande

    2013-01-01

    Full Text Available Adenoid cystic carcinoma (ACC accounts for approximately 6-10% of all salivary gland tumors. Palatal minor salivary glands, parotid, and sub-mandibular glands are usually affected. Rarely, these lesions arising intraosseously have been reported. Mandible is commonly involved than maxilla. The present case is a giant ACC involving the right maxilla. A thorough clinical and radiographic evaluation was performed to assess the involvement of surrounding vital structures along with a meticulous metastatic work-up. Computed tomography showed a giant lesion in maxilla encroaching the left nasal fossa, antrum, buccal space, and oral cavity. No metastasis was noted. Histological evaluation from multiple sites showed both cribriform and solid patterns. Radiotherapy was given as patient did not comply for surgery. Though central ACC is extremely rare, especially in maxilla, it should be included in the differentials for lesions in maxilla. A prompt diagnosis with treatment and long-term follow-up is advised in such cases.

  8. Florid Cementoosseous Dysplasia: A Rare Case Report

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    Mehmet Fatih Şentürk

    2013-01-01

    Full Text Available Florid cementoosseous dysplasia (FCOD is a rare, benign, fibroosseous, and multifocal dysplastic lesion of the jaw that consists of cellular fibrous connective tissue with bone and cementum-like tissue. FCOD is most commonly found in middle-aged black women, is generally asymptomatic, and is usually detected during radiological examination. FCOD associated with multiple impacted teeth and bone expansion is a very rare phenomenon, and there are only a few familial cases reported in the literature. In this report, a 35-year-old male Turkish patient is presented who was diagnosed with nonfamilial FCOD from clinical, radiological, and histopathological findings. To our knowledge this is the first case of the nonfamilial FCOD with this many impacted teeth and severely expanded bones.

  9. Galactosemia: a rare case in pediatric practice

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    S. Ya. Volgina

    2015-01-01

    Full Text Available Galactosemia is a rare life-threatening inherited autosomal recessive disease, the differential diagnosis of which has been very difficult up to date, especially if there are no results of neonatal screening for some reasons. The paper describes a clinical case of a 10-day patient with type I classic galactosemia and reflects the importance of a timely diagnostic search and switching him to lactose-free formulas.

  10. Unilateral pure trigeminal motor nerve neuropathy: A rare case report

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    Nishant K Srivastava

    2014-01-01

    Full Text Available Unilateral pure trigeminal motor nerve neuropathy is an extremely rare and unique condition, characterized by atrophy of the muscles, innervated by the motor branch of the trigeminal nerve. We report such a case in a 25-year-old male patient. The diagnosis was made on the basis of clinical and radiological examinations. Magnetic Resonance Imaging (MRI proved to be the key for establishing the diagnosis, which showed atrophy and fatty infiltration over the affected side of the muscles of mastication. We were unable to establish the cause of the condition even after performing a brain MRI.

  11. Ossifying fibroma in the sinonasal tract: a rare case report.

    Science.gov (United States)

    Charavarty, Shekhar; Datta, Debashish; Boro, Esha; Dutta, Bijita

    2014-08-01

    Ossifying fibroma involving the sinonasal tract is an extremely rare occurrence. We are reporting a case of ossifying fibroma in the left maxillary sinus in a 19-year-old female who presented with a mass in the left cheek and nasal obstruction. On examination, the swelling in the left cheek seemed to originate from the left maxilla and was smooth and bony hard. Patient underwent detailed radiological and image guided cytopathological investigations. Depending on the results of these tests complete surgical excision of the mass was done. Histopathological evaluation of the excised mass revealed it to be an Ossifying Fibroma.

  12. A rare case of complete second arch branchial fistula in a 7-year-old child

    OpenAIRE

    Shankar, Venkateswara Gomathi; Babu, Thirunavukkarasu Arun; Swami, Hartimath Basavanand

    2012-01-01

    Branchial fistulae are formed due to the abnormal persistence of the embryonic branchial clefts. Complete branchial fistula with internal and external opening is extremely rare. We report a rare case of complete second arch branchial fistulae in a 7-year-old boy, which was confirmed by a fistulogram. The tract was completely excised and the patient was successfully treated.

  13. A rare case of complete second arch branchial fistula in a 7-year-old child.

    Science.gov (United States)

    Shankar, Venkateswara Gomathi; Babu, Thirunavukkarasu Arun; Swami, Hartimath Basavanand

    2012-07-01

    Branchial fistulae are formed due to the abnormal persistence of the embryonic branchial clefts. Complete branchial fistula with internal and external opening is extremely rare. We report a rare case of complete second arch branchial fistulae in a 7-year-old boy, which was confirmed by a fistulogram. The tract was completely excised and the patient was successfully treated.

  14. Oral lymphangioma: A rare case report

    Directory of Open Access Journals (Sweden)

    Harsha Bhayya

    2015-01-01

    Full Text Available Lymphangiomas are benign hamartomatous tumors of the lymphatic channels which present as developmental malformations arising from sequestration of lymphatic tissue that do not communicate with the rest of the lymphatic channels. Lymphatic vessels are filled with a clear protein-rich fluid containing few lymph cells. It can also occur in association with hemangioma. The onset of lymphangiomas are either at birth (60% to 70% or up to two years of age (90% and rare in adults. Lymphangiomas have marked predilection for the head and neck region (50-70%. The most common location in the mouth is the dorsum of tongue, followed by lips, buccal mucosa, soft palate, and floor of the mouth. On tongue, they may present as a localized or a diffused growth which may enlarge to cause macroglossia, impaired speech and difficulty in mastication. Herewith, we present a rare case of lymphangioma of tongue leading to macroglossia in a 8-year-old boy.

  15. A Rare Case of Petrified Ear

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    Kathryn E. Buikema

    2012-01-01

    Full Text Available Calcification or ossification of the auricle, also referred to as petrified ear, is a rare diagnosis in dermatology. In medical literature, it has most often been attributed to trauma, hypothermia and frostbite, or hypercalcemia secondary to a metabolic or endocrine disorder, such as Addison's disease. Here, we report the clinical and radiologic findings of a 79-year-old African American male whose unilateral petrified auricle was an incidental finding. He had a preceding history of hyperparathyroidism and subsequent hypercalcemia treated with a subtotal parathyroidectomy three years prior to presentation. In addition to laboratory analysis, a history and physical examination was performed which revealed no other signs of hypercalcemia. Radiologic studies demonstrated partial ossification of the external auricular cartilage on the left side. The patient was diagnosed with the rare occurrence of a petrified ear. In light of this case, we provide a discussion concerning the possible etiologies of this diagnosis including appropriate patient evaluation and possible treatment recommendations.

  16. A Rare Case of Malignant Glomus Tumor of the Esophagus

    Directory of Open Access Journals (Sweden)

    Gurvinder Singh Bali

    2013-01-01

    Full Text Available Glomus tumors are rare neoplasms that usually occur on the hands in a subungual location, or sometimes in palms, wrists or soles of the feet. They are described as purple/pink tiny painful lesions with a triad of pain, local point tenderness, and cold hypersensitivity. They are almost always benign, but rare malignant variants have been reported. They have also been reported to be present at unusual locations, like the lung, stomach, or liver. Gastrointestinal glomus tumors are extremely rare tumors and very few cases have been reported in the literature. Most that have been reported were usually benign in nature. A rare esophageal glomangioma, mimicking a papilloma, was reported in 2006. We report a case of glomangiosarcoma (malignant glomus tumor in a 49-year-old female, who presented with symptoms of dysphagia including some spasm and hoarseness and subjective unintentional weight loss. On endoscopic exam, she was found to have a distal esophageal mass with malignant features. Radiologically, the mass had a size of about 8 cm on the CT scan without evidence of metastases. Pathology and immunostaining of the biopsy showed features resembling a malignant glomus tumor. She underwent an endoscopic and laparoscopic staging of the tumor along with ultrasound. Based on the laparoscopic findings, which were consistent with the preoperative diagnosis, she was scheduled for an esophagectomy. Histopathology and immunophenotypic features of the excised mass were consistent with a diagnosis of malignant glomus tumor.

  17. Spontaneous Superior Mesenteric Artery Branch Pseudoaneurysm: A Rare Case Report

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    Mina Guirgis

    Full Text Available : Background: Visceral arterial pseudoaneurysms (VAPAs are rare vascular entities with serious consequences. Traditionally, they are associated with trauma, infection, and inflammatory disease, or they can arise as a post-operative complication. Report: An 87 year old man presented with abdominal pain and was found to have a spontaneous VAPA on a computed tomography angiogram. Serial imaging 4 months previously had demonstrated no aneurysm. Between scans, warfarin was changed to apixaban for aortic valve replacement, but he had no other changes to any other medications. He required urgent endovascular coiling of the pseudoaneurysm, with satisfactory recovery and outcome. Discussion: VAPAs are extremely rare, with splenic artery VAPAs the most commonly reported. Regardless, fewer than 250 cases of splenic artery pseudoaneurysm have been reported. Superior mesenteric artery (SMA pseudoaneurysms are the rarest type of VAPAs. Early identification and urgent treatment are warranted because of the associated high mortality risk, with a 50% risk of rupture in any given VAPA. Treatment options range from open operation to endoscopic and endovascular procedures. Apixaban has been proposed to contribute to pseudoaneurysm formation by slow and continuous bleeding that results in the formation of the pseudoaneurysm. Conclusions: Spontaneous VAPAs are extremely rare and this is the first time a VAPA has been associated with the novel oral anticoagulant “apixaban”. Urgent management of any VAPAs is important because of the high risk of rupture and potential life threatening haemorrhage. Keywords: Visceral arterial pseudoaneurysm, Superior mesenteric artery, Apixaban, Coils

  18. Facial Localization of Malignant Chondroid Syringoma: A Rare Case Report

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    Deniz Tural

    2013-01-01

    Full Text Available First described by Hirsch and Helwig in 1961, chondroid syringomas (CSs are rare, benign tumors of the skin arising from the eccrine sweat glands with tumor differentiation in the epithelial and mesenchymal tissues. They most commonly occur in the head and neck, although they may be also found in the axilla, trunk, limbs, and genitalia. The incidence of CS is <0.01% of all primary skin tumors. Malingnant chondroid syringomas (MCS, which are also called malignant mixed tumors of the skin, are extremely uncommon. MCSs commonly involve the limbs and rarely head and neck. In this article, we present a case of malignant chondroid syringoma localized in the face at the left nasolabial region in the light of literature review.

  19. Retroperitoneal Malignant Peripheral Nerve Sheath Tumour: A Rare Case Report.

    Science.gov (United States)

    Deger, Ayse Nur; Bayar, Mehmet Akif; Caydere, Muzaffer; Deger, Hakki; Tayfur, Mahir

    2015-09-01

    Malignant nerve sheath tumours (MPNST) are rare neoplasias and retroperitoneal cases are fairly rare and clinically difficult to be detected, but they are very agressive neoplasias. MPNST are frequently seen in head, neck and upper extremities. In patients with NF1; MPNST, a poor-prognostic lesion, may result from a malignant degeneration of a former plexiform neurofibroma. It is necessary to be aware of a potential malignancy in patients diagnosed with plexiform neurofibroma. We present a 21-year-old female with a diagnosis of MPNST. The patient was admited to the hospital because of a tumour in the subcutaneous region on her left buttock. The surgeon's clinical diagnosis was lipoma. After the pathological examination of biopsy specimen, the lesion was identified as "plexiform neurofibroma" and then the patient was diagnosed with Neurofibromatosis Type 1 (NF1). Simultaneously, another mass on the retroperitoneal region was identified as malignant peripheral nerve sheath tumour (MPNST).

  20. A Rare Case of Metastatic Choriocarcinoma of Lung Origin

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    Parth Rali

    2017-01-01

    Full Text Available Choriocarcinoma is part of the spectrum of gestational trophoblastic disease that occurs in women of reproductive age. Although the most common metastatic site of choriocarcinoma is the lung, primary pulmonary choriocarcinoma is rare. To diagnose primary pulmonary choriocarcinoma, the patient should have no previous gynecologic malignancy, have elevated human chorionic gonadotropin, and have pathological confirmation of the disease excluding gonadal primary site of the tumor. Due to the paucity of data, there are no guidelines for treatment. Prognosis of this malignancy is extremely poor. We report a rare case of metastatic primary lung choriocarcinoma in a 69-year-old postmenopausal woman who was treated with combination of surgery, chemotherapy, and radiation. The patient had a good outcome and is doing well after 1-year follow-up.

  1. HARLEQUIN ICTHYOSIS: A RARE CASE REPORT

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    Pradipprava Paria

    2016-01-01

    Full Text Available Harlequin ichthyosis is the most severe form of congenital ichthyosis. It is a rare autosomal recessive disorder (1:300,000. The vast majority of affected individuals are due to mutation in the ABCA12 gene, which cause a deficiency of the epidermal lipid transporter, resulting in hyperkeratosis and abnormal barrier function of skin. Infants are very susceptible to metabolic abnormalitites and infections. They usually do not survive for very long. we report here a case of a new born with harlequin ichthyosis of consaguinious parentage who had a history of similar birth previously.

  2. Seckel syndrome: A rare case report

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    Rinky Sisodia

    2014-01-01

    Full Text Available Seckel syndrome (SS is a rare, autosomal recessive syndrome; characterized by severe intrauterine and postnatal growth retardation, microcephaly, mental retardation, and typical facial appearance with beaklike protrusion of the midface (bird headed. In addition to the characteristic craniofacial dysmorphism and skeletal defects, abnormalities have been described in the cardiovascular, hematopoietic, endocrine, gastrointestinal, and central nervous systems. Usually such patients have poor psychomotor development. This case report presents an 8-year-old child with SS born to parents, exposed in Bhopal gas disaster.

  3. Benedikt's Syndrome: A Rare Case Report

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    Aslı Aksoy Gundogdu

    2017-08-01

    Full Text Available Benedikt syndrome is a rare midbrain syndrome which is associated with the damage of the median mesencephalic tegmentum. The most common etiology of this syndrome is ischemic stroke. The occlusion of the posterior cerebral artery or the paramedian branches of the basilar artery results with the ischemia of this midbrain territory. Ipsilateral occulomotor cranial nerve palsy, contralateral hemiparesis, hemihypoesthesia, hemiataxia and korea or tremor are the clinical symptoms of this syndrome. In this article, we reported a case of Benedikt syndrome with an etiologic cause of cardioembolic stroke, who was diagnosed by the neurological examination and neuroradiological findings.

  4. Xeroderma pigmentosum: A rare case report with review of literature

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    B Anand

    2012-01-01

    Full Text Available Xeroderma pigmentosum, or XP, is an autosomal recessive genetic disorder in which the ability to repair DNA damage caused by ultraviolet (UV light is deficient. In extreme cases, all exposure to sunlight must be forbidden, no matter how small. As such, individuals with the disease are often colloquially referred to as ′Children of the Night′. Mutations in XP genes that regulate nucleotide excision repair, not only predispose persons with xeroderma pigmentosum to multiple malignancies, but also promote premature cutaneous and ocular ageing, and in some cases promote progressive neurodegenerative changes. There is a great involvement of many parts of the body, especially head and neck. The oral manifestations are mainly related to the occurrence of malignant tumors in the lips, tongue and buccal mucosa. This paper reports a rare case of XP in a 40-year-old female presenting with dermatological, oral and ophthalmological involvement.

  5. A Rare Case of Idiopathic Plastic Bronchitis

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    Mohammed Raoufi

    2017-01-01

    Full Text Available Plastic bronchitis is a rare disorder characterized by formation of large, branching bronchial casts, which are often expectorated. We present an interesting case of a 35-year-old woman who presented for evaluation of a chronic cough productive of voluminous secretions. Clinical and radiological examination confirmed a total left lung atelectasis without any pathological mediastinal node. Flexible bronchoscopy demonstrated tenacious, thick, and sticky whitish secretions blocking the left stem bronchus. This material was extracted, and inspection demonstrated a bronchial cast, whose pathological analysis revealed necrotic epithelial cells, some eosinophils, and Charcot-Leyden crystals. Two days after bronchoscopy, the patient rejected more bronchial casts, and dyspnea improved. Control of chest x-ray revealed complete left lung aeration and the diagnosis of idiopathic plastic bronchitis was obtained. This article shows the interest in clinical practice to evoke the diagnosis of plastic bronchitis in front of a productive chronic cough. Our case illustrates a rare clinical presentation represented by an atelectasis of an entire lung.

  6. Familial gingival fibromatosis: A rare case report

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    Shweta Sharma

    2012-01-01

    Full Text Available Hereditary gingival fibromatosis is a rare condition that can occur as an isolated disease or as part of a syndrome or chromosomal abnormality. In severe cases, the gingival enlargement may cover the crowns of teeth and cause severe functional and aesthetic concerns. Here, we present a case of an 8-year-old girl with severe enlargement of gums in maxilla and mandible. Both deciduous and permanent teeth were not erupted in the oral cavity at all. Mutation in the Son-of-Sevenless (SOS-1 gene has been associated with the disease. The diagnosis was made based on clinical examination and family history. Surgical removal of the hyperplastic tissue was performed under general anesthesia.

  7. Using Extreme Phenotype Sampling to Identify the Rare Causal Variants of Quantitative Traits in Association Studies

    OpenAIRE

    Li, Dalin; Lewinger, Juan Pablo; Gauderman, William J.; Murcray, Cassandra Elizabeth; Conti, David

    2011-01-01

    Variants identified in recent genome-wide association studies based on the common-disease common-variant hypothesis are far from fully explaining the hereditability of complex traits. Rare variants may, in part, explain some of the missing hereditability. Here, we explored the advantage of the extreme phenotype sampling in rare-variant analysis and refined this design framework for future large-scale association studies on quantitative traits. We first proposed a power calculation approach fo...

  8. A rare case of thyroid metastasis from pancreatic adenocarcinoma.

    LENUS (Irish Health Repository)

    Kelly, Michael E

    2012-02-01

    CONTEXT: Thyroid metastasis from pancreatic adenocarcinoma is extremely rare, with only two previous cases in the literature. We report a case of pancreatic adenocarcinoma metastasising to the thyroid. We review the incidence, diagnosis, and management of this rare occurrence. CASE REPORT: A 38-year-old man with a synchronous 6-month history of thyroid swelling, presented with epigastric pain and signs of obstructive jaundice. He was investigated by abdominal computerised tomography and endoscopic retrograde cholangiopancreatography. The diagnosis of pancreatic neoplasm was made. His thyroid neoplasm was investigated at another tertiary centre and thought to be a papillary neoplasm. He underwent a pancreaticoduodenectomy and recovered well post-operatively. Eight weeks later he had a total thyroidectomy. Histology confirmed that the thyroid mass was both morphologically and immunophenotypically similar to the pancreatic neoplasm. CONCLUSION: This case demonstrates the importance of a full investigation when a patient with suspected neoplastic history presents with a thyroid nodule. We outline the crucial role that immunohistochemistry plays in detecting and classifying primary and secondary thyroid neoplasms. The detection of a solitary thyroid metastasis from pancreatic adenocarcinoma may indicate a poor prognosis, and it is debatable whether resection of the primary should be undertaken when it presents with a solitary metastasis.

  9. Urachal adenocarcinoma: a rare case report

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    Bo Bao

    2017-03-01

    Full Text Available Urachal carcinoma is a rare and aggressive form of bladder cancer involving the urachus, a fibrous remnant of the allantois that extends from the bladder to the umbilicus. We report this case of a 49-year-old women with primary urachal adenocarcinoma treated with partial cystectomy who relapsed 5 years after surgery with lung metastases. This patient with unremarkable medical history presented with abdominal discomfort and a palpable pelvic mass. Follow-up imaging reveals a large mass on the dome of the bladder extending from the urachus. Subsequent ultrasound-guided biopsy result was suggestive of an urachal mucinous adenocarcinoma. The patient was treated surgically with a partial cystectomy.

  10. Prune Belly syndrome: A rare case report.

    Science.gov (United States)

    Samal, Sunil Kumar; Rathod, Setu

    2015-01-01

    Prune Belly syndrome (PBS) is a rare congenital anomaly of uncertain etiology almost exclusive to males. We report a case of term male baby born to a 39-year-old grand multipara with previous four normal vaginal births. There was no history of genetic or congenital anomaly in her family. Examination of the baby revealed hypotonia, deficient abdominal muscle, cryptorchidism, palpable kidney, and bladder. Ultrasound examination of the abdomen revealed bilateral gross hydronephrosis and megaureter. Provisional diagnosis of PBS was made and the baby was admitted in neonatal intensive care units for further management. Routine antenatal care with ultrasonography will help in detecting renal anomalies, which can be followed postnatally. Early diagnosis of this syndrome and determining its optimal treatment are very important in helping to avoid its fatal course.

  11. A rare case of Moebius sequence

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    Abhishek Kulkarni

    2012-01-01

    Full Text Available We report a case of an 18-year-old male who presented with watering and inability to close the left eye completely since 6 months and inability to move both eyes outward and to close the mouth since childhood. Ocular, facial, and systemic examination revealed that the patient had bilateral complete lateral rectus and bilateral incomplete medial rectus palsy, left-sided facial nerve paralysis, thickening of lower lip and inability to close the mouth, along with other common musculoskeletal abnormalities. This is a typical presentation of Moebius syndrome which is a very rare congenital neurological disorder characterized by bilateral facial and abducens nerve paralysis. This patient had bilateral incomplete medial rectus palsy which is suggestive of the presence of horizontal gaze palsy or occulomotor nerve involvement as a component of Moebius sequence.

  12. A RARE CASE OF HUGE CERVICAL FIBROID

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    Tushar Tatyaba Palve

    2017-07-01

    Full Text Available PRESENTATION OF CASE Leiomyomas are the most common tumor of uterus and the female pelvis. It is impossible to determine true incidence, all though frequently quoted incidence of 50% seems reasonable at post mortem examination1 . However only 1- 2% of it are confined to cervix. Cervical leiomyomata are mostly single and are subserous or interstitial in origin. Generally these tumours presents with retention of urine, constipation, sensation of something coming out of vagina, & rarely at times present with abdominal mass which may mimic ovarian cancer. 47 year old unmarried nulligravida with complaints of severe dysmenorrhagia since one year and lump in abdomen which has grown over period of one year. There was no bowel and bladder complaints, On physical examination pallor present, on abdominal examination 26-28 week firm solid mass arising from pelvis with restricted mobility was present, Clinically no ascites, patient was investigated on OPD basis.

  13. Congenital epulides: A rare case report

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    Saurabh Kumar

    2014-01-01

    Full Text Available Congenital epulis (CE or "Granular cell epulis" also previously termed as "Neumann′s tumor" is a benign growth arising from the mucosa of the gingiva, typically seen as a mass protruding from the infant′s mouth, often interfering with respiration and feeding. These tumors generally present as a single mass arising from the upper alveolus. We report a rare case of two congenital epulides arising from the maxillary and mandibular alveolus in a day old female infant, which was surgically excised, allowing for early initiation of breast feeding. The tumor cells stained negative for S100 protein hence differentiating from other granular cell tumors. The clinical presentation, differential diagnosis in regard to the various neonatal oral swellings and the management of CE has been discussed.

  14. Isolated pulmonary candidiasis in a patient with diabetes mellitus: A rare case report

    OpenAIRE

    Hakamifard, Atousa; Khorvash, Farzin; Raisi, Arash

    2016-01-01

    Fungal infections are as a cause of morbidity and mortality in immunocompromise patients. Because the respiratory tract is colonized with Candida, the presence of this agent in respiratory specimens makes the diagnosis of Candida pneumonia problematic. Candida pneumonia is a rare infection, and the majority of cases are secondary to hematogenous dissemination. Furthermore isolated Candida pneumonia originating from endotracheal inoculation is an extremely rare entity. We describe a case of is...

  15. Onychomycosis: A Rare Presentation of Fungal Urinary Tract Infection in an Extremely Preterm Neonate

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    Shilpa Kalane

    2015-06-01

    Full Text Available Onychomycosis refers to nail infections, caused by fungi including yeasts and non-dermatophyte moulds. One or several toenails or fingernails (seldom all may be involved in this condition. Many cases of fingernail onychomycosis are due to yeasts. Fungal infection has emerged as an important cause of neonatal infection, associated with significant morbidity and mortality, especially in very low birth weight (< 1500 g and extremely low birth weight (< 1000 g infants. Herein, we report a case of a 24-day-old male infant, who presented with onychomycosis on the left ring fingernail, associated with fungal urinary tract infection (UTI. The evaluation of nails helped us detect fungal UTI. To date, there have been no reports suggesting onychomycosis as a presentation of fungal UTI. We could not find the association between onychomycosis and neonatal fungal UTI. Hence, retrospectively, it can be said that onychomycosis was a presentation of fungal UTI. Further studies are required to evaluate the etiology and treatment of neonatal onychomycosis. Moreover, dermatologists should pay particular attention to this rare event.

  16. Urethral triplication with meatuses terminating on the penis: a rare case presenting with bifid urinary stream

    International Nuclear Information System (INIS)

    Hirselj, Daniel A.; Jayanthi, Venkata R.; Lowe, Gregory K.

    2009-01-01

    Urethral duplication is an uncommon clinical finding, and classification systems have been described to explain the clinical findings. Urethral triplication is an extremely uncommon diagnosis, with few published case reports. We present the rare case of an infant noted to have a bifid urinary stream and found to have urethral triplication on voiding cystourethrography. (orig.)

  17. Primary Small Cell Neuroendocrine Carcinoma of Vagina: A Rare Case Report

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    Jignasa N. Bhalodia

    2011-01-01

    Full Text Available Primary small cell neuroendocrine carcinoma of vagina is an extremely rare disease. There have been only 26 previously reported cases in literature. Here, we report a case of primary small cell neuroendocrine carcinoma of vagina. Immunohistochemistry (IHC showed tumor cells positive for synaptophysin, chromogranin, and neuron-specific enolase (NSE.

  18. Urethral triplication with meatuses terminating on the penis: a rare case presenting with bifid urinary stream

    Energy Technology Data Exchange (ETDEWEB)

    Hirselj, Daniel A.; Jayanthi, Venkata R. [Nationwide Children' s Hospital, Department of Pediatric Urology, Columbus, OH (United States); Lowe, Gregory K. [The Ohio State University Medical Center, Department of Urology, Columbus, OH (United States)

    2009-11-15

    Urethral duplication is an uncommon clinical finding, and classification systems have been described to explain the clinical findings. Urethral triplication is an extremely uncommon diagnosis, with few published case reports. We present the rare case of an infant noted to have a bifid urinary stream and found to have urethral triplication on voiding cystourethrography. (orig.)

  19. PRIMARY MULTILOCULAR HYDATID CYST OF NECK : A RARE CASE REPORT

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    Deepak Ramraj

    2015-02-01

    Full Text Available Hydatid disease, also known as echinococcosis or hydatidosis , is an infectious disease caused by Echinococcus. Echinococcus granulosus is the most common Echinococcus species affecting human beings. It may affect any organ and tissue in the body, in particular the liver and lung. Musculoskeletal or soft tissue hyda tidosis accounts for about 0.5% 5% of all echinococcal infections in endemic areas, and is almost always secondary to the hepatic or pulmonary disease. Even in regions where echinococcosis is endemic, hydatidosis of cervicofacial region is extremely rare. Herein, we present exceptionally rare case in a 55 year old female with an unusual localization of primary multilocular hydatid cyst in the right supraclavicular region of the neck. A high index of suspicion is required to diagnose hydatid cyst in rare loc ations like this. Hydatid cyst should be considered in differential diagnosis of benign swellings of head and neck region, so that it can be managed during surgery to prevent acute anaphylaxis

  20. Detecting rare variants in case-parents association studies.

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    Kuang-Fu Cheng

    Full Text Available Despite the success of genome-wide association studies (GWASs in detecting common variants (minor allele frequency ≥0.05 many suggested that rare variants also contribute to the genetic architecture of diseases. Recently, researchers demonstrated that rare variants can show a strong stratification which may not be corrected by using existing methods. In this paper, we focus on a case-parents study and consider methods for testing group-wise association between multiple rare (and common variants in a gene region and a disease. All tests depend on the numbers of transmitted mutant alleles from parents to their diseased children across variants and hence they are robust to the effect of population stratification. We use extensive simulation studies to compare the performance of four competing tests: the largest single-variant transmission disequilibrium test (TDT, multivariable test, combined TDT, and a likelihood ratio test based on a random-effects model. We find that the likelihood ratio test is most powerful in a wide range of settings and there is no negative impact to its power performance when common variants are also included in the analysis. If deleterious and protective variants are simultaneously analyzed, the likelihood ratio test was generally insensitive to the effect directionality, unless the effects are extremely inconsistent in one direction.

  1. Rare case of nephrotic syndrome: Schimke syndrome.

    Science.gov (United States)

    Pedrosa, Anna Kelly Krislane de Vasconcelos; Torres, Luiz Fernando Oliveira; Silva, Ana Corina Brainer Amorim da; Dantas, Adrianna Barros Leal; Zuntini, Káthia Liliane da Cunha Ribeiro; Aguiar, Lia Cordeiro Bastos

    2016-01-01

    Schimke syndrome corresponds to dysplasia of bone and immunity, associated with progressive renal disease secondary to nephrotic syndrome cortico-resistant, with possible other abnormalities such as hypothyroidism and blond marrow aplasia. It is a rare genetic disorder, with few reports in the literature. The most frequent renal involvement is nephrotic syndrome with focal segmental glomerulosclerosis and progressive renal failure. The objective of this study was to report a case of Schimke syndrome, diagnostic investigation and management of the case. Resumo A síndrome Schimke corresponde à displasia imuno-óssea, associada à doença renal progressiva secundária à síndrome nefrótica córtico-resistente, podendo haver outras anormalidades como hipotireoidismo e aplasia de medula óssea. Trata-se de uma patologia genética rara, com poucos relatos na literatura. O acometimento renal mais frequente é uma síndrome nefrótica por glomeruloesclerose segmentar e focal e falência renal progressiva. O objetivo deste estudo foi relatar um caso de síndrome de Schimke, investigação diagnóstica e condução do caso.

  2. A rare case of primary amenorrhea

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    Himadri Bal

    2016-01-01

    Full Text Available Crytomenorrhea arises usually due to the imperforate hymen, sometimes due to vaginal septum, rarely due to cervical agenesis. Here, we present a case of primary amenorrhea resulting from cervical agenesis in a 38-year-old woman. She presented with primary amenorrhea and cyclic lower abdominal pain. She had undergone some surgery 5 years back, details of which were not available. On examination secondary sexual characters were normal, per abdomen there was an 18 weeks size firm abdominopelvic mass. Local vaginal examination showed a blind vaginal pouch. A clinical diagnosis of hematometra due to transverse vaginal septum was made. However, magnetic resonance imaging pelvis suggested hematometra with cervical stenosis. The patient was taken up for examination under anaesthesia (EUA and exploratory laparotomy. On opening the abdomen uterus found to be enlarged with dense adhesions all around and signs of endometriosis. Extensive adhesiolysis revealed bilateral chocolate cysts of ovaries with hematosalpinges and peritubal adhesions. Hysterotomy and drainage of tarry contents were followed by an exploration of the uterine cavity. The lower pole ended blindly with no evidence of any cervix. Peroperative diagnosis of cervical agenesis leading to hematometra and endometriosis was made. A subtotal hysterectomy with bilateral salpingo-oophorectomy was done. Histopathological examination confirmed ovarian endometriosis and adenomyosis of uterus. Though reconstructive surgery for cervical dysgenesis has been successful in some cases, hysterectomy is generally recommended for cervical agenesis.

  3. Rare earth - no case for government intervention

    OpenAIRE

    Georg Zachmann

    2010-01-01

    China has officially restricted exports of rare earth for several years and announced this year it will further tighten exports. Rare earth is a group of 17 different metals, usually found clustered together. These metals have hundreds of different industry applications. For example, they are used in certain high capacity magnets, batteries and lasers. As the rare earth elements are used in sectors that are assumed to have an over-proportionate growth potential (eg. green-technology), policy ...

  4. Serous Carcinoma of the Uterine Cervix, an Extremely Rare Aggressive Entity: A Literature Review.

    Science.gov (United States)

    Jonska-Gmyrek, Joanna; Zolciak-Siwinska, Agnieszka; Gmyrek, Leszek; Michalski, Wojciech; Poniatowska, Grazyna; Fuksiewicz, Malgorzata; Wiechno, Pawel; Kucharz, Jakub; Kowalska, Maria; Kotowicz, Beata

    2018-01-23

    Serous carcinoma of the uterine cervix (USCC) is an extremely rare subtype. To establish the treatment strategy in patients with USCC is an important issue. MEDLINE (PubMed) was searched for all articles published after the first publication by Lurie et al. [Eur J Obstet Gynecol Reprod Biol 1991; 40: 79-81], reporting woman diagnosed with USCC. Because of limited numbers of studies on the topic of the study, we could not keep a restriction of eliminating smaller sample sizes. A search of PubMed demonstrated that 113 cases of USCC have been reported in the literature since the first publication. The current treatment modality adopted for early cervical cancer is hysterectomy with bilateral iliac-obturator lymphadenectomy and postoperative radiotherapy (RT) or radiochemotherapy (RT-CT) if risk factors for cervical carcinoma appear. The treatment strategy for locally advanced USCC is preoperative RT-CT or chemotherapy (CHTH) with the intention to treat the patient surgically. The treatment option for disseminated disease is CHTH with paclitaxel and carboplatin. Risk factors and a more advanced clinical stage of USCC have an impact on poor outcomes despite the use of standard treatment methods, adapted for cervical cancer. The outside-pelvic failures tend to seek effective systemic treatment. © 2018 S. Karger AG, Basel.

  5. A rare case of primary gastric plasmacytoma: An unforeseen surprise

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    Krishnamoorthy Navin

    2010-01-01

    Full Text Available Primary plasmacytoma of the gastrointestinal tract is a rare entity. We report a case of a primary gastric plasmacytoma in a 57-year-old man who presented with upper-gastrointestinal bleeding. Endoscopy showed a nodular gastric mass with central umblication. Histological examination of the gastrectomy specimen revealed a monoclonal lambda-chain extramedullary plasmacytoma. Further staging was found to be negative for multiple myeloma. As other more common pathologic processes at this site may also be endowed with numerous plasma cells, awareness of this entity and distinction using immunohistochemistry are extremely crucial. Because systemic disease ultimately develops in many patients with localized plasmacytoma, such patients should be followed closely for the appearance of clinical, biochemical, and roentgenologic evidence of multiple myeloma.

  6. Rare case of pancreatic cancer with leptomeningeal carcinomatosis

    Science.gov (United States)

    Yoo, In Kyung; Lee, Hong Sik; Kim, Chang Duk; Chun, Hoon Jai; Jeen, Yoon Tae; Keum, Bora; Kim, Eun Sun; Choi, Hyuk Soon; Lee, Jae Min; Kim, Seung Han; Nam, Seung Joo; Hyun, Jong Jin

    2015-01-01

    Leptomeningeal carcinomatosis occurs very rarely in patients with pancreatic cancer. Leptomeningeal carcinomatosis is characterized by multifocal seeding of the leptomeninges by malignant cells that originate from a solid tumor. To the best of our knowledge, brain metastasis from pancreatic cancer is extremely rare. Leptomeningeal carcinomatosis is estimated to occur in 3% to 8% of cases of solid tumors. The clinical manifestation usually involves neurological symptoms, including dizziness, headache, vomiting, nausea, and hemiparesis, symptoms similar to those of meningitis or brain tumors. Diagnostic methods for leptomeningeal carcinomatosis include brain magnetic resonance imaging and cerebrospinal fluid examination. Here, we describe a case of leptomeningeal carcinomatosis in which the primary tumor was later determined to be pancreatic cancer. Brain magnetic resonance imaging findings showed mild enhancement of the leptomeninges, and cerebrospinal fluid cytology was negative at first. However, after repeated spinal taps, atypical cells were observed on cerebrospinal fluid analysis and levels of tumor markers such as carbohydrate antigen 19-9 in cerebrospinal fluid were elevated. Abdominal computed tomography, performed to determine the presence of extracerebral tumors, revealed pancreatic cancer. Pancreatic cancer was confirmed histopathologically on examination of an endoscopic ultrasound-guided fine needle aspiration specimen. PMID:25624740

  7. A RARE CASE OF BILATERAL MICROSPHEREPHAKIA

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    Pandu

    2015-05-01

    Full Text Available Microspherophakia is rare bilateral congenital anamoly of the crystalline lens. The condition may be isolated , familial or it may be associated with systemic affections like Marfan's syndrome , Weil - Marchesani syndrome , hyperlysinemia and congenital rubella. Microspherophakia results in lenticular myopia , lens dislocation , usually inferiorly and inverse glaucoma. We present a case in a 8 year old child who presented with bilateral microspherophakia and anterior dislocation of lens of right eye. visual acuity in right eye was counting fingers close to face and i n left eye 6/60.IOP with perkins applanation tonometer was 30mmHg in right eye 22mmHg in left eye , cornea was hazy due to edema , anterior chamber was shallow in both eye patient was managed with emergency lens extraction of right eye and secondary ACIOL im plantation. Left eye was managed by laser peripheral iridotomy. IOP was within normal limits postoperatively in both eyes without any antiglaucoma medications. Postoperatively best corrected visual acuity in right was 6/18 and 6/9 in left eye.

  8. A Rare Case: Touraine Solente Gole Syndrome

    Directory of Open Access Journals (Sweden)

    Kamil Şahin

    2017-03-01

    Full Text Available Touraine-Solente-Gole syndrome, also known as pachydermoperiostosis, is transmitted as an autosomal recessive trait. It is characterized by enlargement of fingers and toes, pachyderma, excessive sweating, and pain. In this paper, we present a 9-year-old patient to attract attention to this rare disease. A 9-year-old female patient was brought to our outpatient clinics with sweating and enlargement of hands and feet. She was the fourth child born to consanguineous parents. Her 26-year-old elder sister also had the same symptoms. Her physical examination revealed clubbing of the hands, and thick and sweating fingers. Her test results were unremarkable. Hand x-ray revealed epiphyseal, and metaphyseal thickening of the hands, and periostal hyperosteosis. Pachydermoperiostosis usually begins in childhood, progresses till 20 years of age, then, ceases. Delayed closure of fontanelles, and patent ductus arteriosus may be symptoms of the disease. Patients with deletions and mutations in HPGD (15-hydroxy prostaglandin dehydrogenase gene (4q33-q34 demonstrate this phenotype. This syndrome is more frequent in females, and mimics rheumatic diseases. Ibuprofen therapy may be used for bone pain. Colchicine is the alternative treatment. In cases of excessive hand and feet sweating associated with clubbed fingers pachydermoperiostosis should be brought to mind.

  9. Gelofen Induced Hypersensitivity: A Rare Case Report

    Directory of Open Access Journals (Sweden)

    M. Nikkhah Rankohie

    2016-07-01

    Full Text Available Introduction: Non-steroidal anti-inflammatory drugs (NSAIDs are drugs commonly pre-scribed in dental practice for the management of pain and swelling. But, rarely hypersensitiv-ity reactions are reported. Case Report: A 28 year old woman underwent periodontal plastic surgery (gingival graft. Postoperative analgesics (400 mg Gelofen ,oral and antibiotics were administrated for the patient. Three hours after discharge of patient, she complained of redness, itching , rapid swelling of her eyes in 10 minutes, and watery eye discharge 1 hour after taking the drugs. She was treated with 8mg/2ml mg Dexamethasone IM at the dental department and with Hy-drocortisone 100mg/ml IM and antihistamine drugs at the hospital. Conclusion: There are no published protocols and sensitivity and specifity of skin pick testing and patch testing for Gelofen. So avoidance of re-exposure is the best management strategy. The use of Cox-2 specific medications would be a proper alternative for pain relief. (Sci J Hamadan Univ Med Sci 2016; 23 (2:179-183

  10. Simulation of the 23 July 2012 Extreme Space Weather Event: What if This Extremely Rare CME Was Earth Directed?

    Science.gov (United States)

    Ngwira, Chigomezyo M.; Pulkkinen, Antti; Mays, M. Leila; Kuznetsova, Maria M.; Galvin, A. B.; Simunac, Kristin; Baker, Daniel N.; Li, Xinlin; Zheng, Yihua; Glocer, Alex

    2013-01-01

    Extreme space weather events are known to cause adverse impacts on critical modern day technological infrastructure such as high-voltage electric power transmission grids. On 23 July 2012, NASA's Solar Terrestrial Relations Observatory-Ahead (STEREO-A) spacecraft observed in situ an extremely fast coronal mass ejection (CME) that traveled 0.96 astronomical units (approx. 1 AU) in about 19 h. Here we use the SpaceWeather Modeling Framework (SWMF) to perform a simulation of this rare CME.We consider STEREO-A in situ observations to represent the upstream L1 solar wind boundary conditions. The goal of this study is to examine what would have happened if this Rare-type CME was Earth-bound. Global SWMF-generated ground geomagnetic field perturbations are used to compute the simulated induced geoelectric field at specific ground-based active INTERMAGNET magnetometer sites. Simulation results show that while modeled global SYM-H index, a high-resolution equivalent of the Dst index, was comparable to previously observed severe geomagnetic storms such as the Halloween 2003 storm, the 23 July CME would have produced some of the largest geomagnetically induced electric fields, making it very geoeffective. These results have important practical applications for risk management of electrical power grids.

  11. A rare case of bilateral aspergillus endophthalmitis

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    Saurabh Gupta

    2015-12-01

    Full Text Available Aspergillus endophthalmitis is a devastating inflammatory condition of the intraocular cavities that may result in irreparable loss of vision and rapid destruction of the eye. Almost all cases in the literature have shown an identified source causing aspergillus endophthalmitis as a result of direct extension of disease. We present a rare case of bilateral aspergillus endophthalmitis. A 72-year-old woman with a history of diabetes mellitus, congenital Hirschsprung disease, and recent culture-positive candida pyelonephritis with hydronephrosis status post-surgical stent placement presented with difficulty opening her eyes. She complained of decreased vision (20/200 with pain and redness in both eyes – right worse then left. Examination demonstrated multiple white fungal balls in both retinas consistent with bilateral fungal endophthalmitis. Bilateral vitreous taps for cultures and staining were performed. Patient was given intravitreal injections of amphotericin B, vancomycin, ceftazidime, and started on oral fluconazole. Patient was scheduled for vitrectomy to decrease organism burden and to remove loculated areas of infection that would not respond to systemic antifungal agents. Four weeks after initial presentation, the fungal cultures revealed mold growth consistent with aspergillus. Patient was subsequently started on voriconazole and fluconazole was discontinued due to poor efficacy against aspergillus. Further workup was conducted to evaluate for the source of infection and seeding. Transthoracic cardiogram was unremarkable for any vegetation or valvular abnormalities. MRI of the orbits and sinuses did not reveal any mass lesions or bony destruction. CT of the chest was unremarkable for infection. Aspergillus endophthalmitis may occur because of one of these several mechanisms: hematogenous dissemination, direct inoculation by trauma, and contamination during surgery. Our patient's cause of bilateral endophthalmitis was through an

  12. Case Report: Multiple Myeloma with Gangrene of Extremities

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    N Gharahi

    2009-07-01

    Full Text Available Introduction: Multiple myeloma represents a malignant proliferation of plasma cells derived from a single clone and it results in bone pain or fracture, renal failure, susceptibility to infections, anemia and hypercalcemia. The hyper viscosity syndrome is rare. Cryoglobulins are immunoglubulins that precipitate at temperatures less than 37degrees Celsius. Monoclonal cryoglobulins are usually present along with a specific hematologic disorder and are often asymptomatic. We report a second case of multiple myeloma with gangrene of all four extremities. Case: The Patient was a 77 year–old farmer with a 2 weeks history of coldness, pain and discoloration of the fingers of both the extremities which had extended to the mid forearm and shin regions. It was accompanied by skin erosions of the lower extremities, dark spots on the ear auricles and discoloration of the tip of the nose. On physical examination, quadrigangrene associated with ischemia of the auricles and tip of nose was seen. Serum proteins electrophoresis demonstrated monoclonal gammopathy and serum was positive for cryoglobulin, Bone marrow study showed neoplastic plasma cells infiltration. The patient was diagnosed as cryoglobulinemia based on multiple myeloma and treated accordingly.

  13. Penile Duplication and Two Anal Openings; Report of a Very Rare Case

    OpenAIRE

    Bakheet, Mohamed Abdel Al M.; Refaei, Mohammad

    2012-01-01

    Background Penile duplication (diphallus) is an extremely rare disorder. It is almost always associated with other malformations like double bladder, exstrophy of the cloacae, imperforate anus, duplication of the rectosigmoid and vertebral deformities. Meanwhile anal canal duplication, the most distal and least common duplication of the digestive tube and is a very rare congenital malformation. Case Presentation A 21 days old Egyptian neonate is reported with complete penile duplication and t...

  14. A Rare Case: Struma Ovarii in a 14-Year-Old Girl.

    Science.gov (United States)

    Iltar, Elif; Ureyen, Isin; Toptas, Tayfun; Savas, Melike; Çekiç, Sema; Uysal, Aysel

    2018-02-01

    Ovarian tumors presented with ovarian mass in childhood and adolescence are uncommon but an important part of gynecological cases. Struma ovarii is one of the rare cystic benign ovarian tumors that is observed predominantly in women who are between the ages of 40 and 60 years old. It is extremely rare in adolescents. Herein, we present a 14-year-old adolescent girl with struma ovarii who presented to the emergency room with abdominal pain.

  15. Aspergillus Salpingitis: A Rare Case Report

    African Journals Online (AJOL)

    rare entity, the correct diagnosis is of great importance for the indication of proper therapy. KEY WORDS: America ... Salpingitis is the most common serious infection in women .... immune systems, especially those recipients of stem-cell and.

  16. Megalourethra with Y-Type Duplication of Urethra Presented as Perianal Fistula: A Rare Case Report

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    Shashi Verma

    2015-01-01

    Full Text Available Megalourethra with Y-type duplication is an extremely rare anomaly. We report here one such case, diagnosed with retrograde urethrogram, which was done from both penile meatus and perianal opening simultaneously. Patient was successfully treated by laser optical internal urethrotomy (OIU, excision of duplicated urethra, and reduction urethroplasty in a single stage.

  17. Gingival fibromatosis with hypertrichosis syndrome: Case series of rare syndrome

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    Preetha Balaji

    2017-01-01

    Full Text Available Gingival fibromatosis with hypertrichosis syndrome is an extremely rare genetic condition characterized by profound overgrowth of hair and gums, as well as other variable features. Gingival fibromatosis is characterized by a large increase in the gingival dimension which extends above the dental crowns, covering them partially or completely. They were found to have a genetic origin, may also occur in isolation or be part of a syndrome, or acquired origin, due to specific drugs administered systemically. Congenital generalized hypertrichosis is a heterogeneous group of diseases with continuing excessive growth of terminal hair without androgenic stimulation. It has informally been called werewolf syndrome because the appearance is similar to that of a werewolf. Various syndromes have been associated with these features such as epilepsy, mental retardation, cardiomegaly, or osteochondrodysplasia. As so far very few cases have been reported in literature, we are reporting a series of three cases with management of the same. The excess gingival tissues, in these cases, were removed by conventional gingivectomy under general anesthesia. The postoperative result was uneventful and the patient's appearance improved significantly. Good esthetic result was achieved to allow patient to practice oral hygiene measures. Though this is not a serious condition clinically, psychosocial trauma cannot be neglected owing to the cosmetic disfigurement it produces.

  18. A rare case of acquired lymphangioma circumscriptum of the penis.

    Science.gov (United States)

    Adikari, S; Philippidou, M; Samuel, M

    2017-02-01

    Acquired lymphangioma circumscriptum is a rare occurrence on the penis. We report a case of a 47-year-old man who presented with a single lesion of acquired lymphangioma circumscriptum on the penis resembling genital warts. We report the case to increase awareness of this rare condition which may mimic sexually transmitted infections such as genital warts.

  19. Comparison between Three Rare Cases of Co‑Infection with ...

    African Journals Online (AJOL)

    Co‑infection in immunocompetent patients is rare. Though co‑infection with dengue and leptospira cases is increasingly reported, a co‑infection of this combination along with hepatitis E is rarely thought of. Until date only two case of triple co‑infection have been reported world‑wide. Here, we are reporting a patient with ...

  20. Rare Papillary Serous Carcinoma In A Nigerian: Case Report And ...

    African Journals Online (AJOL)

    Objective: To present a rare case of papillary serous carcinoma of the cervix and review the literature. Materials and Methods: An illustrative case seen by the authors in a fifty-two year old Nigerian woman with stage III carcinoma of the cervix. Results: The clinical and pathological features of this rare tumour are discussed ...

  1. Seckel syndrome: A rare case report

    OpenAIRE

    Rinky Sisodia; Ravi Kadur Sundar Raj; Vipin Goel

    2014-01-01

    Seckel syndrome (SS) is a rare, autosomal recessive syndrome; characterized by severe intrauterine and postnatal growth retardation, microcephaly, mental retardation, and typical facial appearance with beaklike protrusion of the midface (bird headed). In addition to the characteristic craniofacial dysmorphism and skeletal defects, abnormalities have been described in the cardiovascular, hematopoietic, endocrine, gastrointestinal, and central nervous systems. Usually such patients have poor ps...

  2. A RARE CASE OF AZEOTROPIC COPOLYMERIZATION

    NARCIS (Netherlands)

    JONGSMA, T; KIMKES, P; CHALLA, G

    1991-01-01

    Copolymerizations with both reactivity ratios equal to one are rare. Therefore, we report here on the radical copolymerization of styrene and p-tert-butoxy-carbonyl-oxy-styrene in toluene with AIBN as initiator. We found it to be an azeotropic copolymerization for all compositions, i.e. f1 = F1 and

  3. Enamel renal syndrome: A rare case report

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    S V Kala Vani

    2012-01-01

    Full Text Available Enamel renal syndrome is a very rare disorder associating amelogenesis imperfecta with nephrocalcinosis. It is known by various synonyms such as amelogenesis imperfecta nephrocalcinosis syndrome, MacGibbon syndrome, Lubinsky syndrome, and Lubinsky-MacGibbon syndrome. It is characterized by enamel agenesis and medullary nephrocalcinosis. This paper describes enamel renal syndrome in a female patient born in a consanguineous family.

  4. Necrotizing streptococcal myositis of the upper extremity: a case report.

    Science.gov (United States)

    Reichert, Johannes C; Habild, Götz; Simon, Paul; Nöth, Ulrich; Krümpelmann, Jan B

    2017-08-15

    Necrotizing myositis is a rare but life-threatening soft-tissue infection characterized by rapidly spreading inflammation and subsequent necrosis of the affected tissue. The myositis is often caused by toxin-producing, virulent bacteria such as group A β-hemolytic streptococcus and associated with severe systemic toxicity. It is rapidly fatal unless diagnosed promptly and treated aggressively. However, necrotizing myositis is often initially misdiagnosed as a more benign soft-tissue infection as such fulminant, invasive muscle infections are rare with no more than 30 cases reported over the last century. We illustrate the case of a 74-year-old male Caucasian initially presenting with a progressing swelling and gradually oncoming pain of the upper right extremity. Rapidly, livid discolorations of the skin, blisters, hypoesthesia and severe pain resistant to analgesics treatment developed accompanied by disruption of the arterial blood flow. Due to a manifest compartment syndrome the patient was admitted to theater for fasciotomy of the arm. After multiple revision surgeries wound closure was achieved using a pedicled, fasciocutaneous parascapular flap and a free, ipsilateral anterolateral thigh flap. Microbiological analysis revealed group A β-hemolytic streptococcus, histology a bacterial interstitial myositis with necrotic muscular fibers. A high degree of clinical suspicion is necessary to avert potentially disastrous consequences of necrotizing myositis. Timely diagnosis, broad-spectrum antibiotic therapy, and aggressive surgical debridement of affected tissue are keys to the treatment of this serious, often life-threatening infection.

  5. Dens evaginatus and dens invaginatus in a double tooth: A rare case report

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    Gaurav Sharma

    2015-01-01

    Full Text Available The presence of dens invaginatus (DI and dens evaginatus (DE on same tooth is a rare phenomenon. However, when these dental anomalies occur on a double tooth, it becomes an extremely rare phenomenon. The authors report a rare case of DI and DE on fused permanent maxillary central incisor with supernumerary tooth in a 40-year-old male. The present article also focuses on the differentiating fusion from gemination and also reviews preventive and management strategies for tooth with complex dental anatomy.

  6. Revisiting Cementoblastoma with a Rare Case Presentation

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    Vijayanirmala Subramani

    2017-01-01

    Full Text Available Cementoblastoma is a rare benign odontogenic neoplasm which is characterized by the proliferation of cellular cementum. Diagnosis of cementoblastoma is challenging because of its protracted clinical, radiographic features, and bland histological appearance; most often cementoblastoma is often confused with other cementum and bone originated lesions. The aim of this article is to overview/revisit, approach the diagnosis of cementoblastoma, and also present a unique radiographic appearance of a cementoblastoma lesion associated with an impacted tooth.

  7. A Rare Case of Oestrogen Dermatitis.

    Science.gov (United States)

    Bourgeault, Emilie; Bujold, Janie; Doucet, Marie-Eve

    Oestrogen dermatitis is a rare disorder characterised by cyclical eruptions in association with a woman's menstrual cycle. A 43-year-old woman with an 8-year history of cyclical inguinal dermatitis, with a negative patch test, was tested with intradermal progesterone and oestrogen. Intradermal testing was positive for oestrogen only. In a female patient with cyclical dermatitis, it is important to consider oestrogen or progesterone dermatitis in the differential diagnosis.

  8. Cartap poisoning: A rare case report.

    Science.gov (United States)

    Kumar, A S Praveen; Amalnath, Deepak; Dutta, T K

    2011-10-01

    Cartap is a pesticide commonly used to control weevil and caterpillars. It is an analogue of nereistoxin, a neurotoxic substance isolated from the marine annelid Lumbriconereis heteropoda. It causes neuromuscular blockade. Poisoning with cartap is very rare and not yet reported from India. We report a 35-year-old lady with cartap poisoning who presented with nausea, vomiting, and dyspnea. She improved with N-acetyl cysteine and symptomatic management.

  9. Cartap poisoning: A rare case report

    OpenAIRE

    Kumar, A. S. Praveen; Amalnath, Deepak; Dutta, T. K.

    2011-01-01

    Cartap is a pesticide commonly used to control weevil and caterpillars. It is an analogue of nereistoxin, a neurotoxic substance isolated from the marine annelid Lumbriconereis heteropoda. It causes neuromuscular blockade. Poisoning with cartap is very rare and not yet reported from India. We report a 35-year-old lady with cartap poisoning who presented with nausea, vomiting, and dyspnea. She improved with N-acetyl cysteine and symptomatic management.

  10. Isolated Subclavian Vein Injury: A Rare and High Mortality Case

    Directory of Open Access Journals (Sweden)

    Sahin Iscan

    2013-01-01

    Full Text Available Isolated subclavian vein injuries are rarely seen without concomitant arterial injury, bone fracture, damage to brachial plexus, and thoracal traumas. Our case was brought to the emergency service 6 hours after he had been shot at the shoulder with a firearm. After detection of extravasation from the left axillary and subclavian vein on arteriographic and venographic examinations, he was operated on. An autogenous saphenous vein graft was interposed between subclavian and axillary veins. Cardiac arrest developed twice because of hypovolemia, which was resolved with medical therapy. Subclavian vein injuries have a more mortal course when compared with the injuries to the subclavian arteries. Its most important reason is excessive blood loss and air embolism because of delayed arrival to hospital. As is the case in all vascular injuries, angiography is the most important diagnostic examination. If the general health state of the patient permits, arteriography and venography should be performed in patients potentially exposed to vascular injuries. In patients with extreme blood loss and deteriorated health state, direct surgical exploration of the injury site, containment of the bleeding, and venous repair are life-saving approaches.

  11. Hydatid disease of the spine: A rare case

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    Mona Agnihotri

    2017-01-01

    Full Text Available Hydatid disease or hydatidosis is the most widespread zoonosis caused by Echinococcus granulosus. Liver and lungs are the most common sites. Bone involvement is rare and reported in 0.5%–4% with spinal involvement reported in 50% of these cases. We present a case of spinal hydatidosis in a 35-year-old male presenting with lower extremity weakness and numbness. Magnetic resonance imaging (MRI of the spine showed multiple cystic lesions at the T9–T11 level with involvement of the paraspinal muscles. The lesion was seen intraspinal, intradural, intramedullary, and epidural. Radiological impression was aneurysmal bone cyst. The patient underwent laminectomy, and the excised cysts showed characteristic features of hydatid cyst (HC on histopathology. The patient was started on antihelminthic therapy postoperatively. MRI is a diagnostic modality for HC, but the unusual location and absence of characteristic features can cause diagnostic difficulty. A high index of suspicion should be kept in patients residing in endemic areas and presenting with unusual cystic lesion of spine.

  12. A Rare Case of Neonatal Hypophosphatasia: A Case Report

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    Nasim Pouralizadeh

    2018-03-01

    Full Text Available Hypophosphatasia is a rare hereditary disorder of bone metabolism.In this article, we presented the case of a male infant with a soft skull and short, deformed limbs at birth, followed by seizures and respiratory distress during admission in the neonatal intensive care unit (NICU. Prenatal ultrasound showed limb hypoplasia, skull hypomineralization, and polyhydramnios. Seizures occurred on day nine of admission. The neonate was intubated after pneumonia on day 12of birth and died due to the same cause and respiratory failure on day 14. Clinical presentation and low alkaline phosphatase (ALP confirmed the diagnosis of hypophosphatasia. The disorder covers a spectrum of severe neonatal type with severe hypomineralization to various adult types with osteomalacia and dental problems. Prenatal hypophosphatasia is diagnosed based on the clinical signs, including soft skull, short limbs, breathing difficulty, seizures, respiratory distress, laboratory results (low ALP and high pyridoxal 5-phosphate, and radiographic findings (hypomineralization and metaphyseal dysplasia.

  13. A rare case of vulvar squamous cell carcinoma; case presentation

    Directory of Open Access Journals (Sweden)

    Cristina N. Cozma

    2018-05-01

    Full Text Available Objectives. Vulvar cancer is a rare gynecological malignancy, with an incidence of 1.5 per 100 000 women/year. The most common vulvar cancer is developed in squamous cells, the most encountered type of skin cells. Case report. We report a case of a 72-year-old female admitted in the Department of Plastic Surgery of Emergency Clinical Hospital “Prof. Dr. Agrippa Ionescu” with a 5/4.2 cm painful ulcerated tumoral mass located in the vulvar area. The lesion slowly increased in size over the past 12 months. The tumour was surgically removed with oncological safety margins and sent for histopathological evaluation. The histopathological examination revealed an ulcerated squamous carcinoma with lymphovascular and perineural invasion, but with negative margins. Postoperative results were favorable, and no local or general complications were observed. Conclusion. We highlight this case due to its unusual presentation in the clitoral area. Moreover, considering the potential for recurrence we point out the importance of the radical vulvectomy with regional lymphadenectomy and histopathological examination, in order to put a precise diagnosis and ensure the best possible treatment for the patient.

  14. Canalicular adenoma: A rare case report

    Directory of Open Access Journals (Sweden)

    Swati Phore

    2018-01-01

    Full Text Available Canalicular adenomas (CAs are uncommon benign salivary gland neoplasms of the oral cavity. They are typically located on the upper lip, buccal mucosa, and infrequently found on the palate and derived from minor salivary glands. Due to benign character of the tumor, CAs rarely present with bone erosion. Histologically, trabecular type of basal cell adenoma, pleomorphic adenoma, and polymorphous low-grade adenocarcinoma should be discriminated from CAs. A-36-year-old female patient with CA was presented. The lesion was managed surgically under local anesthesia, and 2 months follow-up was uneventful.

  15. A rare case of acute lymphoblastic leukaemia with hemophilia A

    Directory of Open Access Journals (Sweden)

    John Biju

    2009-12-01

    Full Text Available Abstract A rare case of Acute lymphoblastic leukemia with hemophillia in a 12 year old boy is presented in the article. Patient was known case of hemophillia (factor VIII deficiency. He was diagnosed as a case of ALL based on bone marrow examination and immunophenotypic study. Patient was treated as per Children Cancer group guidelines. The main aim of reporting this rare association lies in developing treatment strategies in preventing life threatening bleeding due to this rare association which though may be accidental but need further research.

  16. A rare case of malignant triton tumor in the cerebellopontine angle

    Directory of Open Access Journals (Sweden)

    Gong Li

    2012-04-01

    Full Text Available Abstract Malignant triton tumor (MTT is defined as malignant peripheral nerve sheath tumor with rhabdomyoblastic differentiation. Intracranial MTT is extremely rare, and only four cases have been reported in the literature. Here, we report a case of MTT occurring in the cerebellopontine angle, and describe its histopathological characteristics, immunohistochemical features, and prognosis. Virtual slides The virtual slide(s for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/1336227313684480

  17. A rare case of malignant triton tumor in the cerebellopontine angle.

    Science.gov (United States)

    Gong, Li; Liu, Xiao-Yan; Zhang, Wen-Dong; Han, Xiu-Juan; Yao, Li; Zhu, Shao-Jun; Lan, Miao; Li, Yan-Hong; Zhang, Wei

    2012-04-19

    Malignant triton tumor (MTT) is defined as malignant peripheral nerve sheath tumor with rhabdomyoblastic differentiation. Intracranial MTT is extremely rare, and only four cases have been reported in the literature. Here, we report a case of MTT occurring in the cerebellopontine angle, and describe its histopathological characteristics, immunohistochemical features, and prognosis. The virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/1336227313684480.

  18. A Rare Renal Epithelial Tumor: Mucinous Cystadenocarcinoma Case Report and Review of the Literature

    Directory of Open Access Journals (Sweden)

    Abdulkadir Tepeler

    2011-01-01

    Full Text Available Primary renal mucinous cystadenocarcinoma is a very rare lesion of kidney which originates from the metaplasia of the renal pelvic uroepithelium. Only one case with primary mucinous cystadenocarcinoma has been reported in the English literature. We report second case of mucinous cystadenocarcinoma which was radiologically classified as type-IIF Bosniak cyst in peripheral localization. We aimed to present this extreme and unusual entity with its radiological, surgical, and pathologic aspects under the light of literature.

  19. External auditory canal leech: a rare case report of paediatric ...

    African Journals Online (AJOL)

    Leeches are blood sucking organism feed on human blood. While human bites are common, they rarely cause human internal infestation. We describe a rare case of a parasitic leech infestation of the External Auditory Canal (EAC). A two month old child presented to the Emergency department with a seven day history of ...

  20. A rare case of juvenile dermatomyositis and review of literature

    Directory of Open Access Journals (Sweden)

    Anjali T Bharani

    2017-01-01

    Full Text Available Idiopathic inflammatory myopathies are rare group of systemic connective tissue diseases. The hallmark of these disorders is symmetrical chronic inflammation and weakness of proximal muscles. Juvenile dermatomyositis (JDM is the most common inflammatory myositis in children. We describe a rare case of JDM in a 4-year-old female child who presented with characteristic cutaneous rash and proximal muscle weakness.

  1. A rare case of a sharp foreign body on the vocal cord

    Directory of Open Access Journals (Sweden)

    Khairunnisak Misron

    2017-08-01

    Full Text Available A foreign body (FB in the upper aerodigestive tract is a common clinical problem that presents as as acute emergency. Sharp FB, such as fish bone or chicken bone, commonly lodges in the tonsil, base of tongue, vallecula or pyriform fossa. Dislodgement of a FB into the laryngopharynx is very rare and specifically onto the vocal cord is extremely uncommon. This case report illustrates a rare case of a sharp FB that was dislodged into the airway and stuck on to the right vocal cord, which was removed under local anaesthesia.

  2. Isolated pulmonary candidiasis in a patient with diabetes mellitus: A rare case report.

    Science.gov (United States)

    Hakamifard, Atousa; Khorvash, Farzin; Raisi, Arash

    2016-01-01

    Fungal infections are as a cause of morbidity and mortality in immunocompromise patients. Because the respiratory tract is colonized with Candida, the presence of this agent in respiratory specimens makes the diagnosis of Candida pneumonia problematic. Candida pneumonia is a rare infection, and the majority of cases are secondary to hematogenous dissemination. Furthermore isolated Candida pneumonia originating from endotracheal inoculation is an extremely rare entity. We describe a case of isolated pulmonary candidiasis in the form multiple nodular lesions in a patient with long-term history of diabetes mellitus without evidence of fungemia or systemic involvement who responded to antifungal therapy.

  3. Isolated pulmonary candidiasis in a patient with diabetes mellitus: A rare case report

    Directory of Open Access Journals (Sweden)

    Atousa Hakamifard

    2016-01-01

    Full Text Available Fungal infections are as a cause of morbidity and mortality in immunocompromise patients. Because the respiratory tract is colonized with Candida, the presence of this agent in respiratory specimens makes the diagnosis of Candida pneumonia problematic. Candida pneumonia is a rare infection, and the majority of cases are secondary to hematogenous dissemination. Furthermore isolated Candida pneumonia originating from endotracheal inoculation is an extremely rare entity. We describe a case of isolated pulmonary candidiasis in the form multiple nodular lesions in a patient with long-term history of diabetes mellitus without evidence of fungemia or systemic involvement who responded to antifungal therapy.

  4. Case Study: Camptocormia, a Rare Conversion Disorder.

    Science.gov (United States)

    Rajmohan, Velayudhan; Thomas, Biju; Sreekumar, Kumaran

    2004-01-01

    Camptocormia is a condition characterized by severe frontal flexion of the spinal cord and knees, with passive drooping of both arms. It occurs as a form of conversion disorder. Some cases are associated with behavioral problems. A case of camptocormia of 2-year duration in a south Indian adolescent girl with oppositional defiant disorder and…

  5. A very rare, petro-clival, neurothekeoma tumor. Case Report

    Directory of Open Access Journals (Sweden)

    Iacob G.

    2015-09-01

    Full Text Available Known as nerve sheath myxoma too, neurothekeoma are benign tumors, usually arise in the skin of the head, neck region and upper extremities, in young females. Cerebral neurothekeoma are very rare, a few cases were already described in the parasellar area, in the middle cranial and posterior fossa. We present a petro-clival neurotekoma. A 78-year-old male was admitted for two years left fronto-temporal headache completed in the last 6 months with left trigeminal V1 neuralgia, left facial hypoesthesia, diplopia, swallowing disorders for liquid foods, balance disorders. From his medical records we noticed: stage 2 chronic kidney disease, hypertension, prostate adenoma, dyslipidemia hypercholesterolemia. The MRI showed a macronodular petroclival mass in hiper T2, hipo T1, flair iso signal; normal cerebral angiography. The patient was operated on using a left retrosigmoid, retromastoidian approach. A 4/3/3 cm tumor, gray, encapsulated, soft consistency, partially cystic, less bleeding, attached to the dura, displacing the basilar artery and brain stem controlateral, encasing the trigeminal nerve. The tumor was totally removed with a good postop evolution. Six months follow up he had no more facial pain, but only persistant left facial hypoesthesia. Histologically the tumor had lobular appearance with spindle or stellate cells embedded in abundant myxoid background. The tumor cells were diffusely positive for S100, PGP9.5’, CD 34” positive in vessels, Ki67’positive in 5%. Cranial MRI performed one month after surgery did not show any residual tumor. Also known as nerve sheath myxomas, neurothekoma are rare benign tumors. For intracranial neurothekoma surgical indication is mandatory

  6. Primary Orbital Chondromyxoid Fibroma: A Rare Case.

    Science.gov (United States)

    Mullen, Martin G; Somogyi, Marie; Maxwell, Sean P; Prabhu, Vikram; Yoo, David K

    A 56-year-old male with history of chronic sinusitis was found to have a 3 cm left orbital lesion on CT. Subsequent MRI demonstrated a multilobulated enhancing soft tissue lesion at the superotemporal region of the left orbit. Initial biopsy was reported as a low-grade sarcoma. On further evaluation, a consensus was made that the lesion was likely a benign mixed mesenchymal type tumor but should nonetheless be surgically removed. Left lateral orbitotomy was performed which revealed a tumor originating in the lateral orbital bone with segments eroding through the wall of the orbit. Intraoperative frozen sections revealed myoepitheliod tissue with locally aggressive features and the tumor was completely removed. The final histopathologic analysis of the tissue was consistent with a chondromyxoid fibroma. Chondomyxoid fibroma is a rare entity in the orbital bones and is more commonly seen in long bones.

  7. Two case studies on NARCCAP precipitation extremes

    Science.gov (United States)

    Weller, Grant B.; Cooley, Daniel; Sain, Stephan R.; Bukovsky, Melissa S.; Mearns, Linda O.

    2013-09-01

    We introduce novel methodology to examine the ability of six regional climate models (RCMs) in the North American Regional Climate Change Assessment Program (NARCCAP) ensemble to simulate past extreme precipitation events seen in the observational record over two different regions and seasons. Our primary objective is to examine the strength of daily correspondence of extreme precipitation events between observations and the output of both the RCMs and the driving reanalysis product. To explore this correspondence, we employ methods from multivariate extreme value theory. These methods require that we account for marginal behavior, and we first model and compare climatological quantities which describe tail behavior of daily precipitation for both the observations and model output before turning attention to quantifying the correspondence of the extreme events. Daily precipitation in a West Coast region of North America is analyzed in two seasons, and it is found that the simulated extreme events from the reanalysis-driven NARCCAP models exhibit strong daily correspondence to extreme events in the observational record. Precipitation over a central region of the United States is examined, and we find some daily correspondence between winter extremes simulated by reanalysis-driven NARCCAP models and those seen in observations, but no such correspondence is found for summer extremes. Furthermore, we find greater discrepancies among the NARCCAP models in the tail characteristics of the distribution of daily summer precipitation over this region than seen in precipitation over the West Coast region. We find that the models which employ spectral nudging exhibit stronger tail dependence to observations in the central region.

  8. Malignant mandibular tumors: two case reports of rare mandibular ...

    African Journals Online (AJOL)

    Arun Kumar Agnihotri

    2014-02-26

    Feb 26, 2014 ... present two cases of rare malignant mandibular tumors in a single institution. KEY WORDS: .... Spiculated osteoblastic periosteal reaction was noted with mild ... displacement of right 2nd premolar and 2nd molar teeth were ...

  9. Primary histiocytic sarcoma of the uterine cervix: an extremely rare entity

    Directory of Open Access Journals (Sweden)

    Binesh Fariba

    2017-03-01

    Full Text Available Objective. Histiocytic sarcoma (HS is an extraordinary rare tumor and it has an offensive clinical course. HS of the uterine cervix is a far uncommon tumor with just a few cases described so far. Here we presented a case of primary HS of the uterine cervix in a 62-year-old female initially misdiagnosed as large cell non-keratinizing squamous cell carcinoma. To the best of our knowledge, this is the first reported case of HS of the uterine cervix from Iran.

  10. Extremely long posterior communicating artery diagnosed by MR angiography: report of two cases.

    Science.gov (United States)

    Uchino, Akira; Suzuki, Chihiro; Tanaka, Masahiko

    2015-07-01

    We report two cases of an extremely long left posterior communicating artery (PCoA) diagnosed by magnetic resonance (MR) angiography. The PCoA arose from the normal point of the supraclinoid internal carotid artery and fused with the posterior cerebral artery (PCA) at its posterior ambient segment, forming an extremely long PCoA and extremely long precommunicating segment of the PCA. To our knowledge, this is the first report of such variation. Careful observation of MR angiographic images is important for detecting rare arterial variations. To identify these anomalous arteries on MR angiography, partial maximum-intensity-projection images are useful.

  11. Ovarian Ectopic Pregnancy: a Rare Case Report

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    N Lotfian

    2017-02-01

    Full Text Available Background and aim: Ovarian pregnancy is an uncommon form of ectopic pregnancy which usually diagnosed so late. The aim of this study is to report a case of ovarian ectopic pregnancy Case presentation: A 19 years old woman, with a history of polycystic ovary, first pregnancy, gestation age 9 weeks and 4 days, visited the doctor. She was complaining of severe abdomen pain and vaginal spotting and she was bedridden because of threatened miscarriage. She had discharged from hospital with progesterone suppository prescription. Requesting a transvaginal ultrasound and heterogeneous echogenic mass (size18×8/5 was shown near the left ovary. It was shown as ectopic pregnancy. The patient was hospitalized by ectopic pregnancy in ovary diagnosis and she was treated by methotrexate. Conclusion: In pregnant women that complain of bleeding and spotting in early pregnancy, in addition to threatened abortion, ectopic pregnancy should exist even in the absence of clinical symptoms, should be considered.

  12. Xanthogranulomatous Appendicitis: A Rare Case Report

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    Nikhil Mehrotra

    2017-07-01

    Full Text Available Xanthogranulomatous inflammation is a form of chronic inflammation where the exact eitiology is not known. It is commonly reported in organs like kidney and gall bladder Very few cases of xanthogranulomatous inflammation of appendix have been reported in the literature. We report a case of 30 year female presenting with pain abdomen and fever for which provisional diagnosis of appendicitis was made. Open appendicectomy was performed which on intraoperative examination showed that appendix was adherent to surrounding structures forming a mass. Resection of the mass was done and sent for histopathological examination. Gross examination of the received specimen revealed multiple irregular tissue bits and one tubular tissue bit. Multiple sections studied from resected tissue showed features of xanthogranulomatous inflammation in appendix and periappendicular tissue.

  13. Gorlin-Goltz syndrome: A rare case

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    Satyaki Ganguly

    2015-01-01

    Full Text Available Gorlin-Goltz syndrome or nevoid basal cell carcinoma syndrome is characterized by multiple basocellular epitheliomas, keratocysts in the jaws, bifid ribs, palmar and/or plantar pits and ectopic calcifications of the falx cerebri. We describe a case of Gorlin-Goltz syndrome illustrating the importance of a thorough examination including the examination of palms and soles and detailed investigations in a patient having lesions suggestive of basal cell carcinoma and multiple naevi.

  14. Gorlin-goltz syndrome: a rare case.

    Science.gov (United States)

    Ganguly, Satyaki; Jaykar, Kranti C; Kumar, Rajesh; Jha, Abhijeet Kumar; Banerjee, P K

    2015-01-01

    Gorlin-Goltz syndrome or nevoid basal cell carcinoma syndrome is characterized by multiple basocellular epitheliomas, keratocysts in the jaws, bifid ribs, palmar and/or plantar pits and ectopic calcifications of the falx cerebri. We describe a case of Gorlin-Goltz syndrome illustrating the importance of a thorough examination including the examination of palms and soles and detailed investigations in a patient having lesions suggestive of basal cell carcinoma and multiple naevi.

  15. Gorlin-Goltz Syndrome: A Rare Case

    OpenAIRE

    Ganguly, Satyaki; Jaykar, Kranti C; Kumar, Rajesh; Jha, Abhijeet Kumar; Banerjee, PK

    2015-01-01

    Gorlin-Goltz syndrome or nevoid basal cell carcinoma syndrome is characterized by multiple basocellular epitheliomas, keratocysts in the jaws, bifid ribs, palmar and/or plantar pits and ectopic calcifications of the falx cerebri. We describe a case of Gorlin-Goltz syndrome illustrating the importance of a thorough examination including the examination of palms and soles and detailed investigations in a patient having lesions suggestive of basal cell carcinoma and multiple naevi.

  16. Factitious cheilitis: A rare case report

    Directory of Open Access Journals (Sweden)

    Swati Phore

    2017-01-01

    Full Text Available Self-injurious behavior (SIB can be defined as the destruction or damage of body tissue without suicidal intent. Oral and perioral structures can be traumatized by SIB which involves biting of lips, cheek, lateral surface of the tongue, or buccal mucosa. Depending on its frequency and severity, SIB can lead to various degrees of self-injury. We hereby present a case of patient having lip lesion with positive history of lip chewing.

  17. Nance–Horan Syndrome: A Rare Case Report

    OpenAIRE

    Sharma, Shambhu; Datta, Pankaj; Sabharwal, Janak Raj; Datta, Sonia

    2017-01-01

    Dentofacial anomalies may guide us to the diagnosis of many congenital and hereditary syndromes. A 9-year-old boy was diagnosed with Nance–Horan syndrome. This syndrome is an extremely rare X-linked genetic disorder which is entirely expressed in males with semi-dominant transmission which results from mutations occurring in male gametes. It is characterized by facial dysmorphism such as long face, prominent nose and mandibular prognathism, ocular abnormalities such as congenital cataract, mi...

  18. PRIMARY CONJUNCTIVAL TUBERCULOSIS – A RARE CASE REPORT

    Directory of Open Access Journals (Sweden)

    Kulkarni Dinesh R, Sulegaon Ritesh V, Chulki Shashidhar F

    2015-07-01

    Full Text Available Tuberculosis is an endemic disease in India. Primary conjunctival tuberculosis is an uncommon condition and with better treatment of pulmonary tuberculosis it is now becoming extremely rare. Primary conjunctival Tuberculosis can present as unilateral conjunctivitis, hence laterality, chronicity and non-resolution of symptoms on treatment are indications for biopsy. In our patient conjunctival Tuberculosis was diagnosed on histopathology, which resulted in early implementation of antikochs’ treatment and complete resolution of the disease condition.

  19. Robinow Syndrome: A Rare Case Report and Review of Literature

    OpenAIRE

    Soman, Cristalle; Lingappa, Ashok

    2015-01-01

    ABSTRACT Robinow syndrome is an extremely rare genetic disorder. Short-limbed dwarfism, abnormalities in the head, face, and external genitalia, as well as vertebral defects comprise its distinct features. This disorder exists in dominant and recessive patterns. Patients with the dominant pattern exhibit moderate symptoms. More physical characteristics and skeletal abnormalities characterize the recessive group. The syndrome is also known as Robinow-Silverman-Smith syndrome, Robinow dwarfism,...

  20. Extremity Soft Tissue Sarcoma: A Review of 19 Cases. | Eyesan ...

    African Journals Online (AJOL)

    Background: Although soft tissue sarcoma is a rare tumour, it accounts for a significant proportion of malignancies seen in many orthopaedic practices. The objectives of this study are to evaluate the pattern of presentation of extremity soft tissue sarcoma and the treatment outcome in our patients. Method: This is a 3 year ...

  1. A RARE CASE OF THORACIC ACTINOMYCOSIS

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    Priyanka Das

    2017-10-01

    Full Text Available PRESENTATION OF CASE Actinomycetes are branching gram-positive anaerobic bacteria belonging to Actinomycetaceae family and are commensals in human oropharynx, gastrointestinal tract and female genitalia. Thoracic or pulmonary actinomycosis is an uncommon bacterial infection. The diagnosis of pulmonary or thoracic actinomycosis is often confounding because of its shared clinical features with malignant lung diseases and chronic suppurative lung diseases. However, chest physicians should be aware of actinomycosis being a differential diagnosis in persistent shadows in lung as early diagnosis leads to good prognosis. 1

  2. Rapunzel syndrome: a rare postpartum case.

    Science.gov (United States)

    Tegene, Teshome; Foda, Yahia; Hussain, Omar; Oloniyo, Kolawole; Ha, Ngoc-Tram; Manikonda, Geeta

    2013-01-01

    The Rapunzel syndrome describes a disorder in which a significant amount of hair is swallowed, forming a trichobezoar that extends past the stomach into the small intestines. Given the indigestible nature of hair, it subsequently leads to obstruction within the gastrointestinal system. Clinically, patients may present with symptoms of gastrointestinal obstruction, including abdominal complaints such as pain, nausea, vomiting, and diarrhea. However, due to its broad and nonspecific presenting symptoms, the diagnosis of Rapunzel syndrome warrants consideration once other common etiologies have been excluded. Surgical intervention is often required to remove the abdominal mass. This unusual syndrome is often associated with psychiatric disorders, affecting young women most commonly. In this report, we will discuss a unique case of Rapunzel syndrome in a one-month postpartum woman.

  3. Rapunzel Syndrome: A Rare Postpartum Case

    Directory of Open Access Journals (Sweden)

    Teshome Tegene

    2013-01-01

    Full Text Available The Rapunzel syndrome describes a disorder in which a significant amount of hair is swallowed, forming a trichobezoar that extends past the stomach into the small intestines. Given the indigestible nature of hair, it subsequently leads to obstruction within the gastrointestinal system. Clinically, patients may present with symptoms of gastrointestinal obstruction, including abdominal complaints such as pain, nausea, vomiting, and diarrhea. However, due to its broad and nonspecific presenting symptoms, the diagnosis of Rapunzel syndrome warrants consideration once other common etiologies have been excluded. Surgical intervention is often required to remove the abdominal mass. This unusual syndrome is often associated with psychiatric disorders, affecting young women most commonly. In this report, we will discuss a unique case of Rapunzel syndrome in a one-month postpartum woman.

  4. Cutaneous sarcoidosis: A rare case report

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    Bindu Suparna M, Joshi Shivani

    2014-07-01

    Full Text Available Sarcoidosis is a Greek word (Sarco means flesh and Eido means type or like. Cutaneous sarcoidosis occurs in up to one third of patients with systemic sarcoidosis. This disease is characterised by the presence of non – caseating epitheloid cell granulomas in the skin. Cutaneous sarcoidosis presents as a diagnostic challenge to the dermatopathologists due to its varied presentations and almost identical histologic pictures. Hence, exclusion of infectious causes and compatibility with clinical and radiologic picture serve as significant criteria to come up to a diagnosis. Sometimes; skin lesions are the first manifestation of systemic sarcoidosis. This is not a contagious or allergic disease. There is a risk of development of systemic manifestations at a later date; for which a close follow up is a must. We are presenting a case of cutaneous sarcoidosis, which later on progress to sarcoidosis with systemic manifestations.

  5. A RARE CASE OF PAROTID CYST

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    Rambabu

    2015-09-01

    Full Text Available A 28 years old male presented with a case of painless soft fluctuant swelling of right parotid gland is reported to our hospital. The lesion was found to be a cystic lesion through the pre - operative examinations and investigations. The cyst was completely excised, taking care not to injure the lower division of the facial nerve. Po st recovery was uneventful with no defect of the facial nerve functions. The histologic picture confirmed that the cyst was lymphoepithelial cyst which is so called “branchial cyst”. Through the literature reviews of parotid lymphoepit h elial cyst the discu ssions on prevalence, origin, diagnosis, histological finding, investigation and the modes of treatment are made. The ultra sound was found to be valuable in the pre - operative evaluation of the parotid swelling furthermore it is non - invasive, harmless, pai nless and relatively quick

  6. Extremely rare collapse and build-up of turbulence in stochastic models of transitional wall flows.

    Science.gov (United States)

    Rolland, Joran

    2018-02-01

    This paper presents a numerical and theoretical study of multistability in two stochastic models of transitional wall flows. An algorithm dedicated to the computation of rare events is adapted on these two stochastic models. The main focus is placed on a stochastic partial differential equation model proposed by Barkley. Three types of events are computed in a systematic and reproducible manner: (i) the collapse of isolated puffs and domains initially containing their steady turbulent fraction; (ii) the puff splitting; (iii) the build-up of turbulence from the laminar base flow under a noise perturbation of vanishing variance. For build-up events, an extreme realization of the vanishing variance noise pushes the state from the laminar base flow to the most probable germ of turbulence which in turn develops into a full blown puff. For collapse events, the Reynolds number and length ranges of the two regimes of collapse of laminar-turbulent pipes, independent collapse or global collapse of puffs, is determined. The mean first passage time before each event is then systematically computed as a function of the Reynolds number r and pipe length L in the laminar-turbulent coexistence range of Reynolds number. In the case of isolated puffs, the faster-than-linear growth with Reynolds number of the logarithm of mean first passage time T before collapse is separated in two. One finds that ln(T)=A_{p}r-B_{p}, with A_{p} and B_{p} positive. Moreover, A_{p} and B_{p} are affine in the spatial integral of turbulence intensity of the puff, with the same slope. In the case of pipes initially containing the steady turbulent fraction, the length L and Reynolds number r dependence of the mean first passage time T before collapse is also separated. The author finds that T≍exp[L(Ar-B)] with A and B positive. The length and Reynolds number dependence of T are then discussed in view of the large deviations theoretical approaches of the study of mean first passage times and

  7. Extremely rare collapse and build-up of turbulence in stochastic models of transitional wall flows

    Science.gov (United States)

    Rolland, Joran

    2018-02-01

    This paper presents a numerical and theoretical study of multistability in two stochastic models of transitional wall flows. An algorithm dedicated to the computation of rare events is adapted on these two stochastic models. The main focus is placed on a stochastic partial differential equation model proposed by Barkley. Three types of events are computed in a systematic and reproducible manner: (i) the collapse of isolated puffs and domains initially containing their steady turbulent fraction; (ii) the puff splitting; (iii) the build-up of turbulence from the laminar base flow under a noise perturbation of vanishing variance. For build-up events, an extreme realization of the vanishing variance noise pushes the state from the laminar base flow to the most probable germ of turbulence which in turn develops into a full blown puff. For collapse events, the Reynolds number and length ranges of the two regimes of collapse of laminar-turbulent pipes, independent collapse or global collapse of puffs, is determined. The mean first passage time before each event is then systematically computed as a function of the Reynolds number r and pipe length L in the laminar-turbulent coexistence range of Reynolds number. In the case of isolated puffs, the faster-than-linear growth with Reynolds number of the logarithm of mean first passage time T before collapse is separated in two. One finds that ln(T ) =Apr -Bp , with Ap and Bp positive. Moreover, Ap and Bp are affine in the spatial integral of turbulence intensity of the puff, with the same slope. In the case of pipes initially containing the steady turbulent fraction, the length L and Reynolds number r dependence of the mean first passage time T before collapse is also separated. The author finds that T ≍exp[L (A r -B )] with A and B positive. The length and Reynolds number dependence of T are then discussed in view of the large deviations theoretical approaches of the study of mean first passage times and multistability

  8. Primary chondrosarcoma of breast - cytology with histopathological correlation: A rare case report with review of literature

    Directory of Open Access Journals (Sweden)

    Sankappa P. Sinhasan

    2014-01-01

    Full Text Available Malignant mesenchymal tumors of the breast other than angiosarcoma are extremely rare and comprise <0.5% of breast tumors. Primary chondrosarcoma of the breast is an extremely rare entity and only 10 cases are reported as single case reports in literature until date. A diagnosis of primary mammary sarcoma can be established only after excluding metaplastic carcinomas and malignant phyllodes by extensive sampling for evidence of in situ or invasive carcinoma. Here, we report a primary chondrosarcoma of breast in a 55-year-old lady diagnosed precisely on fine-needle aspiration cytology and confirmed by histopatholigcal examination after total mastectomy. We emphasize on diagnostic difficulties encountered in cytology smears and discuss differential diagnoses.

  9. Tetra-phocomelia: a rarest of rare case.

    Science.gov (United States)

    Shukla, Anil Kumar; Sanjay, S C; Krishna, L; Krishnappa, N

    2015-03-01

    We present a rarest of rare case of Tetra-Phocomelia evaluated by antenatal Ultrasonography. It is a condition seen in 0.62 per 100,000 live births. An ultrasonogram was done at 18 wk of pregnancy to assess the fetus and after termination gross specimen was evaluated and X-ray infantograms were done to confirm the findings. The case showed classic Tetra-Phocomelia with limbs like flippers of a seal. Our findings make it rarest of rare as only few cases have been so far reported.

  10. A Rare Case of Congenital Diabetes Insipidus

    Directory of Open Access Journals (Sweden)

    Tanvi eRege

    2015-07-01

    Full Text Available Congenital nephrogenic diabetes insipidus (NDI is a conformation disease resulting from protein misfolding. Ninety percent of mutations result from the inactivating mutations of the arginine vasopressin receptor 2 (AVPR2 gene transmitted in an X-linked fashion, blocking the response to vasopressin, resulting in the inability to concentrate urine. Clinical features include polyuria, polydipsia, dehydration, and hypernatremia. They are generally more severely in affected males but present variably in females due to skewed inactivation of the X chromosome. We describe a case of a 40 year old woman with a history of Type 2 diabetes mellitus, hyperlipidemia, and obesity who presents with debilitating polyuria since the age of five with no clear diagnosis. Interestingly, her son was subsequently diagnosed with NDI. Genetic testing revealed that she was heterozygous for the Val88Met mutation in the AVPR2 gene while her son was hemizygous for the same. The patient has since been successfully treated with diuretics and a low solute diet. We highlight that although X-linked NDI patients are mostly males, it should be considered in symptomatic females to prevent delays in the diagnosis. Conformational diseases such as NDI are presently the subject of research using pharmacological chaperones to restore proper receptor membrane localization and function.

  11. A Rare Case of Congenital Diabetes Insipidus.

    Science.gov (United States)

    Rege, Tanvi; Polsani, Srujana; Jim, Belinda

    2015-01-01

    Congenital nephrogenic diabetes insipidus (NDI) is a conformation disease resulting from protein misfolding. Ninety percent of mutations result from the inactivating mutations of the arginine vasopressin receptor 2 (AVPR2) gene transmitted in an X-linked fashion, blocking the response to vasopressin, resulting in the inability to concentrate urine. Clinical features include polyuria, polydispsia, dehydration, and hypernatremia. They are generally more severely in affected males but present variably in females due to skewed inactivation of the X chromosome. We describe a case of a 40-year-old woman with a history of Type 2 diabetes mellitus, hyperlipidemia, and obesity, who presents with debilitating polyuria since the age of 5 with no clear diagnosis. Interestingly, her son was diagnosed with NDI. Genetic testing revealed that she was heterozygous for the Val88Met mutation in the AVPR2 gene while her son was hemizygous for the same. The patient has since been successfully treated with diuretics and a low solute diet. We highlight that although X-linked NDI patients are mostly males, it should be considered in symptomatic females to prevent delays in the diagnosis. Conformational diseases such as NDI are presently the subject of research using pharmacological chaperones to restore proper receptor membrane localization and function.

  12. Macrodystrophia lipomatosa – MR imaging of a rare congenital anomaly: review of 3 cases

    Directory of Open Access Journals (Sweden)

    Pallavi Aga

    2010-12-01

    Full Text Available Macrodystrophia lipomatosa (MDL results from progressive overgrowth of all the mesenchymal elements, with a disproportionate increase in fibro-adipose tissue. This rare congenital abnormality occurs most frequently in the distribution of the median nerve in the upper, and medial plantar nerve in the lower, extremity. Excess of unencapsulated fibro-adipose tissue on magnetic resonance imaging (MRI is virtually diagnostic and can differentiate this entity from other diseases with similar presentations. Involvement of the ulnar aspect of the hand, though rare, has been described; however, the lateral aspect of the foot as seen in our second case has not been reported, to the best of our knowledge.

  13. Rare Infratentorial and Supratentorial Localization of Juvenile Angiofibroma: A Case Report.

    Science.gov (United States)

    Pašalić, Ivan; Trninić, Ines; Nemir, Jakob; Jednačak, Hrvoje; Žarković, Kamelija; Mrak, Goran

    2016-01-01

    Angiofibromas are rare tumors of the head and neck that mostly occur in the sphenopalatine region. We present a case of angiofibroma in a young male patient with an unusual and extremely rare localization, which to our knowledge has not been described before. It was situated in the tentorium and spread to the supratentorial and infratentorial regions. The patient initially presented with symptoms of increased intracranial pressure. After a diagnostic evaluation was done, the whole tumor was successfully removed using the supratentorial and infratentorial approach and the microsurgical technique. © 2016 S. Karger AG, Basel.

  14. Transfusion reaction in a case with the rare Bombay blood group

    Directory of Open Access Journals (Sweden)

    Hayedeh Javadzadeh Shahshahani

    2013-01-01

    Full Text Available Bombay phenotype is extremely rare in Caucasian with an incidence of 1 in 250,000. When individuals with the Bombay phenotype need blood transfusion, they can receive only autologous blood or blood from another Bombay blood group. Transfusing blood group O red cells to them can cause a fatal hemolytic transfusion reaction. In this study, we report a case with the rare Bombay blood group that was misdiagnosed as the O blood group and developed a hemolytic transfusion reaction. This highlights the importance of both forward and reverse typing in ABO blood grouping and standard cross-matching and performing standard pretransfusion laboratory tests in hospital blood banks.

  15. Myoepithelial carcinoma of the male breast: a rare case report ...

    African Journals Online (AJOL)

    Myoepithelial carcinoma (malignant myoepithelioma) of the breast is a rare entity and in the male breast it is even rarer. Two cases of benign myoepithelioma in the male breast have been reported so far. Here we report, probably the first case of Myoepithelial carcinoma in a male breast with clinical features mimicking ...

  16. A Rare Case of Zosteriform Cutaneous Metastases from Squamous ...

    African Journals Online (AJOL)

    A Rare Case of Zosteriform Cutaneous Metastases from Squamous Cell Carcinoma of Hard Palate. ... examination, the patient had a superficial ulcer over the hard palate. A provisional diagnosis of zosteriform ... Majority of these cases can be misdiagnosed as herpes zoster and were treated with antiviral drugs. Distant ...

  17. A Rare Case of Craniopharyngioma in the Temporal Lobe

    Directory of Open Access Journals (Sweden)

    Sasan Razmjoo

    2017-01-01

    Full Text Available Herein, we report on a rare case of craniopharyngioma arising in the left temporal lobe with no prior history of head trauma or surgery. There was a solid-cystic mass in the left temporal lobe on MR images. To the best of our knowledge, this is the second case of a craniopharyngioma occurring in the temporal lobe.

  18. A rare presentation in two cases with review of literature

    African Journals Online (AJOL)

    2011-07-29

    Jul 29, 2011 ... liver, lungs, and the eyes.[1] Lymphadenopathy is a rare mode of presentation of cysticercus infestation.[2,3] We report two cases of cysticercus lymphadenitis involving the cervical lymph nodes. Case Report. A 7-year-old male child and a 28-year-old woman, both vegetarians by their food habit, presented ...

  19. Dipylidium caninum infection in a child: A rare case report

    OpenAIRE

    M V Narasimham; P Panda; I Mohanty; S Sahu; S Padhi; M Dash

    2013-01-01

    Dipylidiasis is a zoonotic parasitic infestation caused by the dog tapeworm Dipylidium caninum. Human dipylidiasis has been rarely reported in English literature. Young children are mostly at risk of acquiring the infection due to their close association with dogs and cats. We report a rare case of Dipylidium caninum infection in a 4 year old male child. The diagnosis was based on microscopic examination of stool. Confirmation of the proglottid segments was done by histopathological examinati...

  20. Monilethrix – Case report of a rare disease

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    Tasleem Arif

    2015-01-01

    Full Text Available Monilethrix is a rare genetic disorder of hair characterized by beaded appearance of the hair shaft leading to hair fragility and patchy dystrophic alopecia. In this disorder, the hair shaft has alternate widenings (nodes and constrictions (internodes that lead to fracture of hair shaft and varying degree of alopecia. We report an eight year old Kashmiri boy who presented with diffuse hair loss since infancy. As monilethrix is a rare disease entity which prompted us to report this case.

  1. A Rare Case of Aneurysmal Bone Cyst in the Paranasal Sinus

    Directory of Open Access Journals (Sweden)

    Seyyed Mostafa Hashemi

    2015-09-01

    Full Text Available Introduction: Aneurysmal Bone cysts (ABC are extremely rare in the head and neck region and even rarer in sinuses.  ABC is a benign multicystic mass that is locally-destructive and rapidly expandable. Hemorrhagic fluid content (like in this case and septated appearance are the characteristic feature of ABC. Established treatment options for ABCs include sclerotherapy, embolization, radiotherapy, simple curettage, surgical excision, or a combination of methods.   Case Report: In this article, a 5 year-old boy with a recurrent nasal mass is presented. The patient was finally diagnosed with this rare entity: ABC of the paranasal sinuses. The patient was treated through complete surgical removal.   Conclusion:  ABC can be considered as a rare differential diagnosis of recurrent nasal hemorrhagic mass in a pediatric population.

  2. Left Sided Amyand's Hernia, A Rare Occurance: A Case Report.

    Science.gov (United States)

    Ravishankaran, Praveen; Mohan, G; Srinivasan, A; Ravindran, G; Ramalingam, A

    2013-06-01

    This is a case report about a 35 year old man admitted with complains of obstructed left sided inguinal hernia. On exploration of the left inguinal canal to our surprise a normal appendix was found in addition to a gangrenous omentum. Resection of the gangrenous omentum was done. Appendectomy was done. This case is reported for its rare occurance as only three such cases of left sided amyand's hernia has been reported so far in literature[4-6].

  3. Gorlin Syndrome with Bilateral Polydactyly: A Rare Case Report

    Science.gov (United States)

    Acharya, Sonu; Panda, Swagatika; Sahoo, Sujit Ranjan; Ray, Prayas

    2013-01-01

    ABSTRACT Gorlin's syndrome is a rare disorder transmitted as an autosomal dominant trait. It is characterized by multiple disorders involving multiple systems. We present a case of 11-year-old male child presenting with multiple odontogenic keratocyst to the dental clinic. Retrograde diagnosis of Gorlin-Goltz syndrome was made after clinical and radiological investigation. How to cite this article: Acharya S, Panda S, Dhull KS, Sahoo SR, Ray P. Gorlin Syndrome with Bilateral Polydactyly: A Rare Case Report. Int J Clin Pediatr Dent 2013;6(3):208-212. PMID:25206225

  4. Dipylidium caninum infection in a child: A rare case report

    Directory of Open Access Journals (Sweden)

    M V Narasimham

    2013-01-01

    Full Text Available Dipylidiasis is a zoonotic parasitic infestation caused by the dog tapeworm Dipylidium caninum. Human dipylidiasis has been rarely reported in English literature. Young children are mostly at risk of acquiring the infection due to their close association with dogs and cats. We report a rare case of Dipylidium caninum infection in a 4 year old male child. The diagnosis was based on microscopic examination of stool. Confirmation of the proglottid segments was done by histopathological examination. To the best of our knowledge this is the first human case of Dipylidium caninum reported from this part of the country.

  5. Dipylidium caninum infection in a child: a rare case report.

    Science.gov (United States)

    Narasimham, M V; Panda, P; Mohanty, I; Sahu, S; Padhi, S; Dash, M

    2013-01-01

    Dipylidiasis is a zoonotic parasitic infestation caused by the dog tapeworm Dipylidium caninum. Human dipylidiasis has been rarely reported in English literature. Young children are mostly at risk of acquiring the infection due to their close association with dogs and cats. We report a rare case of Dipylidium caninum infection in a 4 year old male child. The diagnosis was based on microscopic examination of stool. Confirmation of the proglottid segments was done by histopathological examination. To the best of our knowledge this is the first human case of Dipylidium caninum reported from this part of the country.

  6. Adenoid cystic carcinoma of child: a rare case.

    Science.gov (United States)

    Mathai, Meera; Sherubin, J Eugenia; Agnihotri, Pg; Sangeetha, Gs

    2014-01-01

    Adenoid cystic carcinoma (ACC) is the second most common malignant tumor affecting both major and minor salivary glands. Clinically, it is a slowly growing tumor with high propensity for local invasion, recurrence and distant metastasis. It is predominantly seen in the ffith and sixth decades of life. Here, we report a rare case of ACC affecting the right maxilla of a 12-year-old girl. How to cite this article: Mathai M, Sherubin JE, Agnihotri PG, Sangeetha GS. Adenoid Cystic Carcinoma of Child: A Rare Case. Int J Clin Pediatr Dent 2014;7(3):206-208.

  7. Rare Case of Duodenal Metastasis From Pulmonary Squamous Cell Carcinoma

    Directory of Open Access Journals (Sweden)

    Zain Memon DO

    2017-10-01

    Full Text Available Pulmonary squamous cell carcinoma is the second most common non–small cell malignancy of the lung. It commonly metastasizes to the adrenal glands, bone, liver, brain, and kidneys. Most occurrences of metastatic squamous cell carcinoma involving the gastrointestinal tract originate from primary lung tumors. Metastasis to the duodenum, however, is exceedingly rare, with very few cases of stomach or duodenal involvement described in the literature. We report the case of a patient with stage IV pulmonary squamous cell carcinoma metastasizing to the duodenum with an uncommon presentation to add to the paucity of literature available regarding this rare finding.

  8. Metastatic hepatocellular carcinoma in the maxilla and mandible, an extremely rare presentation

    Directory of Open Access Journals (Sweden)

    Satya Ranjan Misra

    2015-01-01

    Full Text Available Malignancy is characterized by anaplasia, invasiveness, and metastasis. Primary oral squamous cell carcinoma is the most prevalent oral malignancy, but secondary malignancy from distant sites have also been reported. Hepatocellular carcinoma (HCC is a common primary liver malignancy that frequently metastasizes during the course of the disease, but < 1% of cases show oral involvement. Such secondary neoplasms do not have any pathognomonic clinical or radiologic findings, and therefore they pose a diagnostic challenge. Hence, in the differential diagnosis of malignant tumors of the oral cavity, it is essential to consider the occurrence of both primary as well as metastatic tumors despite the low incidence of the latter. A rare case of HCC metastasizing to both the maxilla and mandible is presented, in which the patient succumbed to the disease as a result of the delay in diagnosis.

  9. Nance-Horan Syndrome: A Rare Case Report.

    Science.gov (United States)

    Sharma, Shambhu; Datta, Pankaj; Sabharwal, Janak Raj; Datta, Sonia

    2017-01-01

    Dentofacial anomalies may guide us to the diagnosis of many congenital and hereditary syndromes. A 9-year-old boy was diagnosed with Nance-Horan syndrome. This syndrome is an extremely rare X-linked genetic disorder which is entirely expressed in males with semi-dominant transmission which results from mutations occurring in male gametes. It is characterized by facial dysmorphism such as long face, prominent nose and mandibular prognathism, ocular abnormalities such as congenital cataract, microcornea, microphthalmia and strabismus, and dental anomalies including mulberry molars and screwdriver-shaped incisors. Heterozygous females inherit this disease and also suffer from this syndrome but in a milder form. Approximately one-third of the affected males show signs of developmental delay and intellectual abnormalities. This syndrome is very rare and the incidence of the disease has not been established so far. The present article describes the clinical and radiological features and the genetic implications of this syndrome.

  10. Nance–Horan syndrome: A rare case report

    Directory of Open Access Journals (Sweden)

    Shambhu Sharma

    2017-01-01

    Full Text Available Dentofacial anomalies may guide us to the diagnosis of many congenital and hereditary syndromes. A 9-year-old boy was diagnosed with Nance–Horan syndrome. This syndrome is an extremely rare X-linked genetic disorder which is entirely expressed in males with semi-dominant transmission which results from mutations occurring in male gametes. It is characterized by facial dysmorphism such as long face, prominent nose and mandibular prognathism, ocular abnormalities such as congenital cataract, microcornea, microphthalmia and strabismus, and dental anomalies including mulberry molars and screwdriver-shaped incisors. Heterozygous females inherit this disease and also suffer from this syndrome but in a milder form. Approximately one-third of the affected males show signs of developmental delay and intellectual abnormalities. This syndrome is very rare and the incidence of the disease has not been established so far. The present article describes the clinical and radiological features and the genetic implications of this syndrome.

  11. Nance–Horan Syndrome: A Rare Case Report

    Science.gov (United States)

    Sharma, Shambhu; Datta, Pankaj; Sabharwal, Janak Raj; Datta, Sonia

    2017-01-01

    Dentofacial anomalies may guide us to the diagnosis of many congenital and hereditary syndromes. A 9-year-old boy was diagnosed with Nance–Horan syndrome. This syndrome is an extremely rare X-linked genetic disorder which is entirely expressed in males with semi-dominant transmission which results from mutations occurring in male gametes. It is characterized by facial dysmorphism such as long face, prominent nose and mandibular prognathism, ocular abnormalities such as congenital cataract, microcornea, microphthalmia and strabismus, and dental anomalies including mulberry molars and screwdriver-shaped incisors. Heterozygous females inherit this disease and also suffer from this syndrome but in a milder form. Approximately one-third of the affected males show signs of developmental delay and intellectual abnormalities. This syndrome is very rare and the incidence of the disease has not been established so far. The present article describes the clinical and radiological features and the genetic implications of this syndrome. PMID:29042737

  12. Small cell extraskeletal osteosarcoma: a rare case report

    Directory of Open Access Journals (Sweden)

    Neelam Sood

    2014-01-01

    Full Text Available Extraskeletal osteosarcoma is a rare malignant mesenchymal neoplasm and its small cell variant is one among the rarest variant. This article describes a 60-year-old woman presenting with a large, lobulated, painful mass in left thigh with associated history of trauma since 18 months. Her magnetic resonance imaging showed a variegated mixed intensity lesion with associated cystic degeneration, necrosis and matrix arborizing nearby muscles. Fine needle aspiration cytology showed a small cell lesion with very scant osteoid. Tumor was excised and histopathological diagnosis was small cell osteosarcoma involving adjacent muscles and fat with sparing of lymph nodes. The aim of this article is to present the clinical, radiological, cyto-histological and immunohistochemical features of this extremely rare lesion.

  13. Hemolymphangioma of the lower extremities in children: two case reports

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    Kosmidis Ilias

    2010-08-01

    Full Text Available Abstract Background and purpose Hemo-lymphangiomas are rare benign tumors that arise from congenital malformation of the vascular system. They are usually diagnosed at birth or early in childhood. The management of hemo-lymphangiomas in children remains challenging because complete resection is often difficult to be achieved and recurrences are common. Methods We present the case of two children with a mass on their left tibia. Imaging modalities, plain radiograph, Ultrasonography and Magnetic Resonance were used to investigate the nature of the mass, the anatomical relationship to the neighboring tissues and help planning the surgical resection. The dominant diagnosis was hemo-lymphangioma. Both lesions increased in size in a short period of follow-up thus we decided to proceed to surgical excision. The diagnosis of hemo-lymphangioma was confirmed by histological examination of the surgical specimen. Post-operatively, seroma was formed to the first patient, managed by placing a drainage and immobilizing the limb on a splint. The second patient experienced no complications post-operatively. After 12 months of follow-up both patients had no complications or recurrence. Conclusions Very few cases of hemo-lymphangiomas of the extremities have been reported in the literature. Those tumors can grow slowly and remain asymptomatic for a long period of time or may become aggressive and enlarge rapidly, without invasive ability though. Radical resection is the choice of treatment offering the lowest recurrence rates. Other therapeutic methods are: aspiration and drainage, cryotherapy, injection of sclerotic agents and radiotherapy; although none of those offers better results that the surgical excision.

  14. Oral malignant melanoma: a rare case with unusual clinical ...

    African Journals Online (AJOL)

    Primary Oral malignant melanoma is a rare tumor with an indigent prognosis. This is a case report of 47-year-old Sudanese female diagnosed as Oral malignant melanoma of the mandible with an unusual pattern of growth and clinical presentation. Furthermore, a possibility of intraosseous origin is suggested. Pan African ...

  15. Eye and rare genetic diseases: Case series and literature review ...

    African Journals Online (AJOL)

    Genetic diseases are generally characterised by a multi visceral pathogenesis. Although orphan, these diseases interest many disciplines due to their clinical expression. Eye is sometimes part of the clinical polymorphism of some rare genetic diseases. Ocular signs are in some cases leading to the diagnosis of these ...

  16. Fryns anophthalmia-plus syndrome: two rare cases.

    Science.gov (United States)

    Bozkurt, O; Bidev, D; Sari, F N; Dizdar, E A; Ulu, H O; Uras, N; Oguz, S S; Canpolat, F E; Dilmen, U

    2014-01-01

    Fryns anophthalmia-plus syndrome is a rare syndrome with clinical diversity primarily including anophthalmia/microphthalmia, facial clefts, cleft lip/palate, ear and nasal deformities. Here we present two different cases of APS with anopthalmia/microphthalmia, cleft palate, low set ears, ventriculomegaly and one of which had intestinal non-fixation anomaly not described in the literature before.

  17. A rare case of congenital Y-type urethral duplication

    Directory of Open Access Journals (Sweden)

    Charu Tiwari

    2015-11-01

    Full Text Available Duplication of urethra is a rare congenital anomaly. We report a case of Y-type of urethral duplication with the accessory urethra arising from posterior urethra and opening in the perineum. The orthotopic urethra was normal. The accessory urethral tract was cored, transfixed and divided. At 1 year of follow-up, the patient has no urinary complaints

  18. A Rare Case of β-Ketothiolase Deficiency

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    B. Modh

    2015-06-01

    Full Text Available We are reporting a case of β-ketothiolase deficiency, a rare disorder of amino acid metabolism. A 10 month old child presented with complaints of vomiting, convulsions, fever and altered sensorium that on investigations showed metabolic acidosis, hyperammonemia and ketosis. Gas chromatography/ mass spectroscopic examination was suggestive of β-ketothiolase deficiency.

  19. A Rare Case: Gastric Cancer; Involving Primery Thoracal Vertebral Metastases

    Directory of Open Access Journals (Sweden)

    Harun Arslan

    2013-06-01

    Full Text Available Primery bone metastases rarely occur in gastric cancer. Bone metastases indicate that the prognosis is bad. In that article we present a case that is diagnosed as a gastric cancer with primary bone metasteses that caused pathologic thoracal vertebral fracture seenby computer ised tomography.

  20. Neurenteric cyst - a case report of this rare disorder

    NARCIS (Netherlands)

    Schurink, M.; van Herwaarden-Lindeboom, M. Y. A.; Coppes, M. H.; Veldhuizen, A. G.; Koetse, H. A.; de Langen, Z. J.

    Neurenteric cysts are rare congenital lesions that are believed to be the result of the split notochord syndrome. We report the clinical case of a 5-year-old boy presenting with vague gastrointestinal symptoms and fatigue, who had undergone resection of a small intestine duplication cyst as a

  1. Aneurysmal bone cyst of maxillary alveolus: A rare case report

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    Subhas Chandra Debnath

    2016-01-01

    Full Text Available Aneurysmal bone cyst (ABC is a nonneoplastic rare pathologic entity of the jaws. Its locally aggressive nature and high recurrence rate after curettage make surgical resection a better treatment option. Here, we present a case of ABC of maxillary alveolus and its management by alveolectomy followed by white head varnish pack application in the surgical defect.

  2. Aneurysmal bone cyst of maxillary alveolus: A rare case report

    Science.gov (United States)

    Debnath, Subhas Chandra; Adhyapok, Apurba Kumar; Hazarika, Kriti; Malik, Kapil; Vatsyayan, Ashutosh

    2016-01-01

    Aneurysmal bone cyst (ABC) is a nonneoplastic rare pathologic entity of the jaws. Its locally aggressive nature and high recurrence rate after curettage make surgical resection a better treatment option. Here, we present a case of ABC of maxillary alveolus and its management by alveolectomy followed by white head varnish pack application in the surgical defect. PMID:27041915

  3. Primary Renal Synovial Sarcoma: A Rare Case Report

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    Taha Numan Yıkılmaz

    2016-12-01

    Full Text Available Synovial sarcoma (SS is mainly derived from soft tissues. Primary renal SS is a very rare malignancy with around 60 cases reported in the literature. We report a renal mass which was undistinguishable from urothelial carcinoma clinically and pathologically but diagnosed as a primary renal SS at the definitive pathological diagnosis.

  4. Neurological manifestations in speech after snake bite: A rare case ...

    African Journals Online (AJOL)

    Neurological manifestations in speech after snake bite: A rare case. D Vir, D Gupta, M Modi, N Panda. Abstract. No Abstract. Full Text: EMAIL FREE FULL TEXT EMAIL FREE FULL TEXT · DOWNLOAD FULL TEXT DOWNLOAD FULL TEXT · http://dx.doi.org/10.4314/pamj.v4i1.53597 · AJOL African Journals Online. HOW TO ...

  5. Primary intraspinal extradural primitive neuroectodermal tumor: A rare case.

    Science.gov (United States)

    Rege, Shrikant V; Tadghare, Jitendra; Patil, Harshad; Narayan, Sharadendu

    2016-01-01

    Primitive neuroectodermal tumors (PNETs) are aggressive childhood malignancies and are difficult to treat. Primary intraspinal PNETs are rare. These patients have poor prognosis with short survival time even after surgery and chemoradiation. As there are no standard guidelines exist for the management of these tumors, a multidisciplinary approach has been employed with varying success. According to the review of literature, only few cases of primary intraspinal extradural PNETs have been reported. Herein, author has described a case of intraspinal, extradural PNET.

  6. KIKUCHI-FUJIMOTO DISEASE (KFD): A Rare Case Report

    OpenAIRE

    Dr. Gunja Jain; Dr. Mayank Gupta; Dr. Laxmikant Goyal; Dr. Jai Purohit; Dr. Sudhir Mehta

    2017-01-01

    Abstract— Kikuchi-Fujimoto disease (KFD) is a rare disease. It has a worldwide distribution with a higher prevalence in Asians. KFD is usually a self limiting disease and benign in nature. Clinically it presents as regional cervical lymphadenopathy and sometimes may presents as generalized lymphadenopathy as well. Night sweats and low grade fever may also be associated in some cases. A case of a 36 year old female had attended in SMS Hospital. She presented with fever, weight loss and tender ...

  7. ASPERGILLOSIS OF MANDIBLE : A RARE CASE OF OSTEOMYELITIS

    OpenAIRE

    ÖZTÜRK, Nurdan; ERSOY, Burak; SÖNMEZ, Ahmet; ÇELEBİLER, Özhan; NUMANOĞLU, Ayhan

    2007-01-01

    Aspergillus species are saprophyticus fungi which may be the cause of infection predominantly in immunocompromised hosts. Aspergillosis is usually manifested in the respiratory system and bone involvement is rarely encountered. Osteomyelitis of the mandible with aspergillus species has been reported in only two cases in the literature. Treatment mainly consists of antimicrobial therapy in combination with surgery. We report a case of aspergillosis of the mandible in a patient who was under im...

  8. Cerebrotendinous xanthomatosis (a rare lipid storage disorder): a case report

    OpenAIRE

    Razi, Syed Mohd; Gupta, Abhinav Kumar; Gupta, Deepak Chand; Gutch, Manish; Gupta, Keshav Kumar; Usman, Syeda Iqra

    2016-01-01

    Background Cerebrotendinous xanthomatosis is a very rare autosomal recessive lipid storage disorder affecting bile acid biosynthesis. It is manifested by subtle neurological and non-neurological symptoms due to abnormal tissue lipid deposition. Diagnosis is usually delayed but early diagnosis and replacement therapy can prevent devastating neurological sequelae. Case presentation We present a case of a 25-year-old Asian Indian woman who presented with gait difficulty, fusiform swellings of bi...

  9. [Rare side effects in management of hyperthyroidism. Case report].

    Science.gov (United States)

    Sohár, Gábor; Kovács, Mónika; Györkös, Andrea; Gasztonyi, Beáta

    2016-05-29

    The authors present the case history of a patient suffering from hyperthyroidism. The diagnostic procedures revealed the presence of propylthiouracyl induced vasculitis with renal involvement, that recovered completely after the withdrawal of propylthiouracyl and corticosteroid treatment. Thereafter, the patient was treated with thiamasol, that caused agranulocytosis with fever. After transient litium carbonate therapy a succesful thyreoidectomy was performed. Cumulative serious side effects of antithyroid drugs are rare. This case highlights some of the challenges and complications encountered in the management of hyperthyroidism.

  10. Distant Migration of Multiple Siliconomas in Lower Extremities following Breast Implant Rupture: Case Report

    OpenAIRE

    Oh, Joo Hyun; Song, Seung Yong; Lew, Dae Hyun; Lee, Dong Won

    2016-01-01

    Summary: Siliconoma from ruptured breast implants has been reported in multiple body sites, including but not limited to the breast parenchyma, axillary lymph nodes, upper arm, and even lower leg. In this regard, we report a rare case of distant silicone migration to the lower extremities after traumatic breast implant rupture. A 55-year-old Asian woman who received bilateral augmentation mammoplasty 20 years ago presented with ruptured breast implants from a car accident 2 years earlier. Mag...

  11. Granulicatella adiacens abscess: Two rare cases and review

    Directory of Open Access Journals (Sweden)

    Sangita Gupta

    2018-01-01

    Full Text Available Granulicatella adiacens is a nutritionally variant streptococcus species. These bacteria are rarely isolated in the laboratory due to their fastidious growth requirements. These have been mostly reported from bloodstream infections, infective endocarditis, infections of orbit, nasolacrimal duct and breast implants. Here, we are reporting two cases of subcutaneous abscesses caused by G. adiacens. In first case, it was isolated from abscess around elbow joint and second case was a suprapatellar abscess. We have also reviewed the published data concerning diagnosis and antimicrobial susceptibility pattern of Granulicatella infections and included some Indian cases.

  12. An Unusual Presentation of Metanephric Adenofibroma: A Rare Case Report

    Directory of Open Access Journals (Sweden)

    Kiran Agarwal

    2017-10-01

    Full Text Available Metanephric adenofibroma is a rare renal neoplasm with only a few case reports in literature. In majority of cases, it is asymptomatic. However, it may present with haematuria, polycythemia or hypertension. Radiologically, it is indistinguishable from other solid renal tumours. Definitive diagnosis can only be made on the basis of histopathology. It is a benign neoplasm and requires only surgical excision with no need for chemotherapy. Involvement of urinary bladder and presentation as bladder mass has never been reported. In this case report, we present a case of metanephric adenofibroma in a two-year-old male child manifesting with haematuria and urinary bladder mass.

  13. Chondroblastoma of the thoracic spine: a rare location. Case report with radiologic-pathologic correlation

    Energy Technology Data Exchange (ETDEWEB)

    Venkatasamy, A. [University Hospital of Strasbourg, Department of Radiology, Strasbourg (France); Chenard, M.P. [University Hospital of Strasbourg, Department of Pathology, Strasbourg (France); Massard, G. [University Hospital of Strasbourg, Department of Thoracic Surgery, Strasbourg (France); Steib, J.P. [University Hospital of Strasbourg, Department of Spine Surgery, Strasbourg (France); Bierry, G. [University Hospital of Strasbourg, Department of Radiology, Strasbourg (France); University Hospital of Strasbourg, Department of Pathology, Strasbourg (France); University Hospital of Strasbourg, Department of Thoracic Surgery, Strasbourg (France); University Hospital of Strasbourg, Department of Spine Surgery, Strasbourg (France)

    2017-03-15

    Chondroblastoma is a rare benign cartilage neoplasm that arises from the appendicular skeleton in the vast majority of the cases (80%). Chondroblastoma of the spine is an even more rare condition (30 cases reported), and vertebral chondroblastomas, unlike chondroblastomas of the extremities, present with the appearance of an aggressive tumor on CT and MR imaging and occur at least a decade later. Even though vertebral chondroblastomas are very uncommon tumors, they should nonetheless be included in the differential diagnosis when encountered with an aggressive vertebral mass, and a histological confirmation should be performed. We present a case of chondroblastoma of the thoracic spine of a 27-year-old female for which detailed radiologic-pathologic correlation was obtained. (orig.)

  14. Glomangiomyoma of the neck in a child in Nepal: a rare case report and literature review

    Directory of Open Access Journals (Sweden)

    Bishow Tulachan

    2017-11-01

    Full Text Available Abstract Background Glomangiomyoma is a rare histological variant of glomus tumour. Clinically, it mimicks as a haemangioma and is challenging to diagnose. Its occurrence in the neck of a child has not been previously described. Case presentation A 3 year old girl presented with the complaints of painless progressive neck swelling in the right side for one and half year. Sonography, computed tomography (CT, magnetic resonance imaging (MRI, CT neck angiography and fine needle aspiration cytology (FNAC were suggestive of vacular malformation i.e. giant haemangioma or arteriovenous malformation. The mass was removed in toto under general anaesthesia without postoperative complications. The histopathology confirmed it to be glomangiomyoma with haemangiopericytoma like features. Conclusion It’s an extremely rare variant of glomus tumour and may be the first report of a glomangiomyoma in the neck of a child. Despite a rare entity, it should be borne in mind during differential diagnosis.

  15. Gingival enlargement unveiling sarcoidosis: Report of a rare case

    Directory of Open Access Journals (Sweden)

    Sabeeha Abbas Kadiwala

    2013-01-01

    Full Text Available Sarcoidosis is classified as an acquired systemic granulomatous disease. Because of the fact that sarcoidosis affects multiple tissues and organs, it is characterized by many potential signs and symptoms, as well as by the presence of non-caseating granulomas in the organs involved. Although oral sarcoidosis is relatively rare, it may however, present in the oral cavity. This report presents a rare case of sarcoidosis with the initial presenting symptom as severe generalized gingival enlargement. The gingival enlargement was treated by gingivectomy. After histopathological examination of gingival biopsy and certain special investigations, a diagnosis of sarcoidosis was made.

  16. A case report on esophageal tuberculosis – A rare entity

    Directory of Open Access Journals (Sweden)

    Vatsal Khanna

    2017-01-01

    Full Text Available This is a case report of a rare form of tuberculosis in a patient presenting with dysphagia. Patient was subjected to upper gastrointestinal endoscopy, which revealed an ulcerative growth in the distal esophagus. Histopathology revealed esophageal tuberculosis. Patient was managed conservatively with Anti-Tuberculosis Treatment (ATT. Follow up endoscopy after two months revealed resolution of the growth and patient was symptomatically better. In spite of the rare nature of the disease, it can be managed effectively with ATT to avoid complications (fistula, stricture, and esophageal perforation, which might warrant surgery.

  17. Sialadenoma papilliferum: A rare case report and review of literature

    Directory of Open Access Journals (Sweden)

    S Sunil

    2017-01-01

    Full Text Available Sialadenoma papilliferum (SP classified under the ductal papillomas by the WHO is a rare benign tumor of minor salivary glands. It is a rare lesion of salivary glands predominantly affecting the minor glands. It has characteristic exophytic and endophytic clinical growth pattern. Histopathologically, it is characterized by papillary projections supported by fibrovascular connective tissue core and infiltrated with mixed inflammatory cells. The ductal lining epithelium of double-layered cells of luminal layer of tall columnar cells and a basilar layer of small cuboidal cells shows additional papillary projections into the lumen. We report a case of SP of mid palate.

  18. Primitive neuroectodermal tumor presenting as a presacral mass: A rare case report with review of literature

    Directory of Open Access Journals (Sweden)

    Pradnya S Bhadarge

    2017-01-01

    Full Text Available Primitive neuroectodermal tumors (PNETs are a group of highly malignant small round cell tumor (SRCT of neuroectodermal origin. They exhibit a great diversity in their clinical manifestations and pathologic similarities with other SRCTs. PNET commonly occurs in the central nervous system, head and neck region, paravertebral region, pelvis, and lower extremities. PNET presenting as a presacral mass is very rare. We present a case of 65-year-old female patient presented with a mass in the abdomen. Exploratory laparotomy with excision of mass was carried out. Histopathology revealed the diagnosis of PNET. The rarity of PNET at presacral region prompted the description of this case.

  19. Ectopic Compound Odontoma in the Buccal Mucosa: Report of a Rare Case

    Directory of Open Access Journals (Sweden)

    Aparna Venigalla

    2015-01-01

    Full Text Available Eruption of tooth into extraosseous locations is an extremely rare condition. We report a case of a six-year-old girl child with tooth-like structure erupting from the right buccal mucosa. Clinical, radiographic, and histopathologic examination suggested the diagnosis of compound odontoma. Very few cases have been reported so far, where tooth has been located completely in the soft tissue and a variety of names have been used for that condition. A brief review of the literature and the ambiguity in naming the situation is discussed.

  20. Biliary Dyskinesia as a Rare Presentation of Metastatic Breast Carcinoma of the Gallbladder: A Case Report

    Directory of Open Access Journals (Sweden)

    A. Markelov

    2011-01-01

    Full Text Available Background. Breast carcinoma is the most common malignancy in women worldwide. It is most commonly associated with metastases to the liver, lung, bone, and the brain. Invasive lobular carcinoma is a less common pathology with slightly higher metastases to the upper gastrointestinal tract. Invasive lobular carcinoma metastasis to the gallbladder is extremely rare. Method. In this paper we are presenting a case of a 67-year-old female with metastases of invasive lobular breast cancer to the gallbladder six years after her therapy. Conclusion. This case clearly signifies the nature of the micrometastatic foci of the invasive lobular carcinoma even many years after a successful treatment.

  1. Low-grade Myxofibrosarcoma in the Mandible: A Rare Case Report

    Directory of Open Access Journals (Sweden)

    Neda Kargahi

    2017-04-01

    Full Text Available Myxofibrosarcoma is a malignant neoplasm of connective tissue origin commonly found in the extremities. It is very rare in the head and neck regions. Only 25 cases of myxofibrosarcoma have been reported in the head and neck regions until 2014. Here we report a 61-year-old male with this neoplasm in the lower border of his mandible. During a two-year follow-up, this case recurred four times despite a complete resection. This study suggested combined surgical and adjuvant radiotherapy for unresectable lesions and tumors with positive margins to prevent recurrence and risk of progression.

  2. Janiceps conjoined twins with extreme asymmetry: case report with complete autopsy and histopathologic findings.

    Science.gov (United States)

    Kastenbaum, Hannah A; McPherson, Elizabeth W; Murdoch, Geoffrey H; Ozolek, John A

    2009-01-01

    Conjoined twinning is a rare form of twinning, in which 2 bodies are attached, and is classified according to the anatomic place of attachment. An extremely rare form of conjoined twinning is janiceps conjoined twinning, in which 2 faces are attached but oriented in opposite directions. In this report, we present an unusual and difficult-to-classify case of conjoined male twins with partial duplication of craniofacial, upper oropharyngeal, and cardiac organs. We believe this to be one of the few reported cases of janiceps asymmetrus. We describe in detail the gross and microscopic pathology and offer some insights into the possible embryogenesis and distinction from the other rare form of conjoined twinning with facial duplication, diprosopus.

  3. Congenital double-double lip: A rare case report

    Directory of Open Access Journals (Sweden)

    Swati Phore

    2017-01-01

    Full Text Available A double lip is a rare anomaly characterized by a horizontal fold of redundant mucosal tissue that is situated proximal to the vermilion border. It may be either congenital or acquired and has no gender or race predilection. It occurs most often in the upper lip, although both upper and lower lips are occasionally involved. Surgical intervention (simple excision produces good functional and cosmetic results. In this report, a case of a nonsyndromic congenital maxillary double upper lip and lower lip, both are described. Double lip is of special interest in dental profession as a dental surgeon is normally the first one to diagnose this rare and uncommon condition. Authors hereby discuss a very rare condition in which both upper and lower lips are involved.

  4. Gastric heterotopia of the anus: Report of two rare cases and review of the literature

    Directory of Open Access Journals (Sweden)

    Rifat Mannan Abul Ala

    2008-04-01

    Full Text Available Heterotopic gastric mucosa is an extremely rare occurrence in the anorectal region, with 41 reported cases till date. Of these, in only nine cases the heterotopic tissue has been found to be located within 2 cm of the dentate line. We report two cases of gastric heterotopia in the anus - one, in a 55-year-old man; and the other, in a 35-year-old woman. Sigmoidoscopy showed presence of a single sessile anal polyp in the first patient and hemorrhoid in the other. Pathologic examination of the biopsy specimens revealed fundic-type gastric mucosa in both the cases. Both the patients had complete resolution of symptoms after the excision. We present these cases to highlight the significance of recognizing this unusual histologic entity. To the best of our knowledge, the second case represents the first reported description of gastric heterotopia in association with hemorrhoid.

  5. Coexistence of morphea and granuloma annulare: a rare case report

    Directory of Open Access Journals (Sweden)

    Şenay Ağırgöl

    2017-11-01

    Full Text Available ABSTRACT CONTEXT: Localized scleroderma (morphea is characterized by fibrosis of skin and subcutaneous tissue. Granuloma annulare is a relatively common disease that is characterized by dermal papules and arciform plaques. CASE REPORT: Here, we present the case of a 42-year-old woman who developed granuloma annulare on the dorsum of her feet and abdominal region, and morphea on the anterior side of her lower limbs. We also discuss the etiological and pathogenetic processes that may cause the rare coexistence of these two diseases. CONCLUSION: Only a few cases in the literature have described coexistence of morphea and granuloma annulare.

  6. Intracystic papillary carcinoma in a male as a rare presentation of breast cancer: a case report and literature review.

    LENUS (Irish Health Repository)

    Romics, Laszlo

    2009-01-01

    The term "intracystic papillary ductal carcinoma in situ" has recently changed and is now more appropriately referred to "intracystic papillary carcinoma". Intracystic papillary carcinoma in men is an extremely rare disease with only a few case presentations published in the literature so far.

  7. Cor triatriatum dexter, a very rare congenital anomaly presented in a complex pathological context – case presentation

    Directory of Open Access Journals (Sweden)

    Gozar Liliana

    2014-08-01

    Full Text Available Background Cor triatriatum dexter is an extremely rare congenital anomaly, and in most cases, without hemodynamic significance. Congenital cytomegalovirus infection is the most common viral infection, the majority of infected infants being asymptomatic and only 5-15% being symptomatic in the neonatal period.

  8. Extreme ultraviolet fluorescence spectroscopy of pure and core-shell rare gas clusters at FLASH

    Energy Technology Data Exchange (ETDEWEB)

    Schroedter, Lasse

    2013-08-15

    The interaction of rare gas clusters with short-wavelength radiation of free-electron lasers (FELs) has been studied extensively over the last decade by means of electron and ion time-of-flight spectroscopy. This thesis describes the design and construction of a fluorescence spectrometer for the extreme ultraviolet (XUV) spectral range and discusses the cluster experiments performed at FLASH, the Free-electron LAser in Hamburg. Fluorescence of xenon and of argon clusters was studied, both in dependence on the FEL pulse intensity and on the cluster size. The FEL wavelength was set to the giant 4d-resonance of xenon at 13.5 nm and the FEL pulse intensity reached peak values of 2.7.10{sup 15} W/cm{sup 2}. For xenon clusters, charge states of at least 11+ were identified. For argon, charge states up to 7+ were detected. The cluster-size dependent study revealed a decrease of the fluorescence yield per atom with increasing cluster size. This decrease is explained with the help of a geometric model. It assumes that virtually the entire fluorescence yield stems from shells of ions on the cluster surface, whereas ions in the cluster core predominantly recombine non-radiatively with electrons. However, the detailed analysis of fluorescence spectra from clusters consisting of a core of Xe atoms and a surrounding shell of argon atoms shows that, in fact, a small fraction of the fluorescence signal comes from Xe ions in the cluster core. Interestingly, these ions are as highly charged as the ions in the shells of a pure Xe cluster. This result goes beyond the current understanding of charge and energy transfer processes in these systems and points toward the observation of ultrafast charging dynamics in a time window where mass spectrometry is inherently blind. (orig.)

  9. Extreme ultraviolet fluorescence spectroscopy of pure and core-shell rare gas clusters at FLASH

    International Nuclear Information System (INIS)

    Schroedter, Lasse

    2013-08-01

    The interaction of rare gas clusters with short-wavelength radiation of free-electron lasers (FELs) has been studied extensively over the last decade by means of electron and ion time-of-flight spectroscopy. This thesis describes the design and construction of a fluorescence spectrometer for the extreme ultraviolet (XUV) spectral range and discusses the cluster experiments performed at FLASH, the Free-electron LAser in Hamburg. Fluorescence of xenon and of argon clusters was studied, both in dependence on the FEL pulse intensity and on the cluster size. The FEL wavelength was set to the giant 4d-resonance of xenon at 13.5 nm and the FEL pulse intensity reached peak values of 2.7.10 15 W/cm 2 . For xenon clusters, charge states of at least 11+ were identified. For argon, charge states up to 7+ were detected. The cluster-size dependent study revealed a decrease of the fluorescence yield per atom with increasing cluster size. This decrease is explained with the help of a geometric model. It assumes that virtually the entire fluorescence yield stems from shells of ions on the cluster surface, whereas ions in the cluster core predominantly recombine non-radiatively with electrons. However, the detailed analysis of fluorescence spectra from clusters consisting of a core of Xe atoms and a surrounding shell of argon atoms shows that, in fact, a small fraction of the fluorescence signal comes from Xe ions in the cluster core. Interestingly, these ions are as highly charged as the ions in the shells of a pure Xe cluster. This result goes beyond the current understanding of charge and energy transfer processes in these systems and points toward the observation of ultrafast charging dynamics in a time window where mass spectrometry is inherently blind. (orig.)

  10. A rare case of 46,XX gonadal dysgenesis and Mayer-Rokitansky-Kuster-Hauser syndrome.

    Science.gov (United States)

    Manne, Sriharibabu; Veeraabhinav, C H; Jetti, Mounica; Himabindu, Yalamanchali; Donthu, Kiranmai; Badireddy, Mutyalarayudu

    2016-01-01

    46,XX gonadal dysgenesis is a rare genetically heterogeneous disorder characterized by underdeveloped ovaries with consequent, impuberism, primary amenorrhea, and hypergonadotropic hypogonadism. Mullerian agenesis or Mayer-Rokitansky-Kuster-Hauser (MRKH) syndrome is characterized by congenital aplasia of the uterus and the upper part (2/3) of the vagina in a woman with normal development of secondary sexual characteristics and a normal 46,XX karyotype. The phenotypic manifestations of MRKH syndrome may sometimes overlap with various other syndromes and require accurate delineation. The coexistence of both these disorders is extremely rare. Here, we report a case of 46,XX gonadal dysgenesis and MRKH syndrome with anatomically dispersed congenital anomalies unique among reported cases.

  11. Cystic fibroadenoma: report of a rare case with review of literature.

    Science.gov (United States)

    Bhat, Amoolya; Vijaya, C; Gowda, V S Shankare

    2015-01-01

    Fibroadenomas with a predominant cystic change are called cystic fibroadenomas. These are extremely rare forms of fibroadenomas and only one case has been reported so far. They are classified under the category of complex fibroadenomas. Complex fibroadenomas are a rare variant of fibroadenomas occurring in elderly females. They are characterized by presence of one of the complex features along with the usual patterns of fibroadenoma such as cysts more than 3 mm, papillary apocrine metaplasia, or sclerosing adenosis. Patients with these lesions have higher chances of developing carcinoma of breast. We present a case of 35 years old lady with a freely mobile mass in the left breast diagnosed as cystic fibroadenoma after thorough histopathological examination of the lesion.

  12. A rare case of Weil's disease with alveolar haemorrhage

    Directory of Open Access Journals (Sweden)

    Abhiram Chakrabarti

    2014-05-01

    Full Text Available Leptospirosis, a disease of protean manifestations occurs sporadically throughout the year with a peak seasonal incidence during the rainy season mimicking other febrile viral illness. In the rare case, the disease leads to renal and hepatic involvement with hemorrhage which may be associated with multisystem organ dysfunction in form of pulmonary, cardiac and central nervous system, when it is known as Weil's disease. Rarely haemorrhagic manifestations are assosciated. Early diagnosis is important as sometimes the disease may be life threatening. Proper antibiotics results in dramatic improvement. We hereby presented a case that had clinical features of Weil's disease with cough, dyspnoea and haemoptysis. Leptospirosis was detected on ELISA testing. Patient was cured rapidly with antibiotics.

  13. A rare case of Weil's disease with alveolar haemorrhage.

    Science.gov (United States)

    Chakrabarti, Abhiram; Nandy, Manab; Pal, Dipankar; Mallik, Sudesna

    2014-05-01

    Leptospirosis, a disease of protean manifestations occurs sporadically throughout the year with a peak seasonal incidence during the rainy season mimicking other febrile viral illness. In the rare case, the disease leads to renal and hepatic involvement with hemorrhage which may be associated with multisystem organ dysfunction in form of pulmonary, cardiac and central nervous system, when it is known as Weil's disease. Rarely haemorrhagic manifestations are assosciated. Early diagnosis is important as sometimes the disease may be life threatening. Proper antibiotics results in dramatic improvement. We hereby presented a case that had clinical features of Weil's disease with cough, dyspnoea and haemoptysis. Leptospirosis was detected on ELISA testing. Patient was cured rapidly with antibiotics.

  14. A Rare Case Presentation of a Perforated Giant Sigmoid Diverticulum

    Directory of Open Access Journals (Sweden)

    Jennifer C. Kam

    2013-01-01

    Full Text Available Giant sigmoid diverticulum (GSD is a rare complication of diverticulosis. These lesions arise from herniations of the mucosa through the muscle wall which progressively enlarge with colonic gas to become large air-filled cysts evident on plain X-ray and CT scans. We present a rare case of a 72-year-old female presenting with abdominal distention, abdominal tenderness, and fever who developed a type 1 giant sigmoid diverticulum (pseudodiverticulum that subsequently formed an intra-abdominal abscess and an accompanying type 2 diverticulum as well. The patient was treated with surgical resection of the diverticulum with a primary anastomosis and abscess drainage. The patient’s postoperative course was uneventful. This case helps to support the need for the consideration of GSD in patients aged 60 and older with a history of diverticulosis and presenting with abdominal discomfort and distension.

  15. Rare Case of Polymicrobial Keratitis With Balantidium coli.

    Science.gov (United States)

    Hazarika, Manali; Pai H, Vijaya; Khanna, Vinay; Reddy, Harish; Tilak, Kriti; Chawla, Kiran

    2016-12-01

    To report a rare case of polymicrobial keratitis due to Balantidium coli and gram-negative bacteria, Pseudomonas aeruginosa and Klebsiella pneumoniae, in a soft contact lens (CL) wearer. We report a case of CL-related keratitis due to B. coli, P. aeruginosa, and K. pneumoniae. The culture of the corneal scrapings, the CL cleaning solution, and the CL revealed the growth of a rare ciliated parasite, B. coli, along with gram-negative bacteria, namely, P. aeruginosa and K. pneumoniae. The patient was successfully treated with topical broad-spectrum antibiotics and intravenous metronidazole. Polymicrobial keratitis has seldom been reported with B. coli as the causative agent. CL wear can be a risk factor for this infection. Treatment with topical antibiotics may not suffice, and the intravenous route of antiprotozoal drugs may be a useful adjunct. Increasing awareness, early diagnosis, and treatment may improve the final visual outcome.

  16. Violence is Rare in Autism: When It Does Occur, Is It Sometimes Extreme?

    Science.gov (United States)

    Allely, C S; Wilson, P; Minnis, H; Thompson, L; Yaksic, E; Gillberg, C

    2017-01-02

    A small body of literature has suggested that, rather than being more likely to engage in offending or violent behavior, individuals with autism spectrum disorder (ASD) may actually have an increased risk of being the victim rather than the perpetrator of violence (Sobsey, Wells, Lucardie, & Mansell, 1995 ). There is no evidence that people with ASD are more violent than those without ASD (Im, 2016). There is nevertheless a small subgroup of individuals with ASD who exhibit violent offending behaviours and our previous work has suggested that other factors, such as adverse childhood experiences, might be important in this subgroup (Allely, Minnis, Thompson, Wilson, & Gillberg, 2014 ). Fitzgerald ( 2015 ) highlights that school shootings and mass killings are not uncommonly carried out by individuals with neurodevelopmental disorders, with frequent evidence of warning indicators. The aim of the present review is to investigate this in more detail using the 73 mass shooting events identified by Mother Jones (motherjones.com) in their database for potential ASD features. There are 73 mass shooting events but there are two events where there is a pair of shooters which meant that 75 mass shooter cases were investigated. This exercise tentatively suggests evidence of ASD in six of 75 included cases (8%) which is about eight times higher when compared to the prevalence of ASD found in the general population worldwide (motherjones.com). The 8% figure for individuals with ASD involved mass killings is a conservative estimate. In addition to the six cases which provide the 8% figure, there were 16 other cases with some indication of ASD. Crucially, ASD may influence, but does not cause, an individual to commit extreme violent acts such as a mass shooting episode.

  17. Hereditary hypohidrotic ectodermal dysplasia: report of a rare case.

    Science.gov (United States)

    Paramkusam, Geetha; Meduri, Venkateswarlu; Nadendla, Lakshmi Kavitha; Shetty, Namratha

    2013-09-01

    Hereditary Hypohidrotic Ectodermal Dysplasia (HHED), an X-linked, recessive, Mendelian character, is seen usually in males and it is inherited through female carriers. It is characterised by congenital dysplasia of one or more ectodermal structures and it is manifested by hypohidrosis, hypotrichosis and hypodontia. It results from abnormal morphogenesis of cutaneous and oral embryonic ectoderm. Here, we are presenting a rare case of HHED in a 19 year female with classic features of this condition.

  18. Bilateral double-headed condyles: A rare case report

    Directory of Open Access Journals (Sweden)

    Swati Phore

    2018-01-01

    Full Text Available Bifid mandibular condyle is characterized by the duplicity of the head of the mandibular condyle, so the name double-headed condyle. It is usually diagnosed on routine radiographic examination and is described as a rare entity. Usually, bifid condyle is an incidental finding and its etiology is controversial, with no predilection for sex or ethnic background. Herein, we report a case of bilateral bifid condyles.

  19. Sirenomelia: A Rare Case of Foetal Congenital Anomaly

    OpenAIRE

    Dharmraj, Meena; Gaur, Sumitra

    2012-01-01

    Sirenomelia, alternatively known as ?mermaid syndrome? is a very rare congenital deformity in which the legs are fused together, giving them the appearance of the tail of a mermaid?. Other birth defects are always associated with sirenomelia, most commonly abnormalities of the kidneys, large intestines, and genitalia. The present case is a one of sirenomelia associated with an absent right kidney, mild left hydronephrosis, single umbilical artery, and severe oligohydramnios. We discuss the fi...

  20. Sirenomelia: a rare case of foetal congenital anomaly.

    Science.gov (United States)

    Dharmraj, Meena; Gaur, Sumitra

    2012-10-01

    Sirenomelia, alternatively known as 'mermaid syndrome' is a very rare congenital deformity in which the legs are fused together, giving them the appearance of the tail of a mermaid'. Other birth defects are always associated with sirenomelia, most commonly abnormalities of the kidneys, large intestines, and genitalia. The present case is a one of sirenomelia associated with an absent right kidney, mild left hydronephrosis, single umbilical artery, and severe oligohydramnios. We discuss the findings, relative to the present literature and related etiopathogenesis.

  1. Pyknodysostosis: report of a rare case with review of literature

    OpenAIRE

    Ramaiah, Kiran Kumar Kotagudda; George, Giju Baby; Padiyath, Sheeba; Sethuraman, Rupak; Cherian, Babu

    2011-01-01

    Pyknodysostosis is a rare autosomal recessive disorder characterized by the post natal onset of short limbs, short stature, and generalized hyperostosis along with acro-osteolysis with sclerosis of the terminal phalanges, a feature that is considered essentially pathognomonic. Other features include persistence of fontanelles, delayed closure of sutures, wormian bones, absence of frontal sinuses, and obtuse mandibular gonial angle with relative mandibular prognathism. We report a case of 17-y...

  2. Lumbosacral arachnoid cyst with tethered cord: A rare case report

    Directory of Open Access Journals (Sweden)

    S K Jain

    2012-01-01

    Full Text Available Arachnoid cysts are cerebrospinal fluid collections in the spine that can present with neurological symptoms or be discovered accidentally. Intradural location of such cysts especially in the lumbosacral region is relatively rare. The association of such cysts with other congenital anomalies such as tethered cord lends evidence to the developmental origin of arachnoid cysts. We report a case of lumbosacral arachnoid cyst with tethered cord in a 6-year-old male child and discuss the etiopathogenesis and management options.

  3. Humeral Metastasis from Cervical Cancer: A Rare Case Report

    OpenAIRE

    Sonia Chhabra; KanikaTaneja; Megha Ralli; Sunita Singh; Aditi Arora; Sohrab Arora; Pansi Gupta

    2015-01-01

    Long bone metastasis in cervical cancer is a rare presentation generally seen in the lumbar column or ribs. The reported rates of bone metastases are between 15%-29%. It is associated with poor prognosis. Bone scan and magnetic resonance imaging are useful techniques for diagnosis. In this case report, a 32-year old female with a previous history of cervical carcinoma FIGO stage IIIA presented with severe pain and swelling in her right humerus. X-ray and magnetic resonance imag...

  4. Antibody-Mediated Extreme Insulin Resistance: A Report of Three Cases.

    Science.gov (United States)

    Kim, Han Na; Fesseha, Betiel; Anzaldi, Laura; Tsao, Allison; Galiatsatos, Panagis; Sidhaye, Aniket

    2018-01-01

    Type 2 diabetes mellitus is characterized by relative insulin deficiency and insulin resistance. Features suggesting severe insulin resistance include acanthosis nigricans, hyperandrogenism, weight loss, and recurrent hospital admissions for diabetic ketoacidosis. In rare circumstances, hyperglycemia persists despite administration of massive doses of insulin. In these cases, it is important to consider autoimmune etiologies for insulin resistance, such as type B insulin resistance and insulin antibody-mediated extreme insulin resistance, which carry high morbidity and mortality if untreated. Encouragingly, immunomodulatory regimens have recently been published that induce remission at high rates. We describe 3 cases of extreme insulin resistance mediated by anti-insulin receptor autoantibodies or insulin autoantibodies. All cases were effectively treated with an immunomodulatory regimen. Although cases of extreme insulin resistance are rare, it is important to be aware of autoimmune causes, recognize suggestive signs and symptoms, and pursue appropriate diagnostic evaluation. Prompt treatment with immunomodulators is key to restoring euglycemia in patients with autoimmune etiologies of insulin resistance. Copyright © 2018 Elsevier Inc. All rights reserved.

  5. CASE REPORT Imaging features of a rare case of scapuloiliac ...

    African Journals Online (AJOL)

    SA JOURNAL OF RADIOLOGY • March 2011 7. CASE REPORT which, along with the surrounding skeletal muscles, seemed to be supporting the well-formed femoral heads. Discussion. Scapulo-iliac dysostosis (pelvis-shoulder dysplasia, Kosenow syndrome) is an uncommon hereditary skeletal dysplasia. It is classified as ...

  6. Rare Case of Vasculitis of the Hepatic Artery.

    Science.gov (United States)

    Mali, Padmavathi; Muduganti, Sudheer R; Goldberg, Jerry

    2015-12-01

    Vasculitis is an accumulation of inflammatory leucocytes in the blood vessels with reactive damage to mural structures. Isolated vasculitis of the gastrointestinal tract without systemic involvement is rare. We report a unique case of a female patient who presented with abdominal pain, and was found, on serology, to have elevated inflammatory markers without autoantibodies. A computed tomography scan of the abdomen and pelvis was suggestive of vasculitis of the hepatic artery. To the best of our knowledge, this is the first case, to date, of vasculitis of hepatic artery. © 2015 Marshfield Clinic.

  7. Cervical Paraganglioma Mimicking Thyroid Nodule: A Rare Clinical Case

    Directory of Open Access Journals (Sweden)

    Berna İmge Aydoğan

    2016-01-01

    Full Text Available Objective. Paraganglioma is a rare neuroendocrine tumor. When it is located in the neck, it is commonly misdiagnosed as other thyroid neoplasms. Case Report. We report a case of cervical paraganglioma in a 55-year-old female. Patient was admitted to our clinic with goiter and neck pain. Thyroid ultrasonography revealed a 20 mm solitary, heterogeneous nodule located in the upper pole of left thyroid lobe. Fine needle aspiration cytology was nondiagnostic. She underwent left lobectomy and histopathology showed paraganglioma. Discussion. Cervical paragangliomas should be considered in the differential diagnosis of thyroid nodules.

  8. A rare case of short stature: Say Meyer syndrome

    OpenAIRE

    Karthik, T. S.; Prasad, N. Rajendra; Rani, P. Radha; Maheshwari, Rushikesh; Reddy, P. Amaresh; Chakradhar, B. V. S.; Menon, Bindu

    2013-01-01

    Introduction: Say Meyer syndrome is rare X linked condition characterized by developmental delay, short stature and metopic suture synostosis. We are reporting a case of Say Meyer syndrome presented to our hospital for short stature and developmental delay at age 3½ years. Case Report: A 3½-year-old boy presented to our hospital for decreased growth velocity from the age of 1 year. History revealed the boy had a birth weight of 2.3 kg, had an episode of seizures in the neonatal period. He was...

  9. A RARE CASE OF THIAZIDE-INDUCED BENIGN GYNAECOMASTIA

    Directory of Open Access Journals (Sweden)

    Sandeep Yamsani

    2017-02-01

    Full Text Available BACKGROUND Gynaecomastia is a common disorder of the endocrine system in which there is a noncancerous enlargement of the male breast. The incidence of drug-induced gynaecomastia is about 4-10%. One among them are the thiazide group of diuretics, which result in gynaecomastia by inhibition of androgen synthesis. The present case study is about a 75-year-old patient who is a known case of diabetes and hypertension with primary complaints of bilateral breast enlargement diagnosed with a rare presentation of hydrochlorothiazide adverse drug reaction gynaecomastia.¹

  10. Case report of a rare dermatosis in pregnancy: impetigo herpetiformis.

    Science.gov (United States)

    Ulubay, Mustafa; Keskin, Uğur; Fidan, Ulas; Çiçek, Ali Fuat; Çalışkan, Ercan; Karaca, Rıza Efendi; Fıratlıgil, Fahri Burçin; Ergün, Ali

    2015-02-01

    Impetigo herpetiformis (IH) is a very rare type of dermatosis seen in pregnancy. According to the published work, IH during pregnancy is associated with the risk of stillbirth, and obstetric management in such cases is very important. Early recognition is important to reduce both maternal and fetal morbidity. We present a case of IH resistant to corticosteroid therapy in a 27-year-old pregnant woman where the pregnancy was terminated by the induction of labor. © 2014 The Authors. Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology.

  11. A rare case of acquired methemoglobinemia associated with alkaptonuria.

    Science.gov (United States)

    Isa, Yasuki; Nihei, Shun-ichi; Irifukuhama, Yuna; Ikeda, Tomoya; Matsumoto, Hiroyuki; Nagata, Keiji; Harayama, Nobuya; Aibara, Keiji; Kamochi, Masayuki

    2014-01-01

    We herein present a rare case of acquired methemoglobinemia associated with alkaptonuria. Alkaptonuria is a congenital error of metabolism caused by the deficiency of homogentisic acid oxidase, which subsequently results in the accumulation of homogentisic acid (HGA) in body tissues. As renal dysfunction progresses, the level of HGA excretion in the urine decreases and the blood concentration of HGA increases. HGA oxidizes oxyhemoglobin to methemoglobin, which can induce multiple organ failure accompanied by tissue hypoxia, intravascular hemolysis and metabolic acidosis. The mortality of this disease is high when alkaptonuria is associated with the presence of methemoglobinemia; therefore, treatment should be carefully planned in such cases.

  12. Primary peritoneal serous carcinoma: A rare case and palliative approach

    Directory of Open Access Journals (Sweden)

    Viral M Bhanvadia

    2014-01-01

    Full Text Available Primary peritoneal serous carcinoma (PPSC is a rare primary malignancy that diffusely involves the peritoneum, indistinguishable clinically and histopathologically from primary serous ovarian carcinoma. The origin of PPSC has not been well characterized. Here we present a case of PPSC diagnosed in ultrasonography-guided fine needle aspiration cytology (FNAC in a 76- old female presenting with ascites, abdominal pain, distension and constipation. PPSC is an unusual tumour but cytomorphology is distinctive enough to diagnose preoperatively. In the case report hereby described PPSC is an inoperable malignancy, hence chemotherapy and palliative care are the only offered treatment.

  13. Congenital insensitivity to pain: Case report of a rare entity

    Directory of Open Access Journals (Sweden)

    Swati Dahiya

    2018-01-01

    Full Text Available Hereditary sensory and autonomic neuropathies (HSANs are a group of disorders characterized by insensitivity to noxious stimuli and autonomic dysfunction, associated with pathological abnormalities of the peripheral nerves. Five types of HSAN have been reported in literature, out of which Type V known as congenital insensitivity to pain (CIP is a rare autosomal recessive condition. Self-mutilation is an invariable feature of this disorder, involving the teeth and orofacial structures. This case report describes a case of a 6-year-old girl with CIP brought by her parents for prostheses to replace her self-extracted primary teeth.

  14. Solitary Fibrous Tumor of Retromolar Pad; a Rare Challenging Case

    Science.gov (United States)

    Lotfi, Ali; Mokhtari, Sepideh; Moshref, Mohammad; Shahla, Maryam; Atarbashi Moghadam, Saede

    2017-01-01

    Solitary fibrous tumor has a wide spectrum of histopathologic features and many tumors show similar microscopic features. This similarity poses diagnostic challenges to the pathologists and immunohistochemical analysis is required in many cases. Moreover, it is a rare entity in orofacial region which consequently would make its diagnosis more challenging in oral cavity. The knowledge of various microscopic patterns of this tumor contributes to a proper diagnosis and prevents unnecessary treatment. This study reports a case of solitary fibrous tumor in the retromolar pad area and discusses its various histological features and differential diagnoses. PMID:28620640

  15. Unusually large erupted complex odontoma: A rare case report

    Energy Technology Data Exchange (ETDEWEB)

    Bagewadi, Shivanand B.; Kukreja, Rahul; Suma, Gundareddy N.; Yadav, Bhawn; Sharma, Havi [Dept. of Oral Medicine and Radiology, ITS Centre for Dental Studies and Research, Murad Nagar (India)

    2015-03-15

    Odontomas are nonaggressive, hamartomatous developmental malformations composed of mature tooth substances and may be compound or complex depending on the extent of morphodifferentiation or on their resemblance to normal teeth. Among them, complex odontomas are relatively rare tumors. They are usually asymptomatic in nature. Occasionally, these tumors become large, causing bone expansion followed by facial asymmetry. Odontoma eruptions are uncommon, and thus far, very few cases of erupted complex odontomas have been reported in the literature. Here, we report the case of an unusually large, painless, complex odontoma located in the right posterior mandible.

  16. Gastric Volvulus: A Rare Entity Case Report and Literature Review

    Science.gov (United States)

    Akhtar, Aisha; Sheikh, Abdul Ahad E; Sheikh, Abu Baker; Perisetti, Abhilash

    2018-01-01

    Gastric volvulus is a rare entity defined as an abnormal rotation of the stomach around itself. It is a diagnosis of exclusion; the clinical index of suspicion is always low and is mostly diagnosed on imaging or on the surgery table. When it occurs, it is an emergency due to the risk of strangulation and consequent gangrene of the stomach. Mesentero-axial (MA) gastric volvuli constitute one-third of all cases. Here, we are present an interesting case of acute MA gastric volvulus diagnosed with imaging and treated subsequently. PMID:29755908

  17. Rapidly Growing Chondroid Syringoma of the External Auditory Canal: Report of a Rare Case

    Science.gov (United States)

    Vasileiadis, Ioannis; Kapetanakis, Stylianos; Petousis, Aristotelis; Karakostas, Euthimios; Simantirakis, Christos

    2011-01-01

    Introduction. Chondroid syrinoma of the external auditory canal is an extremely rare benign neoplasm representing the cutaneous counterpart of pleomorphic adenoma of salivary glands. Less than 35 cases have been reported in the international literature. Case Presentation. We report a case of a 34-year-old male in whom a rapidly growing, well-circumscribed tumor arising from the external auditory canal was presented. Otoscopy revealed a smooth, nontender lesion covered by normal skin that almost obstructs the external auditory meatus. MRI was performed to define the extension of the lesion. It confirmed the presence of a 1.5 × 0.8 cm T2 high-signal intensity lesion in the superior and posterior wall of EAC without signs of bone erosion. The patient underwent complete resection of the tumor. The diagnosis was confirmed by histopathologic examination. Conclusion. Although chondroid syringoma is extremely rare, it should always be considered in the differential diagnosis of an aural polyp. Chondroid syringomas are usually asymptomatic, slow-growing, single benign tumors in subcutaneous or intradermal location. In our case, the new information is that this benign tumor could present also as a rapidly growing lesion, arising the suspicion for malignancy. PMID:21941560

  18. Rapidly Growing Chondroid Syringoma of the External Auditory Canal: Report of a Rare Case

    Directory of Open Access Journals (Sweden)

    Ioannis Vasileiadis

    2011-01-01

    Full Text Available Introduction. Chondroid syrinoma of the external auditory canal is an extremely rare benign neoplasm representing the cutaneous counterpart of pleomorphic adenoma of salivary glands. Less than 35 cases have been reported in the international literature. Case Presentation. We report a case of a 34-year-old male in whom a rapidly growing, well-circumscribed tumor arising from the external auditory canal was presented. Otoscopy revealed a smooth, nontender lesion covered by normal skin that almost obstructs the external auditory meatus. MRI was performed to define the extension of the lesion. It confirmed the presence of a 1.5×0.8 cm T2 high-signal intensity lesion in the superior and posterior wall of EAC without signs of bone erosion. The patient underwent complete resection of the tumor. The diagnosis was confirmed by histopathologic examination. Conclusion. Although chondroid syringoma is extremely rare, it should always be considered in the differential diagnosis of an aural polyp. Chondroid syringomas are usually asymptomatic, slow-growing, single benign tumors in subcutaneous or intradermal location. In our case, the new information is that this benign tumor could present also as a rapidly growing lesion, arising the suspicion for malignancy.

  19. Congenital upper lip pit: A rare case report

    Directory of Open Access Journals (Sweden)

    Leena James

    2015-01-01

    Full Text Available One of the rarest developmental malformations of the lip is congenital lip pits. They are usually seen as bilateral depressions in the vermilion zone of the lip and occur on the paramedian portion of the vermilion border of the lip. They are extremely rare in the upper lip. Lip pits are due to the failure of complete union of embryonic lateral sulci of the lip/notching of lip at an early stage of development with fixation of tissues of the base of the notch. Lip pits have also been associated with a variety of other congenital disorders and other malformations. The clinical and pathologic picture and the therapeutic aspects of this condition are discussed in this paper.

  20. Dedifferentiated liposarcoma of the anterior mediastinum. A rare case

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    Harth, S.; Litzlbauer, H.D.; Behrens, C.B.; Roller, F.C.; Gamerdinger, U.; Burchert, D.; Krombach, G.A.

    2016-01-01

    Liposarcoma accounts for approximately 14 % of all malignant soft-tissue tumors, regardless of anatomical location (Kransdorf MJ et al. Imaging of soft tissue tumors. Philadelphia: Lippincott Williams and Wilkins, 2014). Primary mediastinal liposarcomas are rare. Liposarcoma is classified into four histologic subtypes: Myxoid/round cell, pleomorphic, atypical lipomatous tumor/well-differentiated liposarcoma and dedifferentiated liposarcoma. Dedifferentiated liposarcoma occurs most commonly in the retroperitoneum and in the soft tissues of the extremities. Like atypical lipomatous tumor/well-differentiated liposarcoma, it is characterized by amplification of MDM2 and CDK4 genes on chromosome 12 (Crago AM et al. Curr Opin Oncol 2011; 23: 373 - 378). Possible symptoms of mediastinal liposarcoma are dyspnea, wheezing, chest pain, cough, superior vena cava syndrome, and weight loss (Macchiarini P et al. Lancet Oncol 2004; 5: 107 - 118).

  1. Dedifferentiated liposarcoma of the anterior mediastinum. A rare case

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    Harth, S.; Litzlbauer, H.D.; Behrens, C.B.; Roller, F.C.; Gamerdinger, U.; Burchert, D.; Krombach, G.A.

    2016-01-15

    Liposarcoma accounts for approximately 14 % of all malignant soft-tissue tumors, regardless of anatomical location (Kransdorf MJ et al. Imaging of soft tissue tumors. Philadelphia: Lippincott Williams and Wilkins, 2014). Primary mediastinal liposarcomas are rare. Liposarcoma is classified into four histologic subtypes: Myxoid/round cell, pleomorphic, atypical lipomatous tumor/well-differentiated liposarcoma and dedifferentiated liposarcoma. Dedifferentiated liposarcoma occurs most commonly in the retroperitoneum and in the soft tissues of the extremities. Like atypical lipomatous tumor/well-differentiated liposarcoma, it is characterized by amplification of MDM2 and CDK4 genes on chromosome 12 (Crago AM et al. Curr Opin Oncol 2011; 23: 373 - 378). Possible symptoms of mediastinal liposarcoma are dyspnea, wheezing, chest pain, cough, superior vena cava syndrome, and weight loss (Macchiarini P et al. Lancet Oncol 2004; 5: 107 - 118).

  2. Peripheral soft tissue ewing's sarcoma: a rare case report

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    Farzana Shegufta

    2013-07-01

    Full Text Available A 22 years male patient presented with gradual left forearm swelling for 6 months. X ray forearm revealed large soft tissue swelling with tiny calcification and mild scalloping at inner aspect of ulna and ultrasonogram (USG revealed soft tissue mass having calcification and necrotic areas within and spectral Doppler showed arterial type of blood flow with no augmentation. Later computerized tomography (CT scan showed soft tissue mass with necrotic area and calcification with no bony involvement. Magnetic resonance imaging (MRI with contrast revealed a large heterogeneously enhancing lobulated mixed intensity lesion in antero-medial compartment of the left forearm involving flexor group of muscles causing displacement of fat plane. MRI and subsequent histopathology of the lesion revealed it as a rare soft tissue Ewing’s sarcoma / primitive neuroectodermal tumor (PNET in extremity. Ibrahim Med. Coll. J. 2013; 7(2: 43-46

  3. Case Report: A Rare Case Report of Frontal Lobe Syndrome

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    Morteza Nouri- Khajavi

    2003-04-01

    Full Text Available The frontal lobe syndrome is a permanent personality change disorder with characteristic clinical pictures, which followed by frontal lobes damage. Clinical picture include: Affective instability, recurrent aggressive behavior, impaired social judgment, apathy and undifferentiating or suspiciousness and paranoid ideations. According DSM-IV classification frontal lobe syndrome named personality change due to head trauma on Axis I. Herein we report a case of 46 years-old man, who has developed behavioral disturbances following head trauma, about 10 years ago. Main clinical figures in this case are apathy, avolition and, undifferentiating. Clinical pictures are constant during these 10 years. The diagnostic approach has been based on patient’s problems history which, has taken from his family, mental status examination, Neurological examination, Brain imaging and Neuropsychological assessments which related to frontal lobes function. Because of rarity & neglection due to mysterious function of frontal lobes, and also considering that personality change from previous level is prominent figure of this syndrome and also brain imaging findings, which compatible with clinical findings, with this aim, we have reported this case.

  4. A Rare Case Report of Subcutaneous Mycoses by Rhytidhysteron Rufulum

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    Yadav Sarita

    2017-04-01

    Full Text Available Phaeohyphomycosis is a term to describe cutaneous and systemic or disseminated mycoses caused by a variety of dematiaceous fungi. Rhytidhysteron rufulum is a poorly known, common, pantropical phaeoid fungal species, and are rare human pathogens. Here, we describe a clinical case report of subcutaneous phaeohyphomycosis; to the best of our knowledge, this is the fourth report of infection with the Rhytidhysteron rufulum. A 54-year-old male presented to medicine outpatient department with complaints of respiratory distress. The patient was diagnosed as a case of chronic bronchitis with acute exacerbation. On examination, a well-defined soft subcutaneous swelling was detected on anterior aspect of right leg. The molecular identification revealed its homology with Rhytidhysteron rufulum. The phaeohyphomycotic infections are increasingly reported globally. This case highlights the need of awareness and high degree of suspicion for fungal aetiology, so that an early and adequate treatment can be given in such cases.

  5. A rare mitochondrial disorder: Leigh sydrome - a case report

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    Shrikhande Dhananjay Y

    2010-09-01

    Full Text Available Abstract Leigh syndrome is a rare progressive neurodegenerative, mitochondrial disorder of childhood with only a few cases documented from India. The clinical presentation of Leigh syndrome is highly variable. However, in most cases it presents as a progressive neurological disease with motor and intellectual developmental delay and signs and symptoms of brain stem and/or basal ganglia involvement. Raised lactate levels in blood and/or cerebrospinal fluid is noted. It is the neuroimaging, mainly the Magnetic Resonance Imaging showing characteristic symmetrical necrotic lesions in the basal ganglia and/or brain stem that leads to the diagnosis. Here, we report a case of 7 months old female child presenting to us with status epilepticus, delayed developmental milestones and regression of the achieved milestones suspected to be a case of neurodegenerative disorder, which on MRI was diagnosed as Leigh syndrome.

  6. Rare vertebral metastasis in a case of Hereditary Paraganglioma

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    da Silva Manuel Eduardo

    2012-09-01

    Full Text Available Abstract Paragangliomas are rare tumours with a prevalence of 1/10000 to 1/30000. Tumors arising from the paraganglia are characteristically of low malignant potential. Vertebral metastases are exceedingly rare, and only isolated case reports have described them. The authors present the clinical course of a 47 years-old female patient with a familial paraganglioma [PGL] with vertebral metastastization, who underwent an intralesional tumor excision and corpectomy. Genetic screening demonstrated a new germinal frameshift mutation of the SDHB exon 6 [c.587-591DelC]. After surgery there was normalization of the analytical parameters and imagiologic screening. One year later she presented a new image in the the pedicle of T11 on the contralateral side of the surgical incision. She performed 2 treatments with MIBG and 1 cicle of radiotherapy that made the new lesion regress. Currently the patient does not present any clinical or analytical evidence of new metastasis. This case outlines the clinical course of a patient with a PGL syndrome for whom a rare vertebral metastasis was diagnosed. It highlights the importance of identifying patients with germline SDHB mutations, as these patients are at a high risk of developing malignant disease.

  7. Palliative radiation in primary squamous cell carcinoma of thyroid: A rare case report

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    Sushmita Ghoshal

    2013-01-01

    Full Text Available Primary squamous cell carcinoma of the thyroid is an extremely rare neoplasm with aggressive behavior. Until date, only around 60 cases have been reported in the literature. Primary treatment of the patient is radical surgery. With optimum treatment survival is not more than 6 months in this aggressive malignancy. However in our patient surgery it was not possible because of unresectability of the mass due to encroachment of major vessels. Hence, we have delivered radiotherapy alone, with which effective palliation could be achieved and patient is leading a good quality-of-life for last 1 year.

  8. Conceptualizing Rolling Motion through an Extreme Case Reasoning Approach

    Science.gov (United States)

    Hasovic, Elvedin; Mešic, Vanes; Erceg, Nataša

    2017-01-01

    In this paper we are going to show how learning about some counterintuitive aspects of rolling motion can be facilitated by combining the use of analogies with extreme case reasoning. Specifically, the intuitively comprehensible examples of "rolling" polygonal prisms are used as an analogical anchor that is supposed to help the students…

  9. Christ siemens touraine syndrome: A rare case report

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    N Retnakumari

    2016-01-01

    Full Text Available Christ-Siemens-Touraine (CST is a rare hereditary disorder of X-linked recessive trait, characterized by abnormal development of two or more structures or tissues of ectodermal origin. The common clinical findings include hypodontia, hypohydrosis, hypotrichosis, and onychodysplasia. Although hypodontia is common, anodontia is a rare feature. Most of the patients are suffering from social rejection and consequent psychological trauma because of the facial dysmorphism and absence of multiple teeth. Oral rehabilitation is of prime importance for such patients. This article presents a case in a 5½-year-old boy presenting with altered manifestations affecting almost all the ectodermal structures like skin, hair, nails, teeth, sebaceous glands, sweat glands, salivary glands, mammary glands, and tear glands. He also had complete anodontia and dry mouth. A multidisciplinary treatment was given to the patient with the collaboration of various health professionals. The child gained confidence and was relieved from the psychological impact following the prosthetic rehabilitation.

  10. Ankyloglossia with cleft lip: A rare case report

    Science.gov (United States)

    Jangid, Kritika; Alexander, Aurelian Jovita; Jayakumar, Nadathur Doraiswamy; Varghese, Sheeja; Ramani, Pratibha

    2015-01-01

    Ankyloglossia or tongue-tie is a congenital anomaly affecting the tongue, which is characterized by thick, short lingual frenulum. This condition causes many difficulties such as limited tongue protrusion, breastfeeding difficulties, speech impairment and lack of self-confidence. It is very rarely associated with any other congenital craniofacial disorders such as cleft lip, X-linked cleft palate, Van der Woude syndrome, Smith-Lemli-Opitz syndrome, Orofacial digital syndrome, Beckwith Weidman syndrome or Simpson-Golabi-Behmel syndrome. This article presents a rare case of ankyloglossia associated with cleft lip treated with diode laser in a 12-year-old Indian boy who had undergone surgical correction of associated cleft lip soon after birth. Correction of ankyloglossia at a young age would lead to enhanced phonetics, improved oral hygiene, and overall personality development. PMID:26941523

  11. Ankyloglossia with cleft lip: A rare case report

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    Kritika Jangid

    2015-01-01

    Full Text Available Ankyloglossia or tongue-tie is a congenital anomaly affecting the tongue, which is characterized by thick, short lingual frenulum. This condition causes many difficulties such as limited tongue protrusion, breastfeeding difficulties, speech impairment and lack of self-confidence. It is very rarely associated with any other congenital craniofacial disorders such as cleft lip, X-linked cleft palate, Van der Woude syndrome, Smith-Lemli-Opitz syndrome, Orofacial digital syndrome, Beckwith Weidman syndrome or Simpson-Golabi-Behmel syndrome. This article presents a rare case of ankyloglossia associated with cleft lip treated with diode laser in a 12-year-old Indian boy who had undergone surgical correction of associated cleft lip soon after birth. Correction of ankyloglossia at a young age would lead to enhanced phonetics, improved oral hygiene, and overall personality development.

  12. Tuberculous osteomyelitis affecting periodontium: A rare case report

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    Gaurav Bakutra

    2015-01-01

    Full Text Available Tuberculous lesions affecting periodontium are rare and seen as secondary infections localized to the soft tissues. With the advent of effective drug therapy, tuberculous lesions of the oral cavity have become rare. Involvement of the periodontium has seldomly been reported in the recent literature. We report a case of tuberculous osteomyelitis of mandible affecting periodontium leading to gingival recession and bone exposure in the mandibular premolar region in a 42-year-old female patient. The diagnosis was based on patient's medical and dental history, bacterial culture, clinical and radiographic examination, blood investigation, immunologic tests, histopathologic examination of the tissue specimen. Patient was already taking antitubercular chemotherapy prescribed by physician. Sequestrectomy and decortications were carried out to remove the affected bone. Healing was uneventful and there was no recurrence after 1½ year of follow-up. Antitubercular chemotherapy along with sequestrectomy and decortication are the treatment of choice for tuberculous osteomyelitic lesions affecting periodontium.

  13. Ocular side effect of tinidazole: A rare case report

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    Hina Kauser

    2014-01-01

    Full Text Available Ocular side effects in the form of punctate epithelial erosions with the use of tinidazole - a 5-nitroimidazole group of drugs is very rare. A 32-year-old male was prescribed tablet tinidazole for the treatment of amoebiasis but developed adverse effects in the form of blisters on both upper and lower lips with itching and burning sensation, itching and burning on penile and anal area associated with punctate epithelial erosions of cornea of both the eyes. All these are rare manifestations but punctate epithelial erosions of cornea has never been reported in the literature so far. Punctate epithelial erosions of cornea have not previously been reported and should be added to the list of complications of tinidazole. Hence, this case is being reported .

  14. Rare presentation of pancreatic schwannoma: a case report

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    Tofigh Arash

    2008-08-01

    Full Text Available Abstract Introduction Schwannoma is a rare tumor among pancreatic neoplasms. Schwannomas vary in size, and most of them are cystic, mimicking pancreatic cystic lesions. Generally, a definitive diagnosis is made at the time of histological analysis. The mainstay treatment is surgical resection. Case presentation We report an unusual presentation of pancreatic schwannoma with abdominal pain and several episodes of cholangitis in a 54-year-old Caucasian (Iranian man. The condition was not diagnosed pre-operatively and Whipple's procedure was performed. Conclusion Pancreatic schwannoma is an important clinical entity to include in the differential diagnosis of pancreatic lesions. Pre-operative diagnosis is difficult but computed tomographic findings may be helpful. The tumor may also have atypical and rare presentations, such as cholangitis and weight loss. For benign tumors, simple enucleation is usually adequate, whereas malignant tumors require standard oncological resection.

  15. Christ Siemens Touraine syndrome: A rare case report.

    Science.gov (United States)

    Retnakumari, N; Varghese, Manuja; Kannan, V P

    2016-01-01

    Christ-Siemens-Touraine (CST) is a rare hereditary disorder of X-linked recessive trait, characterized by abnormal development of two or more structures or tissues of ectodermal origin. The common clinical findings include hypodontia, hypohydrosis, hypotrichosis, and onychodysplasia. Although hypodontia is common, anodontia is a rare feature. Most of the patients are suffering from social rejection and consequent psychological trauma because of the facial dysmorphism and absence of multiple teeth. Oral rehabilitation is of prime importance for such patients. This article presents a case in a 5½-year-old boy presenting with altered manifestations affecting almost all the ectodermal structures like skin, hair, nails, teeth, sebaceous glands, sweat glands, salivary glands, mammary glands, and tear glands. He also had complete anodontia and dry mouth. A multidisciplinary treatment was given to the patient with the collaboration of various health professionals. The child gained confidence and was relieved from the psychological impact following the prosthetic rehabilitation.

  16. Multicystic mesothelioma--a rare case of ascites: case report.

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    Manuc, M; Lamatic, C; Pop, C; Dobrea, C; Becheanu, G; Grasu, M; Iosif, D; Diculescu, M

    2007-01-01

    We present the case of a 37-year-old male, admitted to our clinic with abdominal tenderness, right supraclavicular tumour, and ascites. The presence of ascites was incidentally reported 6 years before, but no other evaluation was done at that moment or during this period. Abdominal ultrasound and CT scan revealed moderate ascites, perivascular adenopathies, and multiple abdominal cystic lesions, while thoracic CT scan revealed the same lesions in mediastinum. Laboratory data were within normal limits, including the tumoral markers, and the tests for hydatid cysts. A biopsy from the right supraclavicular nodule was performed, and based on usual and immunohistochemical stains (calretinin, mesotheline, CK 5/6, CK 7, CK18 diffusely positive in mesothelial cells, and CEA -M, bcl-2 and vimentin negative), suggested the diagnosis of mesothelioma. Based on these results, the diagnosis of "multicystic mesothelioma" was made. The patient was referred for surgery.

  17. Ingested razor blades within the appendix: A rare case report

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    Jason Cui

    Full Text Available Introduction: Foreign body ingestion is a common clinical presentation with less than 1% of the cases requiring surgical intervention. In this report, we present a rare case of razor blades lodged in the appendix as a result of intentional ingestion. Presentation of case: A 25 year old male prisoner presented to our hospital with persistent right iliac fossa pain after razor blade ingestion. After 5 days of conservative management, there was no sign of transition on serial X-Rays. Laparoscopy with intraoperative image intensification confirmed the presence of the razor blades in the appendix and appendicectomy was subsequently performed without complications. Discussion: Most ingested objected with diameter less than 2.5 cm and length less than 6 cm can pass through the gastrointestinal tract spontaneously in less than one week. The entry of foreign objects into the appendix is thought to be due to relative low motility of the caecum, the dependent position of the appendix and the size of the appendiceal orifice. Radiographic localisation to the appendiceal lumen was complicated by metallic artefact, but was consistent with failure to transit. Appendicectomy was felt to be the safest mode of retrieval. Conclusion: Ingested foreign body lodged in the appendix is a rare event. Once the exact location is confirmed, a simple laparoscopic appendicectomy can be performed to facilitate the removal. Keywords: Appendicitis, Laparoscopy, Appendicectomy, Foreign body ingestion, Razor blades, Case report

  18. Lymphoepithelial carcinoma: a case report of a rare tumor of the larynx.

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    Hammas, Nawal; Benmansour, Najib; El Alami El Amine, Mohamed Nour-Dine; Chbani, Laila; El Fatemi, Hind

    2017-01-01

    Lymphoepithelial carcinoma is a tumor mostly diagnosed in the nasopharynx, but it has also been described in a variety of nonnasopharyngeal sites. It is extremely rare in the larynx and should be distinguished from squamous cell carcinoma. Therefore, it must be known by clinicians, pathologists and oncologists. In this case report, we discuss its etiopathogeny, its epidemiological, clinical, pathological and therapeutic aspects, and its outcome. An 81-year-old Morrocan man, smoker for 40 years, presented with a 1 year history of dysphonia, dyspnea and dysphagia. Laryngoscopy showed a mass occupying supraglottic, glottic and subglottic levels of the larynx. Cervico-thoracic computed tomography scan showed a laryngeal wall thickening with cervical lymphadenopathy. Laryngeal biopsy was performed. Microscopic analysis and immunohistochemistry confirmed the diagnosis of laryngeal lymphoepithelial carcinoma. Immunostaining for LMP1 was negative. Laryngeal lymphoepithelial carcinoma is an extremely rare and an aggressive tumor. It is rarely associated with the EBV. It must be regarded as a distinct entity. Radiotherapy is advisable as the unique therapy for local tumor. A correct diagnosis and a close collaboration between the pathologist and clinicians is mandatory for an optimal treatment strategy.

  19. Cerebrotendinous xanthomatosis (a rare lipid storage disorder): a case report.

    Science.gov (United States)

    Razi, Syed Mohd; Gupta, Abhinav Kumar; Gupta, Deepak Chand; Gutch, Manish; Gupta, Keshav Kumar; Usman, Syeda Iqra

    2016-04-19

    Cerebrotendinous xanthomatosis is a very rare autosomal recessive lipid storage disorder affecting bile acid biosynthesis. It is manifested by subtle neurological and non-neurological symptoms due to abnormal tissue lipid deposition. Diagnosis is usually delayed but early diagnosis and replacement therapy can prevent devastating neurological sequelae. We present a case of a 25-year-old Asian Indian woman who presented with gait difficulty, fusiform swellings of bilateral tendo-Achilles and infrapatellar tendons, along with history of bilateral cataract surgery 1 year earlier. The diagnosis was made on the basis of clinical, biochemical, imaging, and histopathological analysis and replacement therapy was started. The peculiarity of the present case is the absence of any neurological manifestations which are usually the early clues to the diagnosis of cerebrotendinous xanthomatosis. The present case report emphasizes the fact that early age bilateral cataracts along with bilateral tendo-Achilles xanthomas can be early pointers toward the diagnosis of cerebrotendinous xanthomatosis.

  20. Neuroblastoma in early childhood: A rare case report and review of literature

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    Ritesh R Kalaskar

    2016-01-01

    Full Text Available Neuroblastoma is an extremely rare pediatric neoplasm whose prognosis becomes poor and poor as the age advances. It can be sporadic or nonfamilial in origin. It is primarily a tumor of abdominal origin from where it metastasis to lymph nodes, liver, intracranial and orbital sites, and central nervous system. There is no standard dental treatment protocol for the management of neuroblastoma due to its poor survival rate and rarity. However, dental treatment may follow the protocol of preventive and restorative. Surgicals should be performed under supervision as it may trigger metastasis. We report a rare case of neuroblastoma in a 3-year-old child presenting classical oral manifestations such as bilateral palatal swelling, rolled border ulcer on the posterior part of hard palate adjacent to primary molars, and bilateral proptosis.

  1. A Case Report on Upper Extremity Pain of Cardiac Origin

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    Turgay Altınbilek

    2016-08-01

    Full Text Available Upper extremity pain can originate from the musculoskeletal system, or be a reflection of problems originating from various organs. Therefore, it is highly important to perform a detailed clinical evaluation on patients during differential diagnosis. In this case report, we present a 61 year-old male patient who was admitted with pain in both upper extremities and the upper back that presumed to be of cardiac origin following our clinical evaluations. The patient was referred to the cardiology department, where he was diagnosed with coronary heart disease. The patient’s complaints of pain were fully resolved through the application of an intracoronary stent.

  2. Spinal cord injury in Parkour sport (free running: a rare case report

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    Derakhshan Nima

    2014-06-01

    Full Text Available A 24-year-old male was transferred to the emergency department while being in the state of quadriplegia with a history of performing Parkour sport, which is also called double front flip. Neurological examination revealed that the patient’s muscle power was 0/5 at all extremities. The patient did not show any sense of light touch or pain in his extremities. In radiological studies, cervical spine X-ray and CT scan images showed C4-C5 subluxation with bilateral locked facets and spinal cord injury. The results of this very rare case study revealed that exercising Parkour sport without taking into account safety standards could result in irreversible injuries to the cervical spinal cord with fatal outcome. Key words: Spinal cord injuries; Cervical vertebrae; Athletic injuries

  3. A Rare Case of Pneumonia Caused by Shewanella putrefaciens

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    Rajshree Patel

    2012-01-01

    Full Text Available Shewanella putrefaciens is a gram-negative, nonfermentative, oxidase positive, motile bacillus that produces hydrogen sulphide. It is found widely in the nature especially in marine environments. In some very rare cases Shewanella putrefaciens can be a human pathogen. It can produce a wide variety of clinical syndromes including bacteremia as well as skin and soft tissue infections. However, pneumonia due to S. putrefaciens is rare; there are a total of 4 reported cases in the literature. We present a case of 63-year-old male who was presented to emergency room status after cardiac arrest, fell into sea water face down. On the second day of hospitalization, he was diagnosed to have pneumonia based on the clinical, radiological, and laboratory findings. Empirical antibiotic treatment with vancomycin and piperacillin/tazobactam combination was initiated. Gram-stained smear of endotracheal aspirate yielded gram-negative bacteria, and the isolate grown from endotracheal aspirate culture was identified as S. putrefaciens by Biomerieux API 20 NE technique. On review of the literature and according to culture and sensitivity results, therapy in our patient was changed to cefepime. Patient’s pneumonia improved with treatment with cefepime. We believe that our patient developed pneumonia evidently caused by S. putrefaciens, after near drowning in sea water. The pneumonia resolved after treatment with cefepime.

  4. Transverse Vaginal Septum With Secondary Infertility: A Rare Case.

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    Rahman, Hafeez; Trehan, Nikita; Singh, Shuchita; Goyal, Meenakshi

    2016-01-01

    To demonstrate the technique of laparoscopic vaginal reconstruction in a rare case of mid-vaginal septum with secondary infertility. A step-by-step explanation of the technique using videos and pictures (Canadian Task Force classification IV). Transverse vaginal septum is a rare condition, with an incidence of only 1 in 30,000 women. It is usually a congenital mullerian fusion defect; few cases of acquired septum have been reported. Roughly 40% of cases occur in the mid-vagina. Transverse vaginal septum typically presents with primary amenorrhea and hematocolpos. The goal of surgery is to create a patent vagina with restoration of fertility. The laparoscopic approach has proven superiority over the open technique. Laproscopic vaginal reconstruction was performed in a 24 year female with transverse vaginal septum. Dissection was done laproscopically up to mid vagina. Incision was given on vagina excising the septum followed by reconstruction. The laparoscopic approach to vaginal reconstruction avoids the abdominal incision, with its associated pain and possible adhesion formation. It also provides a better view for dissection. In this patient, a patent vagina was created in a single operation, with no postoperative dyspareunia, and fertility was restored. Copyright © 2016 AAGL. Published by Elsevier Inc. All rights reserved.

  5. A rare case of traumatic chylothorax after blunt thoracic trauma

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    Spasić Marko

    2017-01-01

    Full Text Available Introduction. Chylothorax is an accumulation of chyle in the pleural cavity due to a disruption of the thoracic duct. Traumatic chylothoraces are usually a result of a penetrating trauma and disruption of the thoracic duct, but blunt traumatic chylothorax is a rare condition. The aim of this paper is to present a rare case of traumatic chylothorax after blunt thoracic trauma. Case Outline. We present a case of traumatic chylothorax after blunt thoracic trauma in a patient injured in a motor vehicle accident. The patient had a right-sided fracture of rib XI, hydropneumothorax, lung contusion, and signs of pneumomediastinum. We performed thoracic drainage, but a few days later, according to the increase of amount of the fluid daily drained, and the confirmation of laboratory findings of the analyzed fluid, we made a diagnosis of chylothorax and the patient underwent a thoracotomy, where we sutured the thoracic duct. Conclusion. Chylothorax should be considered in patients after chest trauma if they develop a milky pleural effusion. Analysis of pleural fluid and level of triglycerides is important for the diagnosis and treatment of chylothorax. [Project of the Serbian Ministry of Education, Science and Technological Development, Grant no. III41007

  6. Gorlin-Goltz syndrome: A rare case report

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    Chetan A Pol

    2013-01-01

    Full Text Available Gorlin-Goltz syndrome is an uncommon autosomal dominant inherited disorder characterized by numerous basal cell carcinomas, odontogenic keratocysts (OKCs and musculoskeletal malformations. A rare case of this syndrome observed in a 13-year-old male patient is presented in which multiple OKCs were causing disfigurement of the lower jaw as well as displacement and malocclusion of the teeth. Early diagnosis and treatment of this syndrome is important to reduce the severity of complications including cutaneous and cerebral malignancy and oromaxillofacial deformation and destruction due to jaw cysts.

  7. Gorlin-Goltz syndrome: A rare case report.

    Science.gov (United States)

    Pol, Chetan A; Ghige, Suvarna K; Kalaskar, Ritesh R; Gosavi, Suchitra R

    2013-10-01

    Gorlin-Goltz syndrome is an uncommon autosomal dominant inherited disorder characterized by numerous basal cell carcinomas, odontogenic keratocysts (OKCs) and musculoskeletal malformations. A rare case of this syndrome observed in a 13-year-old male patient is presented in which multiple OKCs were causing disfigurement of the lower jaw as well as displacement and malocclusion of the teeth. Early diagnosis and treatment of this syndrome is important to reduce the severity of complications including cutaneous and cerebral malignancy and oromaxillofacial deformation and destruction due to jaw cysts.

  8. Encephalocele presenting as lower lid swelling: A rare case report

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    Vaibhav Kumar Jain

    2018-01-01

    Full Text Available Encephalocele is a rare congenital abnormality characterized by abnormal protrusion of brain and meninges through an opening in the skull. We report an 8-year-old girl who presented with a swelling in the right lower lid for the last 6 years. In her infancy, she had undergone surgery for a very small swelling located in the right nasolacrimal area. On further clinicoradiological evaluation, anterior encephalocele was diagnosed. This case highlights the uncommon site of anterior encephalocele; misdiagnosis and mismanagement of which could result in dreaded complications such as meningitis and cerebrospinal fluid leaking fistula formation.

  9. Two Rare Cases of Hypernatremic Haemorrhagic Encephalopathy in Children.

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    Dr Shital Turakhia

    2016-12-01

    Full Text Available Hypernatremia is defined as increased serum sodium concentration more than 145mEq/L. It occurs most commonly in pediatric patients, geriatric patients and patients with debilitated conditions in ICU who suffers from severe water losses. Newborns and toddlers are at high risk as they have poor thirst control and regulatory mechanism. It occurs when patient is suffering from diarrhea and then mismanagement by over treating the patient with sodium containing fluids. CNS complications and radiological findings are rarely recognized and reported. We hereby report cases of two such pediatric patients who were referred to our hospital from primary health care centers with complain of convulsions and diarrhea.

  10. Rogue-Elephant-Inflicted Panfacial Injuries: A Rare Case Report

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    Santosh Kumar Yadav

    2012-01-01

    Full Text Available Attacks by elephants, the largest of the “large animals,” produce many fatalities a year. Most attacks are provoked, although rogue elephants are occasionally responsible. Trampling, goring, tossing the individual with the trunk, or crushing with the knees produces the injuries. Injuries from encounters with large animals represent a significant health risk for rural communities. Wild-animal-inflicted maxillofacial injuries are rare, and limited literature is available describing their management. We present a case of severe maxillofacial injuries caused by the attack of a rogue elephant.

  11. EXTENSIVE RADICULAR CYST OF THE MANDIBLE: A RARE CASE REPORT

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    Gokul VENKATESHWAR

    2013-03-01

    Full Text Available The radicular cyst is the most common inflammatory odontogenic cystic lesion of the jaws. It usually originates as a sequel to a periapical inflammatory process, following chemical, physical or bacterial injury. Due to its chronic etiology, the cyst usually appears towards the later stage of life. It has a male sex predilection, with the maxillary anterior region as the most common site of involvement. This article reports a rare case of a large radicular cyst in the mandible, its management and follow up along one year.

  12. Acute amebic appendicitis: Report of a rare case

    Directory of Open Access Journals (Sweden)

    Singh Naorem

    2010-10-01

    Full Text Available Acute appendicitis of amebic origin is considered a rare cause of acute appendicitis. We report a case of amebic appendicitis presenting with fever, severe pain in the right lower quadrant of the abdomen and rebound tenderness. Lab investigations revealed neutrophilic leukocytosis. The patient underwent appendectomy. Histopathological examination revealed numerous Entameba histolytica trophozoites in the mucosa of the appendix. Acute appendicitis of amebic origin does not appear frequently. Appendicular amebiasis can give the clinical features of acute appendicitis and should be treated accordingly.

  13. 'Case reporting of rare adverse events in otolaryngology': can we defend the case report?

    LENUS (Irish Health Repository)

    Dias, Andrew

    2012-01-31

    The study of errors in medicine has proliferated since the publication of The Institute of Medicine Report \\'TO ERR IS HUMAN\\' in 2000. Case nuances and process of care issues are valuable areas to explore if the goal is to provide the health care worker with the knowledge to avoid future errors. Meta-analysis and randomized controlled trials provide a large data base of evidence towards improvement and opportunities, but it is suggested that case reports can still provide valuable clinical information. The aim is to use the published literature to produce a series of rare harm case reports in E.N.T. The methods include systematic literature review. Journals searched in PUBMED were 60. Rare harm case obtained from the search were 5,322. Rare harm case reports not reported in any other form of evidence-based medicine were 40. Yes, the case report can be defended as it is an important pillar of evidence-based medicine.

  14. Tea Cup in the brain, a rare case of penetrating brain injury in pediatric patient

    Directory of Open Access Journals (Sweden)

    Sharma Arvind

    2016-03-01

    Full Text Available Head injuries are very common in children. All over the world, the most common mechanism is fall. These injuries are more prevalent in developing countries due to lack of education, poverty, lack of standard and scientific ways to child upbringing. Penetrating injuries in pediatric patients is extremely uncommon and usually occur due to sharp objects like knife, screw driver, drills, nails. We are reporting a rare case of a child with penetrating head injury due to tea cup, very commonly used crockery in every house hold. To the best of our knowledge, no similar case has ever been reported in world literature. Our case also emphasized the need for educating people about child care.

  15. A rare variant of internal anatomy of a third mandibular molar: a case report.

    Science.gov (United States)

    Nimigean, V; Nimigean, Vanda Roxana; Sălăvăstru, D I

    2011-01-01

    The several anatomical variations existing in the root canal system may contribute to failure of the root canal therapy. Knowledge of the internal dental morphology is a complex and extremely important point for planning and performing endodontic therapy. This paper reports the case of a left mandibular third molar that presented only one dental conical root and only one aberrant radicular canal with an initial annular portion situated in the coronar third of the root and a linear portion at the level of the other two thirds of the dental root, which opened through an apical foramen. Root canal therapy and case management are described. Features like wide crown access, adequate illumination and use of exploring files where important for successful completion of the endodontic treatment. The treatment was performed through conventional methods. This clinical case constitutes a rare anatomical variant of internal radicular morphology.

  16. Radiographic diagnosis of a rare case of oculo-dento-digital dysplasia

    Directory of Open Access Journals (Sweden)

    Umesh Chandra Parashari

    2011-12-01

    Full Text Available Oculodentodigital dysplasia (ODDD, also known as oculodento-osseous dysplasia, is an extremely rare autosomal dominant disorder with high penetrance, intra- and interfamilial phenotypic variability, and advanced paternal age in sporadic cases. The incidence of this disease is not precisely known, with only 243 cases reported in the scientific literature, suggesting an incidence of around 1 in 10 million people. It is marked mainly by eye abnormalities, craniofacial dysmorphism, dental anomalies, hand and foot malformations, various skeletal defects, and mildly delayed mental development. Neurological changes may appear earlier in each subsequent generation. This case report describes a radiological diagnosis of ODDD based on physical appearance, clinical features and radiographic findings in a 16-year-old girl.

  17. A rare case of short stature: Say Meyer syndrome

    Directory of Open Access Journals (Sweden)

    T S Karthik

    2013-01-01

    Full Text Available Introduction: Say Meyer syndrome is rare X linked condition characterized by developmental delay, short stature and metopic suture synostosis. We are reporting a case of Say Meyer syndrome presented to our hospital for short stature and developmental delay at age 3½ years. Case Report: A 3½-year-old boy presented to our hospital for decreased growth velocity from the age of 1 year. History revealed the boy had a birth weight of 2.3 kg, had an episode of seizures in the neonatal period. He was born to non-consanguineous marriage. He had global developmental delay and there was a lack of bowel and bladder control. History did not reveal any hearing or visual impairment. No history of any chronic systemic illnesses. Magnetic resonance imaging (MRI brain revealed mild diffuse frontotemporal atrophy with multiple irregular gliotic areas in bilateral frontal lobes. Diffuse white matter volume loss in bilateral cerebral hemispheres. Diffuse thinning of corpus callosum. Diffuse periventricular hyper intensity on T2W and fluid attenuated inversion recovery sequences. Conclusion: Say Meyer syndrome is rare X linked condition characterized by developmental delay, short stature and metopic suture synostosis. Characteristic MRI brain findings include diffuse frontotemporal atrophy with multiple gliotic areas in frontal lobes. Diffuse white matter volume loss in bilateral cerebral hemispheres.

  18. A rare case of bleeding disorder: Glanzmann's thrombasthenia.

    Science.gov (United States)

    Swathi, Jami; Gowrishankar, A; Jayakumar, S A; Jain, Karun

    2017-01-01

    Glanzmann's thrombasthenia (GT) is a rare bleeding disorder, which is characterized by a lack of platelet aggregation. It is characterized by qualitative or quantitative abnormalities of the platelet membrane glycoprotein IIb/IIIa. Physiologically, this platelet receptor normally binds several adhesive plasma proteins, and this facilitates attachment and aggregation of platelets to ensure thrombus formation at sites of vascular injury. The lack of resultant platelet aggregation in GT leads to mucocutaneous bleeding whose manifestation may be clinically variable, ranging from easy bruising to severe and potentially life-threatening hemorrhages. To highlight this rare but potentially life-threating disorder, GT. We report a case of GT that was first detected because of the multiple episodes of gum bleeding. The patient was an 18-year-old girl who presented with a history of repeated episodes of gum bleeding since childhood. Till the first visit to our hospital, she had not been diagnosed with GT despite a history of bleeding tendency, notably purpura in areas of easy bruising, gum bleeding, and prolonged bleeding time after abrasions and insect stings. GT was diagnosed on the basis of prolonged bleeding time, lack of platelet aggregation with adenosine di phosphate, epinephrine and collagen. GT should always be considered as differential diagnosis while evaluating any case of bleeding disorder.

  19. Ovarian tubercular abscess mimicking ovarian carcinoma: A rare case report

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    Abinash Agarwala

    2015-01-01

    Full Text Available Although genito-urinary tuberculosis is common, reports of isolated ovarian tubercular abscess are rare. Ovarian tubercular abscess may mimics that of an ovarian tumor, leading to diagnostic difficulties. We reported a case report of 35 years woman presented with chronic pain abdomen, weight loss, low-grade fever and a right ovarian mass on ultrasound, with a significantly elevated CA-125 level. On clinical and radiological evidence, diagnosis of ovarian carcinoma was made, and laparotomy was performed with resection of the ovary. Postoperative specimen sent for histological examination that revealed classic epithelioid granuloma and acid-fast bacilli were present in Ziehl-Neelsen stain. Patient was put on antitubercular regimen from our Dots center. She is improving clinical after taking antitubercular drug and is on regular follow up at our chest outpatient department. Ovarian tubercular abscess is common in young women living in endemic zones, but case report of isolated tubercular abscess is rarely reported. CA-125 can be raised in both ovarian tubercular abscess and ovarian carcinoma, and only imaging is not always conclusive. Laparotomy followed by tissue diagnosis can be helpful in this situation. As the prognosis and treatment outcome of ovarian tubercular abscess and ovarian carcinoma is different, proper diagnosis by laparotomy should be done. Early diagnosis of ovarian tubercular abscess is vital as untreated disease can lead to infertility.

  20. Adenocarcinoma of the urinary bladder, mesonephroid type: a rare case

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    Mahmoud Abbas

    2013-02-01

    Full Text Available Primary adenocarcinoma of the urinary bladder is a rare disease. It occurs in 0.5-2% of all bladder cancers and is discussed as the malignant counterpart of nephrogenic adenomas. We report a 46-year-old white female presented with gross hematuria for clinical examination. Histopathology revealed pT2, Pn1, L1, G2 adenocarcinoma of the bladder and carcinoma in situ according to the TNM classification. Computed tomography scan diagnostic was unremarkable. Patients with adenocarcinoma of the urinary bladder should be treated vigorously and without time delay. Only 7 cases of adenocarcinoma in the urinary bladder (mesonephroid have been described until now. We present a case of clear cell adenocarcinoma of the urinary bladder, mesonephroid type that early diagnosed and till now 3 months after the cystectomy without symptoms and without complications.

  1. Ectodermal dysplasia-skin fragility syndrome: A rare case report

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    Subhash Kashyap

    2015-01-01

    Full Text Available Ectodermal dysplasia/skin fragility syndrome (ED-SFS is a newly described autosomal recessive disorder characterized by skin fragility and blistering, palmoplantar keratoderma, abnormal hair growth, nail dystrophy, and occasionally defective sweating. It results from mutations in the PKP1 gene encoding plakophilin 1 (PKP1, which is an important component of stratifying epithelial desmosomes and a nuclear component of many cell types. Only 12 cases of this rare genodermatosis have been reported so far. We present an unusual case of ED-SFS in a 12-year boy who was normal at birth but subsequently developed skin fragility, hair and nail deformities, abnormal dentition, palmoplantar keratoderma, and abnormal sweating but no systemic abnormality.

  2. Ectopic third molar in maxillary sinus: A rare case report

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    Abhishek Sinha

    2017-01-01

    Full Text Available Ectopic tooth eruption in a non-dental area is a rare entity, and is most common in oral cavity. There have been a few case reports of teeth erupting in mandibular condyle, chin, palate, coronoid process, and maxillary sinus. Ectopic tooth in the maxillary sinus are found incidentally on routine radiological examination, same time they can be symptomatic and associated with pathologies usually dentigerous cyst or odontogenic keratocyst. Facial pain, purulent rhinorrhoea, epistaxis, headache, swelling, and epiphora-related naso-lacrimal duct obstruction can also be seen. By Caldwell-Luc procedure the ectopic teeth within the maxillary sinus are often removed. In this study, a case of ectopic maxillary third molar tooth on right maxillary sinus is presented.

  3. Mixed Pyolaryngocele: A Rare Case of Deep Neck Infection

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    Rachid Mahdoufi

    2017-07-01

    Full Text Available Introduction: Pyolaryngocele is a very rare and serious complication of laryngocele. It can present as deep neck space infection and mislead the diagnosis. Our aim is to bring this unusual entity to the attention of surgeons and describe its clinical features. Case Report: We report a case of a 45-year-old male patient with a five-week history of neck swelling, dysphonia, dyspnea and odynophagia. An urgent CT scan showed a mixed pyolaryngocele. The management consisted of a high dose antibiotic and an excision of the residual laryngocele via an external approach. Conclusion: A pyolaryngocele is an unusual complication of laryngocele, which becomes secondarily infected, causing many symptoms. Removing the laryngocele is still the best treatment option to prevent this complication and recurrence.

  4. Spontaneous successful pregnancy in posthypophysectomy hypopituitarism: A rare case report

    Directory of Open Access Journals (Sweden)

    Indu Lata

    2014-01-01

    Full Text Available Pregnancy in patients with pan-hypopituitarism following surgery of pituitary adenoma is rare and considered high risk. Hormonal dysfunction in these patients involves more than one axis (gonadotrophic, thyroidal, and adrenal. However, advance in infertility treatment have led to the increased pregnancy rate in hypopituitarism women. We present a case of nonfunctioning pituitary macroadenoma, who after pituitary surgery (hypophysectomy developed hypopituitarism followed by multiple tuberculoma brain with hydrocephalus with arachnoiditis. She conceived spontaneously after 9 years of pituitary surgery and carried her pregnancy to the term. Elective caesarean section was done at 38 weeks and both infant and mother are well. The case highlights the rarity of the phenomenon and the safe outcome of the pregnancy with proper replacement.

  5. A Rare Case of Multifocal Prostatic Blue Nevus

    Directory of Open Access Journals (Sweden)

    Elias J. Farran

    2018-01-01

    Full Text Available Prostatic blue nevus is a rare benign pathologic diagnosis most commonly diagnosed incidentally on many different types of prostate specimens. Blue nevus is the deposition of stromal melanin characterized by spindle cells within the fibromuscular stroma which stains positive for melanin-specific stains Fontana-Masson and S100 and stains negative for CD68, HMB45, and iron stains. We report the case of a multifocal and bilateral blue nevus in a 52-year-old Hispanic male who presented with an elevated prostate-specific antigen of 4.3 and mild obstructive lower urinary tract symptoms, found by transrectal ultrasound-guided prostate needle biopsy. The biopsy also revealed benign prostatic tissue with postatrophic hyperplasia and chronic inflammation. This is the 35th reported case of prostatic blue nevus and the third to show multifocal blue nevus.

  6. Guillain Barre Syndrome Following Traumatic Brain Injury: A Rare Case

    Directory of Open Access Journals (Sweden)

    Kirac Unal

    2016-06-01

    Full Text Available Introduction Guillain-Barre syndrome (GBS is an immune-mediated acute inflammatory disorder of the peripheral nervous system. Infectious agents were usually accused of playing a role in the etiology of GBS. Guillain-Barre syndrome has rarely been reported following subdural and subarachnoid hemorrhage after head trauma. Case Presentation We report on a 63-year-old male patient presenting GBS following Traumatic Brain Injury (TBI. Only five other similar cases are described in the literature. Conclusions Sudden onset of GBS symptoms following trauma may erroneously be assessed as secondary complications of the TBI and can lead to unnecessary procedures such as computerized tomography (CT scan and magnetic resonance imaging (MRI for a definitive diagnosis and may be a waste of time.

  7. A Rare Case Of Graves’ Disease With Splenomegaly And Pancytopenia

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    Elayne Christinne Marcelino e Silva

    2017-07-01

    Full Text Available Introduction: Splenomegaly and pancytopenia are rare complications of Graves' disease with few reports in the literature about this association. The pathogenesis is unknown and immunological mechanisms seem to be involved. The possibility of hyperthyroidsm should always considered in patients with pancytopenia. Objective: Describe  clinical case of association between Grave's disease, splenomegaly and pancytopenia. Method: This is a case report, obtained through data from medical records of a reference hospital located in the city of Juazeiro do Norte, Ceara, Brazil. Case report: Patient, 46 years old, female, sought treatment at a reference hospital with abdominal pain that started two days earlier, prevalent in mesogastric region and left hypochondrium very intense and recurrent, associated with significant consuptive syndrome ( loss of 10 Kg in 4 months, asthenia, dyspnea on minimum exertion, irritability and fine tremor in extremitie. SHe denied fever, palpitations, heat intolerance, skin ou eye changes. A diffuse thyroid enlargement with the presence of thrill and murmur, digital clubbing, fixed and bright look, light exophthalmos and splenomegaly about 6 cm below the left costal margin were abserved after physical examination. Ultrasound examination (USG of the abdomen and CT scan showed moderate splenomegaly. Laboratory tests showed normocytic and normochromic anemia, leukocytosis and mild thrombocytopenia. Thyroid USG showed characteristic features of Graves' disease, a bone marrow biopsy revealed maturation preserved in all strains and lack of fibrosis and megakaryocytes present in normal number without atypia. treatment was set with propylthiouracil 300 mg a day and after the first revaluation after hospital discharge three weeks later a regression of splenomegaly has been observed. Conclusion: This case ilustrates the rare association between hyperthyroidism and splenomegaly with pancytopenia.

  8. Primary Leiomyosarcoma of the Breast: A Rare Case Report

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    Pallavi Agrawal

    2015-08-01

    Full Text Available Background: Primary leiomyosarcoma (LMS of the breast is a very rare neoplasm of the breast arising from the mesenchymal tissue. The clinical presentation of this entity simulates other benign and malignant lesions of mesenchymal tissue of the breast.Case presentation: Case 1: A 40-year-old female presented with a right-sided breast lump, which was suspected to be a malignant spindle cell tumor on needle core biopsy (NCB. A multi-disciplinary team performed modified radical mastectomy (MRM with axillary node dissection on the patient with no post-operative chemo-radiation. Case 2: A 70-year-old female presented with a left sided breast lump and a palpable axillary node. Needle core biopsy diagnosed it as malignant spindle cell tumor. The patient underwent MRM with axillary node dissection. It was confirmed to be a case of breast LMS with axillary nodal metastasis. Both patients were followed up for one year with no evidence of recurrence.Conclusion: Both cases underwent MRM with axillary node dissection in our study. However, the role of axillary dissection in the prognosis and disease-free survival of the patients with primary LMS of the breast with axillary metastasis has not been studied yet. The optimal management of this entity remains to be tumor excision with clear margins.

  9. Pancreatic metastasis from invasive pleomorphic lobular carcinoma of the breast: a rare case report.

    Science.gov (United States)

    Sun, Xiangjie; Zuo, Ke; Huang, Dan; Yu, Baohua; Cheng, Yufan; Yang, Wentao

    2017-07-11

    Invasive pleomorphic lobular carcinoma (PLC) is an aggressive subtype of invasive lobular carcinoma of the breast, which has its own histopathological and biological features. The metastatic patterns for PLC are distinct from those of invasive ductal carcinoma. In addition, pancreatic metastasis from PLC is extremely rare. We report a rare case of a 48-year-old woman presenting with clinical gastrointestinal symptoms and pancreatic metastasis of PLC. The pancreatic tumor was composed of pleomorphic tumor cells arranged in the form of solid sheets and nests and as single files, with frequent mitotic figures, nucleolar prominence, high nuclear to cytoplasmic ratio and loss of cohesion. The malignant cells were positive for p120 (cytoplasmic) and GATA3 and negative for estrogen receptor, progesterone receptor, human epidermal growth factor receptor 2, E-cadherin, gross cystic disease fluid protein 15 and mammaglobin, which indicated a lobular carcinoma phenotype of the breast. To the best of our knowledge, this is one of the few reported cases in the literature of pancreatic metastasis of invasive lobular carcinoma of the breast, of which the definitive diagnosis was obtained only after surgery. Rare metastasis sites should be considered, particularly, when a patient has a medical history of PLC.

  10. A rare case of ectrodactyly in a child in India

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    Mishil S Parikh

    2014-01-01

    Full Text Available Ectrodactyly, termed split-hand/split-foot malformation (SHFM, is a rare genetic condition characterized by defects of the central elements of the autopod (hand/foot. Clinical presentation is with the absence of one or more median rays or digits creating cone-shaped clefts of the hands and/or feet. The present case of severe bilateral SHFM was presented in an 8-year-old girl in India. This case of SHFM involves the complete absence of the central rays of the autopod in which each of the hands and feet is divided into two parts by a cone-shaped cleft tapering proximally, resembling a "lobster claw." SHFM is often associated with other limb anomalies, including monodactyly, syndactyly and aplasia, and/or hypoplasia of the phalanges, metacarpals, and metatarsals. Most cases are sporadic; however, familial forms do exist with predominantly autosomal dominant inheritance. This case is an example of the non-syndromic form of SHFM expressed with isolated involvement of the limbs, while the syndromic form is associated with anomalies such as intellectual disability, ectodermal and craniofacial findings, and hearing loss. Non-syndromic isolated ectrodactyly does not usually require surgical intervention. We recommended against surgical reconstruction due to lack of evidence of functional disability.

  11. Colloidon baby – Rare case with preventable complications

    Directory of Open Access Journals (Sweden)

    Janardhan Bommakanti

    2015-01-01

    Full Text Available Colloidon baby is a rare congenital disorder characterized clinical-ly by parchment like taught membrane covering the whole body at the time of birth, which subsequently develops Non bullous ichthyosiform erythroderma or Lamellar ichthyiosis in most cases and in few cases other ichthyosiform disorders. The colloidon membrane spontaneously desquamates within 2 weeks or up to 3 months in few cases. Herein, we present 2 cases of colloidon babies born to consanguineously married couples of which the first baby was born at term by normal vaginal delivery and second baby born prematurely by caesarean section. Both 1st & 2nd baby were delivered in different private hospitals in villages of Nizamabad district, Telangana state and reported to tertiary level children’s hospital in Hyderabad city on 4nd and 6th day of life respectively with complaints of colloidon membrane and macera-tion of skin in diaper area, was admitted in Neonatal intensive care unit (NICU in humidified incubator, treated with emollients, intra-venous fluids and prophylactic antibiotics to avoid complications. Nursing care is of prime importance. This presentation was aimed at stressing not only the importance of early recognition by pedia-trician & timely referral to dermatologist and ophthalmologist for saving life of affected baby but also equal importance to proper nursing care.

  12. A rare case of short stature: Say Meyer syndrome.

    Science.gov (United States)

    Karthik, T S; Prasad, N Rajendra; Rani, P Radha; Maheshwari, Rushikesh; Reddy, P Amaresh; Chakradhar, B V S; Menon, Bindu

    2013-10-01

    Say Meyer syndrome is rare X linked condition characterized by developmental delay, short stature and metopic suture synostosis. We are reporting a case of Say Meyer syndrome presented to our hospital for short stature and developmental delay at age 3½ years. A 3½-year-old boy presented to our hospital for decreased growth velocity from the age of 1 year. History revealed the boy had a birth weight of 2.3 kg, had an episode of seizures in the neonatal period. He was born to non-consanguineous marriage. He had global developmental delay and there was a lack of bowel and bladder control. History did not reveal any hearing or visual impairment. No history of any chronic systemic illnesses. Magnetic resonance imaging (MRI) brain revealed mild diffuse frontotemporal atrophy with multiple irregular gliotic areas in bilateral frontal lobes. Diffuse white matter volume loss in bilateral cerebral hemispheres. Diffuse thinning of corpus callosum. Diffuse periventricular hyper intensity on T2W and fluid attenuated inversion recovery sequences. Say Meyer syndrome is rare X linked condition characterized by developmental delay, short stature and metopic suture synostosis. Characteristic MRI brain findings include diffuse frontotemporal atrophy with multiple gliotic areas in frontal lobes. Diffuse white matter volume loss in bilateral cerebral hemispheres.

  13. A rare case of fibrostenotic endobronchial tuberculosis of trachea

    Directory of Open Access Journals (Sweden)

    Cassiopia Cary

    2015-12-01

    Full Text Available Endobronchial tuberculosis (EBTB is a sequelae of pulmonary tuberculosis (TB that extends to the endobronchial or endotracheal wall causing inflammation, edema, ulceration, granulation or fibrosis of mucosa and submucosa. This case depicts a 20 year old foreign-born woman with a history of active pulmonary TB on anti-TB chemotherapy, who presented with worsening stridor, dyspnea, cough and weight loss. The disease state was diagnosed with multiple modalities including, spirometry, CT scan of the neck, and bronchoscopy. The biopsies of the tracheal web revealed fibrotic tissue without any granulomas or malignancy establishing the diagnosis of EBTB. Serial balloon dilations and anti-neoplastic therapy with Mitomycin C was used to accomplish sufficient airway patency to relieve her symptoms. ETBT is a rare consequence of TB, which although has a low incidence in the United States, so physicians should have a high clinical suspicion based on the need for prompt intervention.

  14. Rare features associated with Mobius syndrome: Report of two cases

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    Rumela Ghosh

    2017-03-01

    Full Text Available Mobius syndrome is a rare congenital disorder with the preliminary diagnostic criteria of congenital facial and abducent nerve palsy. Involvement of other cranial nerves, too, is common. Prevalence rate of this syndrome is approximately 1 in 100,000 neonates. It is of unknown etiology with sporadic occurrence. However, data regarding the occurrence rate in India is limited. Features such as orofacial malformations, limb defects, and musculoskeletal, behavioral, and cognitive abnormalities might be associated. A thorough evaluation to identify the condition and establishing an adequate treatment plan is of utmost important in this condition. We are reporting clinical and radiographic features of Mobius syndrome in two cases along with unusual findings of limb and neck deformity.

  15. "Wriggling rotters" in the oral cavity: A rare case report

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    Roopashri Govindaraju

    2014-01-01

    Full Text Available Myiasis is derived from a Latin word "Muia," which means fly and "iasis," which means disease. It is a pathological condition in which there is an infestation of living mammals with the dipterous larvae, which, at least for a certain period feed on the host′s dead or living tissue and develop as parasites. Oral myiasis is a rare pathology in humans and is associated with poor oral hygiene, alcoholism, senility, suppurating lesions, severe halitosis, malignancy, low socioeconomic status and others conditions. The treatment consists of mechanical removal of the maggot one by one along with a systemic treatment of ivermectin, a semi-synthetic macrolide antibiotic. We present a case report of a 25-year-old man with intellectual disability, poor oral hygiene, epilepsy, with a deep lacerated wound in the gingiva of maxillary anterior region with acute swelling of the upper lip and presence of larvae of maggot.

  16. Comparatively rare case of a child-patient's cerebral echinococcus

    International Nuclear Information System (INIS)

    Todorov, A.; Simova, E.; Traykova, N.; Stoev, D.; Danev, V.

    2012-01-01

    Full text: Echinococcosis is a parasitic condition with a wide spread in Bulgaria. The possibility of an extensive as well as various localization of the cysts is vastly accounted for. However, according to book references and data available, and medical practice, the brain is somewhat one of the loci with a rare occurrence. The authors panel has taken into consideration the case of an eight-year-old male, who was diagnosed through CT scan with a large cystic lesion in the left cerebral hemisphere with a temporal and parietal localization, as well as a partial destruction of the adjacent pyramid. The presence of an echinococcal cyst has been established through methods of surgical intervention, as well as histologic sample examination. The authors demonstrate the complete anatomical recovery of the encephalic structures of the patient through an ensuing routine CT scan

  17. Radiation-induced spindle cell sarcoma: A rare case report

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    Khan Mubeen

    2009-01-01

    Full Text Available Ionizing radiation has been known to induce malignant transformation in human beings. Radiation-induced sarcomas are a late sequel of radiation therapy. Most sarcomas have been reported to occur after exposure to a radiation dose of 55 Gray (Gy and above, with a dose ranging from 16 to 112 Gys. Spindle cell sarcomas, arising after radiotherapy given to treat the carcinoma of head and neck region is a very uncommon sequel. This is a rare case report of spindle cell sarcoma of left maxilla, in a 24-year-old male, occurring as a late complication of radiotherapy with Cobalt-60 given for the treatment of retinoblastoma of the left eye 21 years back.

  18. [Rare cause for severe hypertriglyceridemia - case 9/2013].

    Science.gov (United States)

    Kahl, Sabine; Roden, Michael; Mörike, Klaus; Müssig, Karsten

    2013-11-01

    We report on a 48-year-old female patient with recently developed severe hypertriglyceridemia. Medical history was remarkable for breast cancer with breast-preserving surgery and chemoradiotherapy. The patient has been treated with 20 mg tamoxifen per day for three months. Laboratory results showed hypertriglyeridemia, hypercholesterolemia and lowered HDL-cholesterol. Findings were consistent with a drug-induced hypertriglyceridemia caused by anti-estrogenic therapy with tamoxifen. After consulting the patient's gynaecologist, we discontinued tamoxifen treatment. Thereupon, triglyceride levels fell consistently. There were no signs of pancreatitis, serum amylase and lipase were in the normal range. Patients with pre-diagnosed metabolic disorders, especially dyslipidemia and type 2 diabetes, should undergo regular controls of serum triglycerides during tamoxifen treatment. Also, one should keep in mind that a subacute, severe rise in serum triglyceride levels may be caused, in rare cases, by tamoxifen treatment. © Georg Thieme Verlag KG Stuttgart · New York.

  19. A rare case of peripartum cardiomyopathy posted for caesarean section

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    Nalini Kotekar

    2007-01-01

    Full Text Available Post Partum Cardiomyopathy (PPCM is a relatively rare form of heart failure associated with pregnancy. It was recognized first in the 19th century by Ritchie and is defined as the onset of acute heart failure in the last trimester or early post partum period in the absence of infections, metabolic, toxic, ischaemic or valvular causes of myocardial dysfunction. Prognosis depends on the degree of cardiomegaly at presentation and in the following 6 months. Initial high risk period carries a mortality of 25 to 50%. Keeping in mind the reduced contractility and ejection fraction with ventricular dilatation proceeding to cardiac failure, the anesthesiologist managing a case of PPCM faces the challenge of avoiding myocardial depression, hypovolemia and increased SVR, all of which may be hazardous

  20. A rare case of hypocalcemia induced by nilotinib

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    Marija Petrić

    2017-03-01

    Full Text Available Chronic myelogenous leukemia (CML is a myeloproliferative disorder characterized by increased proliferation of predominantly myeloid cells in the bone marrow and their accumulation in the peripheral blood. Nowadays, drugs known as tyrosine kinase inhibitors (TKIs are the standard treatment for CML. Since TKIs specifically target BCR-ABL, the activated tyrosine kinase fusion protein, they are expected to cause less hematological and nonhematological side effects than medications used before. We report a case of a 61-year-old patient treated with the second-generation TKI, nilotinib, that presented with very common side effects (skin rash, myalgia, and paresthesia, and also with rare, but severe hypocalcemia and potentially dangerous and fatal QTc elongation.

  1. Pyknodysostosis: report of a rare case with review of literature

    International Nuclear Information System (INIS)

    Ramaiah, Kiran Kumar Kotagudda; George, Giju Baby; Padiyath, Sheeba; Sethuraman, Rupak; Cherian, Babu

    2011-01-01

    Pyknodysostosis is a rare autosomal recessive disorder characterized by the post natal onset of short limbs, short stature, and generalized hyperostosis along with acro-osteolysis with sclerosis of the terminal phalanges, a feature that is considered essentially pathognomonic. Other features include persistence of fontanelles, delayed closure of sutures, wormian bones, absence of frontal sinuses, and obtuse mandibular gonial angle with relative mandibular prognathism. We report a case of 17-year-old girl who presented with a chief complaint of retention of deciduous teeth. General physical examination demonstrated short stature, frontal and parietal bossing, depressed nasal bridge, beaked nose, hypoplastic midface, wrinkled skin over the finger tips, and nail abnormalities. Radiographs showed multiple impacted permanent and supernumerary teeth, hypoplastic paranasal sinuses with acro-osteolysis of terminal phalanges, and open fontanelles, and sutures along with wormian bones in the lambdoidal region.

  2. A rare case of chondroblastioma of the proximal tibia

    International Nuclear Information System (INIS)

    Gecov, P.; Terziev, I.; Hristoskova, R.; Georgiev, G.; Kinov, P.; Tivchev, P.

    2011-01-01

    The authors report a rare case of chondroblastoma which was localised in the proximal tibial epiphysis in a 14-year-old boy. The lesion was evaluated with routine roentgenography and computer tomography. The imaging techniques revealed eccentric lesion, predominantly in the medial part of the bone, periosteal reaction in the metaphysic, calcification and sclerosis. After diagnostic evaluation including biopsy, the lesion was treated surgically with curettage and grafting. He was followed up for two years with routine radiographs and physical examination. Two years after surgery the patient had no recurrence, no pain and had nearly full range of motion of the knee. Roentgenographic characteristics of the disease as well as differential diagnostics are discussed. In conclusion, imaging techniques should be supplemented with an open biopsy for the final diagnosis.

  3. Pyknodysostosis: report of a rare case with review of literature

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    Ramaiah, Kiran Kumar Kotagudda; George, Giju Baby; Padiyath, Sheeba; Sethuraman, Rupak; Cherian, Babu [Mar Baselios Dental College, Kothamangalam (India)

    2011-12-15

    Pyknodysostosis is a rare autosomal recessive disorder characterized by the post natal onset of short limbs, short stature, and generalized hyperostosis along with acro-osteolysis with sclerosis of the terminal phalanges, a feature that is considered essentially pathognomonic. Other features include persistence of fontanelles, delayed closure of sutures, wormian bones, absence of frontal sinuses, and obtuse mandibular gonial angle with relative mandibular prognathism. We report a case of 17-year-old girl who presented with a chief complaint of retention of deciduous teeth. General physical examination demonstrated short stature, frontal and parietal bossing, depressed nasal bridge, beaked nose, hypoplastic midface, wrinkled skin over the finger tips, and nail abnormalities. Radiographs showed multiple impacted permanent and supernumerary teeth, hypoplastic paranasal sinuses with acro-osteolysis of terminal phalanges, and open fontanelles, and sutures along with wormian bones in the lambdoidal region.

  4. A rare case of autoimmune limbic encephalitis: an uncharted territory!

    Science.gov (United States)

    Ibrahim, Hatim; Al Jasser, Abdulelah N; Khan, Sonia A; Tlili, Kalthoum G

    2017-10-01

    Autoimmune encephalitis is rare. Several auto- antibodies are described in autoimmune encephalitis. We describe a case of autoimmune limbic encephalitis associated with positive voltage gated potassium channel (VGKC) antibodies and positive leucine-rich glioma inactivated protein 1 antibodies (LGI1). A 33-year-old Saudi housewife, she presented with 2 months history of cognitive deterioration and recurrent left facio-brachial dystonic seizures followed by generalized tonic clonic seizures. At times the seizures are preceded by rising epigastric aura and shortness of breath. The neurological examination was normal apart from upgoing left plantar reflex. She had borderline IQ of 76 with impaired verbal fluency and impaired visual and verbal memory. Magnetic resonance imaging of the brain showed right mesial temporal non-enhancing lesion. Cerebrospinal fluid examination was positive for LGI1 and VGKC. Optimal seizure control was achieved with immunotherapy.

  5. A rare case of trichotillomania with antisocial personality disorder

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    Priti Singh

    2015-01-01

    Full Text Available Trichotillomania (TTM is characterised by recurrent and irresistible urge to pull out one’s own body hair. It is often associated with trichorrhizophagia in which there is a habit to eat the roots of pulled out hairs. It can also present with many comorbid psychiatric problems including personality disorders. High rates of comorbid mood, anxiety, and substance use disorders have been detected in patients of TTM. The lifetime prevalence of comorbid personality disorders has been much less extensively studied. We present a rare case of 28-year-old male having TTM with antisocial personality disorder and discuss difficult management issues with this comorbidity. Our patient improved with a combination of fluoxetine and sodium valproate.

  6. A Rare Case of Zosteriform Cutaneous Metastasis from Breast Cancer

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    Filiz Topaloğlu Demir

    2017-03-01

    Full Text Available Breast cancer is the most common cancer among women and the second leading cause of cancer deaths, after lung cancer. Cutaneous breast cancer metastases often develop as direct involvement and local spread and often manifest as solid painless nodules in the anterior chest wall. Internal malignant skin metastases rarely present like soft nodules, telangiectasia-like lesions, neoplastic alopecia, erysipeloides carcinoma, erythema annulare-like, herpetiformis or zosteriform, target-like, pyodermic and morphea-like lesions. In this article, we present a 49-year-old female patient describing a sensation of burning pain with erythematous papules and plaques in a zosteriform distribution. The diagnosis of zosteriform cutaneous metastases from a breast cancer was made. Majority of these cases may be misdiagnosed as herpes zoster infection and can be treated with antiviral drugs. Therefore, cutaneous metastases should be kept in mind in the differential diagnosis of lesions in zosteriform distribution.

  7. Dengue Haemorrhagic Encephalitis: Rare Case Report with Review of Literature.

    Science.gov (United States)

    Kutiyal, Aditya Singh; Malik, Chetanya; Hyanki, Gitika

    2017-07-01

    Dengue is an endemic arboviral infection prevalent especially in tropical countries including Southern and Southeast Asia. Central Nervous System (CNS) involvement in dengue infection is uncommon. Haemorrhagic encephalitis is a rare presentation in dengue. This is a case of a 58-year-old male who presented with fever, petechial rash and altered sensorium. Dengue serology IgM was reactive and MRI brain was suggestive of haemorrhagic encephalitis. Patient was managed in Intensive Care Unit (ICU) but eventually succumbed to his illness. We report this fatal outcome of a common viral infection with unusual neurological presentation to propose an association between dengue and neurotropism and the need to look at dengue infection beyond its classical features.

  8. Adderall Induced Acute Liver Injury: A Rare Case and Review of the Literature

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    Rohini R. Vanga

    2013-01-01

    Full Text Available Adderall (dextroamphetamine/amphetamine is a widely prescribed medicine for the treatment of attention-deficit/hyperactivity disorder (ADHD and is considered safe with due precautions. Use of prescribed Adderall without intention to overdose as a cause of acute liver injury is extremely rare, and to our knowledge no cases have been reported in the English literature. Amphetamine is an ingredient of recreational drugs such as Ecstacy and is known to cause hepatotoxicity. We describe here the case of a 55-year-old woman who developed acute liver failure during the treatment of ADHD with Adderall. She presented to the emergency room with worsening abdominal pain, malaise, and jaundice requiring hospitalization. She had a past history of partial hepatic resection secondary to metastasis from colon cancer which was under remission at the time of presentation. She recovered after intensive monitoring and conservative management. Adderall should be used carefully in individuals with underlying liver conditions.

  9. Posterior Uterine Rupture Causing Fetal Expulsion into the Abdominal Cavity: A Rare Case of Neonatal Survival

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    K. Navaratnam

    2011-01-01

    Full Text Available Introduction. Uterine rupture is a potentially catastrophic complication of vaginal birth after caesarean section. We describe the sixth case of posterior uterine rupture, with intact lower segment scar, and the first neonatal survival after expulsion into the abdominal cavity with posterior rupture. Case Presentation. A multiparous woman underwent prostaglandin induction of labour for postmaturity, after one previous caesarean section. Emergency caesarean section for bradycardia revealed a complete posterior uterine rupture, with fetal and placental expulsion. Upon delivery, the baby required inflation breaths only. The patient required a subtotal hysterectomy but returned home on day 5 postnatally with her healthy baby. Discussion. Vaginal birth after caesarean section constitutes a trial of labour, and the obstetrician must be reactive to labour events. Posterior uterine rupture is extremely rare and may occur without conventional signs. Good maternal and fetal outcome is possible with a prompt, coordinated team response.

  10. Osteosarcoma of the Maxilla: A Rare Case with Unusual Clinical Presentation

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    Pouyan Amini Shakib

    2013-08-01

    Full Text Available Osteosarcoma (OS is a malignant mesenchymal tumor, which rarely occurs in the maxilla. Although variable histologic and radiographic features of OS have been reported previously, in the majority of the cases painful swelling of the jaw is mentioned as the first clinical presentation. Furthermore, early diagnosis and wide surgical resection of the tumor are the most important determinant factors of prognosis. Therefore, the unusual clinical presentations of OS should be considered meticulously to expedite the diagnosis process. We describe a case of OS of the maxilla with extremely unusual presentation in a 42-year-old female, that was initially designated as “epulis fissuratum”. Here, we highlight the importance of combining the clinical, radiographic and histopathologic examination to obtain a definitive diagnosis and also the significance of early effective surgical intervention in evaluation of pathologic lesions.

  11. Usher syndrome Type I in an adult Nepalese male: a rare case report.

    Science.gov (United States)

    Sahu, Sabin; Singh, Sanjay Kumar

    2017-07-01

    Usher syndrome, also known as retinitis pigmentosa-dysacusis syndrome, is an extremely rare genetic disorder, characterized by retinitis pigmentosa (RP) and congenital sensorineural hearing loss. It has been estimated to account for 3-6% of the congenitally deaf population, upto 8-33% of individuals with RP and half of all cases with combined deafness and blindness (Vernon M,1969; Boughman JA et al,1983). The prevalence of Usher syndrome have been reported to range from 3.5 to 6.2 per 100,000 in different populations (Vernon M,1969; Boughman JA et al,1983; Yan D et al, 2010). We report a case of Usher syndrome type I in an adult Nepalese male with typical congenital profound hearing loss, and night blindness secondary to retinitis pigmentosa. © NEPjOPH.

  12. Pilomatrixoma of the Arm: A Rare Case with Cytologic Diagnosis

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    Ruchika Gupta

    2012-01-01

    Full Text Available Pilomatrixoma, a benign skin appendageal tumor, is seen commonly in head and neck. Occurrence of pilomatrixoma in the upper extremities is not common and has been reported infrequently in the available literature. Only a few cases with preoperative aspiration cytology have been reported in the literature. A five-year-old girl underwent fine needle aspiration (FNA of a firm subcutaneous nodule on the lateral aspect of left arm. FNA smears showed scattered and few fragments of round to oval cells along with multinucleated giant cells. Few shadow cells were seen. A cytologic impression of pilomatrixoma was rendered, which was confirmed on histopathology. Pilomatrixoma, a common skin appendageal tumor in head and neck region, should be considered in the cytologic differential diagnoses of subcutaneous masses even in unusual locations like arm. The varied cytomorphology should be remembered to avoid misdiagnosis.

  13. An Unusual Endovascular Therapeutic Approach for a Rare Case of May-Thurner Syndrome.

    Science.gov (United States)

    DaSilva-DeAbreu, Adrian; Masha, Luke; Peerbhai, Shareez

    2017-03-06

    BACKGROUND The etiology of deep venous thrombosis (DVT) may pose a significant diagnostic challenge because truly reversible causes of DVT are rare. In this regard, known pelvic anatomic abnormalities such as aortic and iliac aneurysms should be seriously considered as a complicating factor in patients presenting with acute DVT so as not to miss a potentially curable etiology of May-Thurner syndrome (MTS). CASE REPORT We report the case of a 69-year-old man with a known abdominal aortic aneurysm and bilateral iliac artery aneurysms who presented with an acute DVT. A computed tomography scan of the abdomen and pelvis showed increased dilation of his aneurysmal disease with new resultant compression of the left iliac vein representing acquired MTS. The patient underwent endovascular aneurysm repair of the infra-renal abdominal aortic aneurysm and right common iliac artery aneurysm with a Gore Excluder endoprosthesis in lieu of venous stenting, with resolution of symptoms. CONCLUSIONS Infra-renal aortic and iliac aneurysms causing MTS are extremely rare, and patients at risk for MTS through these mechanisms do not fit the classical demographics associated with this syndrome. Furthermore, this is the first case described in which MTS was treated by addressing the aneurysm through an endoprosthetic approach instead of venous stenting, which is the conventional intervention for MTS.

  14. Alveolar Rhabdomyosarcoma of the foot metastasizing to the Iris: report of a rare case

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    Fabian, Ido Didi; Hildebrand, G. Darius; Wilson, Shaun; Foord, Tina; Sagoo, Mandeep S.

    2016-01-01

    Intraocular iris rhabdomyosarcoma is extremely rare, and in the 3 cases reported to date occurred as the primary site of tumour growth. We report a case of rhabdomyosarcoma of the foot metastasizing to the iris. An 18-year-old white female was referred to the London Ocular Oncology Service for management of a metastatic rhabdomyosarcomatous deposit in the iris, a metastasis from alveolar rhabdomyosarcoma of the foot. She was diagnosed nearly 2 years earlier with the primary sarcoma with extensive systemic spread and treated by resection of the foot lesion and chemotherapy, and achieved a partial remission. The left iris deposit was noted while she was receiving systemic chemotherapy, heralding a relapse. However, anterior uveitis and raised intraocular pressure developed and she was referred to our service for further management. A left iris secondary rhabdomyosarcoma deposit was noticed and in addition a lacrimal gland mass, as indicated by ultrasound B scan of the eye and orbit. The patient was treated with external beam radiotherapy to the globe and orbit, but died 2 months after treatment completion. Rhabdomyosarcoma of the iris is very rare and was previously documented only as a primary malignancy in this location. We report that secondary spread to the iris can also occur, in this case as the first sign of widely disseminated systemic relapse

  15. Double Trouble: A Rare Case of Bilateral Upper Pole Ureteropelvic Junction Obstruction

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    Craig A. Peters

    2014-09-01

    Full Text Available A 16-year-old girl presented with bilateral back pain caused by bilateral upper pole ureteropelvic junction obstructions; an extremely rare phenomenon. Bilateral robotically assisted upper pole pyeloplasties were preformed at the same setting with an excellent clinical response. Although rare, upper pole ureteropelvic junction obstruction is a defined entity that urologists should be aware of.

  16. Cyclopia: A Rare Condition with Unusual Presentation - A Case Report

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    Ghassan S.A. Salama

    2015-01-01

    Full Text Available Introduction Cyclopia (alobar holoprosencephaly (OMIM% 236100 is a rare and lethal complex human malformation, resulting from incomplete cleavage of prosencephalon into right and left hemispheres occurring between the 18th and the 28th day of gestation. Holoprosencephaly occurs in 1/16,000 live births, and 1/250 during embryogenesis. Approximately 1.05 in 100,000 births are identified as infants with cyclopia, including stillbirths. Cyclopia typically presents with a median single eye or a partially divided eye in a single orbit, absent nose, and a proboscis above the eye. Extracranial malformations described in stillbirths with cyclopia include polydactyl, renal dysplasia, and an omphalocele. The etiology of this rare syndrome, which is incompatible with life, is still largely unknown. Most cases are sporadic. Heterogeneous risk factors have been implicated as possible causes. Case Presentation A live full-term baby with birth weight of 2900 g, product of cesarean section because of severe fetal bradycardia, was born at Prince Hashem Military Hospital – Zarqa city/Jordan. This newborn was the first baby to a non-consanguineous family, and a healthy 18-year-old mother, with no history of drug ingestion or febrile illnesses during pregnancy. Antenatal history revealed severe hydrocephalus diagnosed early by intrauterine ultrasound but the pregnancy was not terminated because of the lack of medical legitimization in the country. On examination, the newborn was found to have a dysmorphic face, with a median single eye, absence of nose, micrognathia, and a proboscis above the eye, all of which made cyclopia the possible initial diagnosis. Multiple unusual abdominal defects were present that include a huge omphalocele containing whole liver and spleen, urinary bladder extrophy, and undefined abnormal external genitalia, which called for urgent confirmation. Brain MRI was done and revealed findings consistent with alobar holoprosencephaly (cyclopia

  17. A Rare Comorbidity: Dermatitis Herpetiformis and Sarcoidosis - A Case Report

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    Ivanov Pavlov Stoyan

    2016-09-01

    Full Text Available Sarcoidosis is an enigmatic, multisystem granulomatous disease of unknown etiology and wide range of clinical presentations. Case report: A 54-year-old female presented with facial rash: polymorphic, round, infiltrated erythematous plaques, 1 - 3 cm in size, disseminated on several areas of the face. The medical history was consistent with dermatitis herpetiformis and persistent intrahepatic cholestasis. The laboratory test results suggested celiac disease (strong positivity of IgA anti-tissue transglutaminase antibodies but upper endoscopy was not performed to confirm it. The skin biopsy revealed noncaseating epithelioid-cell granulomas, and negative direct immunofluorescence showed IgA deposits in the dermis. Sarcoidosis with cutaneous and hepatic involvement was established based on compatible clinical findings and supportive histology. The period between manifestations of Duhring disease and skin manifestations of sarcoidosis was 20 years. Conclusion: Our clinical case supports the hypothesis for common immune pathogenic factors in gluten-sensitive diseases and sarcoidosis. The simultaneous occurrence of celiac disease and sarcoidosis is rare, but should not be under recognized.

  18. An Unusual Erupted Complex Composite Odontoma: A Rare Case

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    Dawasaz Ali Azhar

    2013-01-01

    Full Text Available Odontomas are malformations of the dental tissues and may interfere with the eruption of the associated tooth. Complex composite odontoma (CO was described as a distinct entity for the first time by Broca in 1866. This lesion takes place due to the developmental disturbances where the dental components are laid down in a disorganized manner, due to failure of normal morphodifferentiation. Very few cases of erupted complex composite odontomas have been reported in the literature. The case reported here is of an odontoma found in the left mandibular body, associated with an impacted second molar of a 17-year-old Saudi male. Under local anesthesia the odontoma was surgically removed. Histopathological examination confirmed the diagnosis of CO. The impacted second molar which was left in the mandibular body erupted clinically after 6 months. Erupted CO is rarely seen in the mandibular left body. The early diagnosis, followed by a proper treatment at the right time, will result in a favorable prognosis.

  19. A rare case of metastatic squamous urachal carcinoma.

    Science.gov (United States)

    Andrei, S; Andrei, A; Rusu Muntean, G; Ungureanu, M; Herlea, V; Becheanu, G; Popescu, I

    2013-01-01

    Squamous cell carcinoma is a very rare type of urachal malignancy, only a few cases being reported in the medical literature. We present the case of a 49-year-old male patient diagnosed with infected squamous cell urachal carcinoma with multiple pulmonary metastases, after complaints of lower abdominal pain, abdominal mass and fever, without respiratory symptoms. The abdominal ultrasonography and the CT scan revealed a tumoral mass in the lower abdomen in contact with the abdominal wall and the urinary bladder dome, displacing the small bowel. Pulmonary nodular lesions were described in the left lobe pyramid. The intraoperative diagnosis was necrotic urachal tumor with urinary bladder dome invasion and suspected pulmonary metastases, and tumor ablation with bladder dome resection and suture of the bladder were performed. The histopathological result was poorly differentiated squamous cell carcinoma (G3), with negative resection margins. The patient recovered well after surgery, but the prognosis is very poor due to the metastatic stage in which the tumor was diagnosed, no standard chemotherapy regimen for the treatment of metastatic urachal carcinoma being known as effective until now. Celsius.

  20. A rare case of multiple meningiomas with different histology

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    Toma I. Papacocea

    2017-11-01

    Full Text Available Meningiomas are generally benign tumors but sometimes they manifest tendency to progress towards malignancy. It is not yet clear if anaplastic meningiomas have an innate malignancy characteristic, or an initially beginning histological appearance that degenerates malignantly in time. According to literature data, the risk of a benign meningioma to progress towards malignant phenotype is about 0.16-2%, such malignant transformation occurring after a variable period of time (2-16 years. A still unanswered question is how many of the malignant meningiomas present this appearance as an innate feature and how many of them originate from benign meningiomas. Multiple meningiomas are defined as the presence of two or more distinct meningiomas. They occur in 6-10% of all patients that present meningiomas. Multiple meningiomas with a distinct histological appearance are rarely discovered. They support the theory of meningiomas that develop independently in the same patient. Different histology of multiple meningiomas is found in less than a third of the patients who suffer from this pathology. We are presenting the case of a patient with multiple meningiomas with distinct histology, one being benign and the other malignant. In connection with this case we are raising a question of therapeutic management in patients diagnosed with malignant meningiomas, namely if other possible small/ benign meningiomas should be also entirely resected.

  1. A Rare Anterior Abdominal Wall Defect: Omphalocele - A Case Report

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    Sandeep Vilasrao Pakhale

    2015-01-01

    Full Text Available Two most common anterior abdominal wall defects are gastroschisis and omphalocoele or exomphalos. Gastroschisis means 'stomach cleft' which is a congenital defect of the abdominal wall, usually to the right of the umbilical cord insertion and abdominal contents herniate into the amniotic sac. Exomphalos is literally translated from the Greek, means 'outside the navel'. It is also called an Omphalocele. It is a congenital abnormality in which the contents of the abdomen herniate into the umbilical cord through the umbilical ring. Textbooks grouped them together but these are different entities. These congenital malformations have a high mortality rate. Only about 60 % of children with such type of malformations survive until the end of first year of age. A male foetus of 32 weeks gestational age was sent from Dr. Ulhas Patil Medical College and Hospital, Jalgaon (Khurd to the Department of Anatomy to examine the fetus for congenital anomalies. A case report of an Omphalocele was presented. Occurrence of such cases is very rare about 2.17 per 10000 live births as reported in literature.

  2. Chronic meningococcemia: a rare presentation of meningococcal disease: case report

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    Antonio Adolfo Guerra Soares Brandão

    2012-03-01

    Full Text Available Chronic meningococcemia is a rare clinical presentation within the spectrumof infections due to Neisseria meningitidis, which was first described in 1902.It is defined as a chronic and benign meningococcal bacteremia withoutmeningeal signs or symptoms with at least one week’s duration, characterizedby intermittent or continuous fever, polymorphic cutaneous rash, and migratoryarthropathy. The incidence is believed to be around 1:200,000 inhabitants. Itaffects predominantly young people and adults, and it is equally distributedbetween genders. Diagnosis may be challenging in the early stages of thedisease because of the difficulty in isolating Neisseria meningitidis (it reaches74% of positivity in advanced stages. Recently, the use of PCR for detectingNeisseria sp antigen in skin biopsies specimens has been considered for thoseculture-negative cases. The authors report a case of a 54-year-old femalepatient who sought medical attention for a five-day fever followed by arthralgiaand skin lesions predominantly in the lower limbs. The patient progressed toa toxemic clinical status that improved after the administration of antibiotictherapy, which consisted of oxacillin and ceftriaxone. The diagnosis of chronicmeningococcemia was performed after the isolation of Neisseria meningitidisin two different blood sample cultures. This is, to our knowledge, the firstcase of chronic meningococcemia described in Brazil (up to the writing of thisreport.

  3. A Rare Case of Chronic Diarrhea in an Elderly Male

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    Ming-Jong Bair

    2009-06-01

    Full Text Available Chronic diarrhea is a common condition in older age groups, and many patients do not seek medical attention unless their diarrhea is associated with other symptoms, such as weight loss, gastrointestinal bleeding or abdominal pain. It is a critical condition in the elderly, especially with systemic disease. We report the case of an elderly patient with chronic diarrhea secondary to intestinal capillariasis. Human intestinal capillariasis is a rare parasitosis of the gastrointestinal tract, which may be a fatal disease if early treatment is not given. The clinical hallmarks of capillariasis include chronic diarrhea, abdominal pain, borborygmi, marked weight loss, protein and electrolyte loss, and cachexia. Most patients die from electrolyte loss resulting in heart failure and/or septicemia. Taiwan, particularly in Taitung County, is a Capillaria-prevalent area. Thus, parasitosis must be considered in the differential diagnosis of patients with debilitating chronic diarrhea, especially in the elderly aboriginal population of Taitung County. A careful dietary and travel history is important in any such case; but even in the absence of clear-cut exposure, a parasitic infection should be considered and carefully investigated.

  4. Religious extremism: The case of Sudan's Mariam Yahya Ibrahim Ishag

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    Noah K. Tenai

    2016-03-01

    Full Text Available The recent case of the arrest, prosecution and imprisonment of Mariam Yahya Ibrahim Ishag of Sudan has drawn attention to the place of Islamic sharia law in contemporary, diverse and multireligious communities and nation states. Islamic sharia law was used to charge Mariam of apostasy; she was subsequently sentenced to 100 lashes followed by hanging. Religious extremism and one of its resultant effects, namely persecution, particularly of women and other minorities, is a persistent hindrance to ongoing efforts against poverty responses. Religious extremism goes against the spirit of Article 18 of the Universal Declaration of Human Rights and Article 18 of the International Covenant of Civil and Political Rights, of which many nation states are signatories. The Catholic vows of consecration � poverty, chastity and obedience � are very helpful perspectives that can assist in pursuing responses to religious extremism and the resultant intolerance, persecution and dispossession.Intradisciplinary and/or interdisciplinary implications: Drawing from the Roman Catholic Church�s vows of consecration, the article argues for a stance that communities can take in situations that call for solidarity with people in vulnerable situations.

  5. Extremely Elevated CA 125 and CA 19-9 in Endometrioma; A Case Series

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    Vugar Bayramov

    2010-03-01

    Full Text Available Although endometriosis is a benign condition, increased levels of the tumor markers CA 125 and CA 19-9 may be seen. However, these tumor markers reach to very high levels, rarely. In this report, 4 patients between 20 and 43 year-old with extremely elevated CA 125, CA 19-9 and CA 15-3 levels are discussed. In endometriosis extremely increased tumor markers are determined in the case of ruptured endometrioma cyst. There are two mechanisms to clarify extremely elevated levels of CA 125 in endometriosis. First, the peritoneal irritation of CA 125 molecule after the rupture of endometioma cyst and CA 125 secretion from the periton. And the second is penetration of the CA 125 moecule easily to the circulation through the peritoneal endothelial surface after the cyst rupture. In conclusion, the diagnosis of ruptured endometrioma cyst should be kept in mind especially in young patients with extremely elevated serum CA 125 levels with regard to the history and ultrasonographical signs and invasive procedures should be avoided.

  6. Diagnosis and Management of a Patient with Congenitally Missing Maxillary First Permanent Molars: A Rare Case Report

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    Megha Gupta

    2016-01-01

    Full Text Available Congenitally missing teeth are the most commonly seen dental anomalies. Agenesis of the permanent first molar has the least frequency of all the tooth types, and it usually occurs in association with oligodontia or anodontia. Thus, agenesis of the bilateral maxillary first permanent molar is an extremely rare occurrence, and no such case has been reported in ethnic Saudi Arabian population. We hereby report a case of nonsyndromic bilateral congenitally missing maxillary first permanent molar in an eight-year-old Saudi female patient. Comprehensive oral rehabilitation was done for the patient. The implications of the tooth agenesis are also discussed. The prognosis of this case is presented.

  7. A rare case of primary urachal mucinous adenocarcinoma

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    Ahmad Naufal Md Alwi

    2017-12-01

    Full Text Available Background: Urachal cancers arising from the urachus or its remnants, is a very rare and aggressive type of cancer in which occurrence is only about 0.01% of all adult cancers. Till date, there is no standard chemotherapy regimes or randomized trials due to the rarity of the disease. In 2007, not more than 350 written case reports in English has been published worldwide. This case report aims to share our department’s modest experience in management of a primary urachal mucinous adenocarcinoma in the adjuvant setting. Case Description: We report a 54-year-old gentleman from Kudat, Sabah, who presented with an umbilical mass and lower abdominaldiscomfort. Investigations revealed a primary urachal mucinous adenocarcinoma in May 2016 and he had undergone en-bloc resection of urachal tumour up to the serosal layer of the dome of bladder. Post-operatively, he received 12 cycles of adjuvant chemotherapy in the form of FOLFOX-4 regime from June 2016 to December 2016. He was last reviewed in September 2017 and is currently well and asymptomatic. Latest CT scan did not show any tumour recurrence nor distant metastasis. Results: This gentleman has been disease free for 16 months with no clinical or radiological evidence of local recurrence or distant metastasis. Conclusion: There are only few picaresque responses treating urachal cancers with chemotherapy regimens used for bladder carcinomas. Taking into consideration the enteric type histology of urachal adenocarcinomas, thechemotherapy regimes used for gastrointestinal malignancies may possibly yield better response and outcome.

  8. Single coronary artery; extremely rare coronary anomaly successfully treated surgically in young adult male.

    LENUS (Irish Health Repository)

    Shah, A R

    2010-05-01

    Single coronary artery arising from aortic root, is a rare congenital anomaly. A 30-year-old male presented with acute myocardial infarction (MI) complaining of chest pain and raised troponin levels. Emergency angiography showed no coronary lesions but both left and right coronary arteries arising from single ostium. Patient was operated electively and perioperative findings confirmed the diagnosis of single coronary artery, as left coronary artery after taking origin from right sinus of valsalva runs through the septum, before dividing into left anterior descending and circumflex branches. The single coronary ostium opened with a slit like incision over the course of left main coronary, making the size of ostium three to four times bigger than the native one. In addition left internal mammary artery was harvested and grafted to the left anterior descending branch distally. Patient made successful recovery. Four months follow up dobutamine stress echo showed no inducible ischemia.

  9. Psychosocial reactions to upper extremity limb salvage: A case series.

    Science.gov (United States)

    Sposato, Lindsay; Yancosek, Kathleen; Cancio, Jill

    2017-11-30

    Case series. A salvaged limb is one that has undergone a major traumatic injury, followed by repeated surgical attempts in order to avoid amputation. Psychological recovery for individuals with lower extremity limb salvage has been examined in a number of studies. However, psychosocial reactions for individuals with upper extremity (UE) limb salvage are understudied in the literature. The purpose of this study was to explore the process of psychosocial adaptation for 3 trauma cases after UE limb salvage. The Reactions to Impairment and Disability Inventory was used to assess psychosocial adaptation. Physical function outcomes (pain, range of motion, edema, sensation, and dexterity) are presented. The Disabilities of the Arm, Shoulder, and Hand measure was used to assess perceived disability. Medical and rehabilitation history are discussed for each case, in order to provide in-depth understanding of the impact of these injuries. Reactions to injury varied across the cases; however, outcomes suggest that psychosocial adaptation may be influenced by the experience of pain, the ability to participate in valued roles and activities, and having a supportive social network. For this population, therapists may consider emphasizing pain management, focusing on client-centered goals and interventions, and facilitating peer support. Providers should closely monitor patients for signs of poor adaptation, such as hand-hiding behaviors. This study is among the first to examine psychological outcomes for the UE limb salvage population. Future research would be beneficial to provide deeper understanding of the psychosocial challenges for these individuals. Copyright © 2017 Hanley & Belfus. Published by Elsevier Inc. All rights reserved.

  10. [Sirenomelia--a rare cause of an oligoanhydramnion in the second trimester--a case report].

    Science.gov (United States)

    Morfeld, C A; Hofstaetter, C; Adolf, S; Radner, H; Schild, R L

    2012-02-01

    Sirenomelia is a rare, but complex and lethal malformation. It is caused by a primary defect of the caudal axial skeleton and damage to the primary streak, which appears due to a vascular steal phenomenon. Sirenomelia appears sporadic with an incidence of 1-64,000 births. A risk for sirenomelia can be also found in patients with poorly controlled diabetes mellitus and in monocygotic twins. Leading ultrasound findings are fusioned lower extremities, bilateral renal agenesis, single umbilical artery and a distinct oligohydramnios. 3D ultrasound and color Doppler sonography can additionally be used for diagnostic, as well as amnioninfusion. There are 3 forms of sirenomelia, depending on missing or presence of the feet it is distinguished as sympus apus, monopus or dipus. We are presenting a case of sirenomelia with sympus dipus, which was transferred for further diagnostic of severe oligohydramnios in 21 weeks of gestation by the gynecologist. © Georg Thieme Verlag KG Stuttgart · New York.

  11. Case report of a rarely seen long-segment middle aortic syndrome.

    Science.gov (United States)

    Yakut, Kahraman; Erdoğan, İlkay

    2017-03-01

    Middle aortic syndrome (MAS) follows a course with distal thoracic and abdominal aorta stenosis. It is a rare disease that is usually diagnosed after the first decade of life. Clinical reflection of MAS is often in the form of hypertension and claudication in the lower extremities. Its etiology is unclear, but is known to be associated with congenital or acquired diseases. This pathology, which is accompanied by malignant hypertension, often does not respond to medical treatment. In patients with MAS, surgical treatment is first line recommendation to prevent complications such as hypertension, heart failure, intracranial bleeding, or aortic rupture. In order to draw attention to this disease, presently described is case of high blood pressure detected during routine examination of a child who had no complaint, and discovery of long-segment stenosis in the abdominal aorta identified with echocardiography and conventional angiography.

  12. Giant osteoma of the skull vault: A rare case of mixed variety

    Directory of Open Access Journals (Sweden)

    Harisha P.N.

    2016-12-01

    Full Text Available Osteoma is the most common primary bone tumor in the craniofacial skeleton. However, most of these are small, asymptomatic and arise from the facial bones or in relation to the paranasal sinuses. Cranial vault osteomas, that too giant and symptomatic are much rarer. We report a case of sixty year-old gentleman presented with a very slowly increasing, painless, hard swelling on the left side of his head. Computerized tomography scan showed the left parietal calvarial tumor to be having large exostotic and enostotic components. He underwent an en-bloc excision of the tumor and cranioplasty. Giant, symptomatic cranial vault osteoma with concurrent exostotic and enostotic components is extremely rare. These lesions can be safely and completely excised with careful planning and attention to detail.

  13. Cytological diagnosis of solitary plasmacytoma of the skull: A rare case report

    Directory of Open Access Journals (Sweden)

    Mohamad Banyameen Iqbal

    2015-01-01

    Full Text Available Solitary plasmacytoma (SPC of the skull (SPS is rare, and only a few cases have been reported in the literature so far. Plasmacytoma of the skull has a wide spectrum of pathology, including a quite benign, SPC, and an extremely malignant, multiple myeloma at the two ends of the spectrum. SPC of bone including SPS is characterized by a radiologically solitary bone lesion, neoplastic plasma cells in the biopsy specimen, fewer than 5% plasma cells in bone marrow, <2.0 g/dl monoclonal protein in the serum when present and negative urine test for Bence Jones protein (monoclonal light chain. For diagnosing, a comprehensive examination and analysis, which includes radiological examination, immunoglobulin, biochemistry, test for Bence Jones protein in the urine and bone marrow is needed.

  14. A Rare Case of Intermittent Claudication Associated with Impaired Arterial Vasodilation

    Directory of Open Access Journals (Sweden)

    J. J. Posthuma

    2017-01-01

    Full Text Available Exercise-related intermittent claudication is marked by reduced blood flow to extremities caused by either stenosis or impaired vascular function. Although intermittent claudication is common in the elderly, it rarely occurs in the young and middle-aged individuals. Here, we report a case of exercise-related claudication in a 41-year-old woman, in the absence of overt vascular pathology. Using a series of imaging and functional tests, we established that her complaints were due to impaired arterial vasodilation, possibly due to a defect in nitrous oxide-mediated dilation. The symptoms were reversible upon administration of a calcium antagonist, showing reversibility of the vascular impairment. Identification of reversible vascular “stiffness” merits consideration in young and otherwise healthy subjects with claudication of unknown origin.

  15. Recurrent diabetic muscle infarction, a rare complication of diabetes: a case report

    Directory of Open Access Journals (Sweden)

    Tariq Bhat

    2017-01-01

    Full Text Available Diabetic muscle infarction is a rare complication of diabetes mellitus that presents as a localized, exquisitely painful swelling and limited range of motion of the involved extremity. The onset is usually acute, persists for several weeks and resolves spontaneously over several weeks to months without the need for intervention. However, as diabetes mellitus is an immunocompromised state and any painful swelling in the limbs is often taken as infectious in aetiology, the patient is inadvertently investigated with invasive procedures and is started on unnecessary antibiotics, adding to the burden of management. Keeping in view the low threshold for starting antibiotics in painful limb swelling in diabetes mellitus in our setting, we hereby describe a case of recurrent painful diabetic muscle infarction, first involving the right upper and later the right lower limb, managed with physical rest and analgesics. This case emphasizes that the treating physician keep this rare complication of diabetes mellitus in consideration in the respective clinical scenario and adopt a less aggressive (a noninvasive method like ultrasound rather than a more aggressive (an invasive method like muscle biopsy approach in diagnosis and take a similar approach towards management.

  16. Laugh syncope as a rare sub-type of the situational syncopes: a case report

    Directory of Open Access Journals (Sweden)

    Nishida Katsufumi

    2008-06-01

    Full Text Available Abstract Introduction Laughter is a good medicine; it enhances cardiovascular health and the immune system. What happens, however, if a person laughs too much or the laughter becomes out of control? Laughter-induced syncope is rare and likely goes unrecognized by many health care providers. It is thought to be another form of Valsalva-induced syncope. Case presentation We report the case of a 56-year-old, moderately obese (body mass index of 35 man with a past medical history of sleep apnea, hypertension and hyperlipidemia who suffered from syncope secondary to intense laughter. The patient also had a history of syncope in the distant past when he collapsed on the floor for several seconds. Treadmill stress testing after the incident revealed no arrhythmia or ischemic disease, although he complained of dizziness after the test and a sudden drop in blood pressure was noted. Conclusion Laughter-induced or gelastic syncope is extremely rare. It is thought to be a sub-type of the situational syncopes.

  17. Clozapine-induced interstitial nephritis - a rare but important complication: a case report

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    Hunter Robert

    2009-08-01

    Full Text Available Abstract Introduction Given the limited range of effective drug treatments for patients with schizophrenia, increasing numbers of patients, often termed 'treatment-resistant' are prescribed clozapine. While the induction of neutropenia or agranulocytosis by clozapine is well appreciated, other rare potentially fatal adverse reactions may also occur including acute interstitial nephritis as reported in this case. Case presentation A 57-year-old Caucasian woman with treatment-resistant chronic schizophrenia developed acute renal failure following initiation of treatment with clozapine. The adverse reaction occurred after only four doses of the drug had been administered (titrated from 12.5 to 25 mg per day. After clozapine had been withdrawn, the patient's renal function returned to normal with no other changes to medication. The patient had been exposed to clozapine about 4 years previously when she had developed a similar reaction. Conclusion Renal reactions to clozapine are extremely rare but, if not recognized promptly, may prove fatal. Psychiatrists need to be aware of this possible complication when clozapine is initiated.

  18. A Rare Case of Diffuse Idiopathic Pulmonary Neuroendocrine Cell Hyperplasia

    Directory of Open Access Journals (Sweden)

    Godwin Ofikwu

    2015-01-01

    Full Text Available Diffuse idiopathic pulmonary neuroendocrine cell hyperplasia (DIPNECH is a rare clinical condition with only about 100 cases reported in the literature. It is characterized by primary hyperplasia of pulmonary neuroendocrine cells (PNECs which are specialized epithelial cells located throughout the entire respiratory tract, from the trachea to the terminal airways. DIPNECH appears in various forms that include diffuse proliferation of scattered neuroendocrine cells, small nodules, or a linear proliferation. It is usually seen in middle-aged, nonsmoking women with symptoms of cough, dyspnea, and wheezing. We present a 45-year-old, nonsmoking woman who presented with symptoms of DIPNECH associated with bilateral pulmonary nodules and left hilar adenopathy. Of interest, DIPNECH in our patient was associated with metastatic pulmonary carcinoids, papillary carcinoma of the left breast, oncocytoma and angiomyolipoma of her left kidney, and cortical nodules suggestive of tuberous sclerosis. She had video assisted thoracoscopic surgery (VATS, modified radical mastectomy with reconstruction, and radical nephrectomy. She is currently symptom-free most of the time with over two years of follow-up.

  19. Fatal thrombocytopenia: A rare case with possible explanation

    Science.gov (United States)

    Barik, Ramachandra; Patnaik, A. N.; Gulati, A. S.

    2012-01-01

    A 22 year old male presented with breathlessness on exertion, ecchymosis, jaundice and features of worsening right heart failure for the last fifteen days. On physical examination, he had a mid diastolic murmur in the tricuspid area and an ejection systolic murmur in the pulmonary area. Bone marrow histopathology report showed an increased in megakaryocytes count. Routine investigations reports were normal. Echocardiography and computerized tomography (CT) revealed a single mobile large intra cardiac mass originating from the right atrium and causing dynamic obstruction of the right ventricular inflow and outflow tract. Associated fatal thrombocytopenia did not respond to intravenous steroids or platelet transfusion. Patient could not be operated because of very low platelet count, and died during hospital stay before excision biopsy could be done. Pathological autopsy was not done. This is a rare case, as the fatal thrombocytopenia observed here was the result of mechanical effects like frictional and shear force, which can be attributed to the physical presence of a large intra cardiac mass resulting in obstruction to flow. PMID:22629036

  20. A RARE CASE OF IPSILATERAL HIP AND KNEE DISLOCATION

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    Deepak

    2015-06-01

    Full Text Available High velocity road traffic accidents leads to complicated lower limb injuries. Such injuries demand highly experienced surgeon and are associated with high morbidity and mortality. Hip or knee dislocations are two different orthopaedic emergencies. Concomitant fracture dislocation of the hip and knee is rare and very few cases have been reported in the literature. A 45 year old man with history of fall from motorcycle came to the casualty. He had ipsilateral hip and knee dislocation. Immediately patient was shifted to operation theatre and closed reduction was performed under general anaesthesia. Reduction was confirmed under fluoroscopy and post-operative x-rays were taken. The functional results were excellent. After 2 months patient made an uncomplicated recovery and had satisfactory functional outcome with right hip having 110⁰ flexion and right knee flexes to 120⁰.There was no neurological deficit. The urgency, that the treating surgeon shows in managing these injuries, significantly affects the prognosis and outcome finally achieved by these patients (golden period in reducing the hip joint has been described to be 6 hours.

  1. A Rare Case of Cranial Osteomyelitis Caused by Proteus Vulgaris

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    Hakan Uslu

    2011-03-01

    Full Text Available Osteomyelitis of the calvarial bones can cause serious complications such as brain abscess, due to the close proximity to adjacent brain structures. Development of the purulent secretion in surgery and traumatic scalp injuries must be considered as a possibility of osteomyelitis possibility. Generally gram positive, rarely gram negative bacteria and mix agents, can be isolated in infection. Especially chronic pyogenic osteomyelitis agents can be isolated from chronic infections such as tuberculosis. In cranial osteomyelitis diagnosis, radiological diagnosis has a very important place together with the clinical diagnosis. However, infection can usually show late findings radiologically. In treatment, antibiotic treatment is absolutely essential as well as removal of the infected part of the bone. Due to antibiotic treatment lasting between 6-12 weeks, organizing the antibiotic protocols according to the results of culture-antibiograms, which were provided from purulent secretions, has the most important role in the success of surgical treatment. In Proteus sp. infections, for choice of suitable treatment, determination of the type of bacteria is important. For exact diagnosis, histopathological examination of the bone tissue must be carried out. In this report, a case with cranial osteomyelitis caused by Proteus vulgaris which is a gram negative bacteria causing anaerobic infections and classified in the Enterobacteriaceae family is presented. The patient was treated with surgery and appropriate antibiotics. Early recognition of this condition, planning the best treatment strategy and taking precautions to prevent complications, is mandatory for a better outcome.

  2. Unusual clinical presentation of rare case of vaginal leiomyoma: a case report

    OpenAIRE

    Ishrat Zuber; Purnima K. Nadkarni; Aditi A. Nadkarni; Akshay Nadkarni

    2016-01-01

    Primary vaginal leiomyoma are rare and usually arise from anterior vaginal wall, approximately 330 cases of vaginal fibroid reported in world literature. Vaginal myoma usually presented as discharge per vaginum, abnormal bleeding, pain lower abdomen, dyspareunia etc. We report a case of primary vaginal leiomyoma arising from lateral vaginal wall which is presented clinically as pain in hip joint and radiating to ipsilateral leg which is unusual clinical presentation creating diagnostic dilemm...

  3. Case Report Meleney's Ulcer; A Rare but Fatal Abdominal Wall ...

    African Journals Online (AJOL)

    KIGZ

    Meleney's Ulcer; A Rare but Fatal Abdominal Wall Disease Complicating ... Meleney's ulcer or post operative synergistic bacterial gangrene is a rare form of ... Central venous catheterization and parenteral ... is no record of full recovery from the intra-abdominal pathology before the ... chronic undermining ulcer of Meleney.

  4. Lymphoepithelioma-like carcinoma of ureter—A rare case report and review of the literature

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    Sheng-Chen Wen

    2012-09-01

    Full Text Available Lymphoepithelioma-like carcinoma (LELC is best known to occur in the nasopharynx. When LELC occurs in the urinary tract, this extremely rare neoplasm most commonly affects the bladder but has also been reported in the renal pelvis, ureter, prostate [1], and urethra [2]. We present a case of LELC arising in the right proximal ureter of a 64-year-old male patient with hydronephrosis and nausea. Computed tomography demonstrated right ureter tumor. On biopsy, the patient was diagnosed with infiltrating urothelial carcinoma. An operation consisting of right nephroureterectomy and bladder cuff removal was carried out. The pathologic examination showed pure subtype of LELC, pT3N0. Unlike lymphoepithelioma in the nasopharynx, immunohistochemical analysis of this urinary LELC was negative for the Epstein–Barr virus. No disease progression was noted at 6 months’ follow-up. Only eight previous cases of LELC involving the ureter have been reported, and a review of the available literature and a summary of ureter cases are presented here. This is the first report of a ureteral LELC case and third urothelial LELC cases [3] in Taiwan.

  5. SERTOLI-LEYDIG CELL TUMOR; A RARE CASE IN A POSTMENOPAUSAL PATIENT – CASE REPORT

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    Petra Krajnc

    2018-02-01

    Full Text Available Background. Sertoli-Leydig cell tumors belong to the group of sex cord stromal tumors of the ovary. They account for less than 0.5 % of all ovarian tumors and occur primarily in young women between 20 and 30 years of age. This type of tumors can secrete androgens, causing virilisation, and are extremely rarely presented in postmenopausis. Methods. A 73-year old multiparous woman was presented to our institution with complaints of abdominal distention and abdominal pain in her lower abdomen. On physical examination, she had a large, fixed palpable abdominal mass, approximately 20 cm in diameter, arising from the pelvis. The laboratoric tests revealed an elevated level of CA125 of 221.3 U/ml of serum. The ultrasound showed a complex cystic and solid pelvic tumor. There was no sign of ascites. Her hormonal status was within normal range and she also showed no signs of virilisation. On laparotomy a complex left ovarian mass, measuring 30 × 27 × 15 cm was found and sent to frozen section. The result of frozen section was a malignant tumor of unknown origin, therefore a radical surgical procedure was performed. The histopathological examination established the diagnosis of a malignant Sertoli-Leydig cell tumor of the left ovary, of intermediate differentiation. Other removed tissue was free of malignant cells. The early postoperative course was uneventful and the patient was released from hospital 10 days after surgery. However, she returned to our institution 16 days after surgery due to a proximal thrombosis of v. saphena magna. The patient was treated with low-molecularweight heparin and later warfarin for 6 weeks post operation. 16 months after the operation she was symptomatically treated for severe microcytic anemia. She showed no signs of a relapse. 27 months after primary surgery she was operated for the second time due to acute bowel obstruction. She had large masses of necrotic tumor removed from abdomen and transversostomia was performed

  6. Severe Rhabdomyolysis without Systemic Involvement: A Rare Case of Idiopathic Eosinophilic Polymyositis

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    Ayesha Farooq

    2015-01-01

    Full Text Available Introduction. Eosinophilic polymyositis (EPM is a rare cause of rhabdomyolysis characterized by eosinophilic infiltrates in the muscle. We describe the case of a young patient with eosinophilic polymyositis causing isolated severe rhabdomyolysis without systemic involvement. Case Presentation. A 22-year-old Haitian female with no past medical history presented with progressive generalized muscle aches without precipitating factors. Examination of the extremities revealed diffuse muscle tenderness. Laboratory findings demonstrated peripheral eosinophilia and high creatinine phosphokinase (CPK and transaminase levels. Workup for the common causes of rhabdomyolysis were negative. Her CPK continued to rise to greater than 100,000 units/L so a muscle biopsy was performed which showed widespread eosinophilic infiltrate consistent with eosinophilic polymyositis. She was started on high dose systemic corticosteroids with improvement of her symptoms, eosinophilia, and CPK level. Discussion. This case illustrates a systematic workup of rhabdomyolysis in the presence of peripheral eosinophilia. Many differential diagnoses must be considered before establishing a diagnosis of idiopathic eosinophilic polymyositis. To our knowledge, our case of eosinophilic polymyositis is unique as it presented with severe rhabdomyolysis without another organ involvement. Clinicians should maintain a high index of suspicion for this physically debilitating disease to aid in prompt diagnosis.

  7. A rare case of retroperitoneal malignant triton tumor invading renal vein and small intestine

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    Mijović Žaklina

    2013-01-01

    Full Text Available Introduction. Malignant Triton tumor is a very rare malignant peripheral nerve sheath tumor with rhabdomyosarcomatous differentiation. Most of those tumors occur in patients with von Recklinghausen’s disease or as a late complication of irradiation and commonly seen in the head, neck, extremities and trunk. Case report. We reported retroperitoneal malignant Triton tumor in a 57-year-old female patient. Skin lesions were not present, and there was no family history of neurofibromatosis or previous irradiation. The presented case is one of a few recorded in the specialized literature that occurs in the retroperitoneal space in sporadic form. In this case, tumor consisted of a multilobular mass was in close relation with the abdominal aorta and inferior vena cava and involved the renal vein with gross invasion of the small intestine. The patient underwent total resection of the tumor and left nefrectomy was performed. The small intestine 10 cm in length was also resected and end-to-end anastomosis was conducted. The postoperative course was uneventful and the patient was discharged from the hospital ten days after the surgery. Conclusion. Diagnostically, it is crucial to recognize this uncommon histological variant because malignant Triton tumor has a worse prognosis than classic malignant peripheral nerve sheath tumor does. The use of the immunohistochemistry is essential in making the correct diagnosis. Only appropriate pathological evaluation supported by immunostaining with S-100 protein and desmin confirmed the diagnosis. Aggressive surgical management treatment improves the prognosis of such cases with adjuvant radiotherapy.

  8. Tree species traits but not diversity mitigate stem breakage in a subtropical forest following a rare and extreme ice storm.

    Directory of Open Access Journals (Sweden)

    Karin Nadrowski

    Full Text Available Future climates are likely to include extreme events, which in turn have great impacts on ecological systems. In this study, we investigated possible effects that could mitigate stem breakage caused by a rare and extreme ice storm in a Chinese subtropical forest across a gradient of forest diversity. We used Bayesian modeling to correct stem breakage for tree size and variance components analysis to quantify the influence of taxon, leaf and wood functional traits, and stand level properties on the probability of stem breakage. We show that the taxon explained four times more variance in individual stem breakage than did stand level properties; trees with higher specific leaf area (SLA were less susceptible to breakage. However, a large part of the variation at the taxon scale remained unexplained, implying that unmeasured or undefined traits could be used to predict damage caused by ice storms. When aggregated at the plot level, functional diversity and wood density increased after the ice storm. We suggest that for the adaption of forest management to climate change, much can still be learned from looking at functional traits at the taxon level.

  9. Could Buerger's disease cause nonarteritic anterior ischemic optic neuropathy?: a rare case report.

    Science.gov (United States)

    Korkmaz, Anil; Karti, Omer; Top Karti, Dilek; Yüksel, Bora; Zengin, Mehmet Ozgur; Kusbeci, Tuncay

    2018-04-05

    We present an interesting case with nonarteritic anterior ischemic optic neuropathy (NAION) accompanied by Buerger's disease. A 43-year-old man was referred to our neuro-ophthalmology clinic with a complaint of visual deterioration in the left eye that started 5 days ago. He suffered from Buerger's disease, and he had acute pain in the right lower limb below the knee. His best corrected visual acuity was 10/10 in the right eye and 2/10 in the left eye by Snellen chart. There was a relative afferent pupil defect in the left eye. The right optic disc was normal on fundus examination, and blurring, hemorrhagic swelling was found at the left optic disc. Inferior altitudinal visual field defect was observed in the left eye. Neurological examination was normal. Computed tomography angiography scan revealed occlusion in the right posterior tibial artery. Brain imaging and laboratory tests such as blood analyses, genetic screening, coagulation, and lipid panels were unremarkable. NAION may occur in patients with Buerger's disease, but it is extremely rare. Therefore, clinicians should be aware of this rare association.

  10. Penile duplication and two anal openings; report of a very rare case.

    Science.gov (United States)

    Bakheet, Mohamed Abdel Al M; Refaei, Mohammad

    2012-03-01

    Penile duplication (diphallus) is an extremely rare disorder. It is almost always associated with other malformations like double bladder, exstrophy of the cloacae, imperforate anus, duplication of the rectosigmoid and vertebral deformities. Meanwhile anal canal duplication, the most distal and least common duplication of the digestive tube and is a very rare congenital malformation. A 21 days old Egyptian neonate is reported with complete penile duplication and two scrotums with each one carrying two palpable testes. Both penises have normal shaft with normally located meatus. Clear urine voids from both meati spontaneously. The child had also a fold of redundant skin about 4×5 cm at the anal region in which two separate anal openings are present. In rectal examination we found two normal anuses passing stool spontaneously. Ascending (voiding) cystourethrography revealed two penises with two separate meatuses and one bladder from which the two urethras go out separately. Intravenous pyelogram (IVP) revealed two normal kidneys and ureters. Barium study revealed duplication of rectum and colon, otherwise normal GIT. In our review of the literature, we did not come across any other case of this variety of the penile duplication and congenital presence of two anuses. Unfortunately the patient expired before any surgical correction.

  11. A very rare case report of long-term survival: A patient operated on in 1994 of glioblastoma multiforme and currently in perfect health

    Directory of Open Access Journals (Sweden)

    Riccardo Caruso

    2017-01-01

    Conclusion: The fact that there are extremely rare cases of long-term survival and even zero recurrence of the glioblastoma should serve as a stimulus to continue the research effort and not give up the fight against this tumor on a day-to-day basis.

  12. Well-circumscribed deep-seated lipomas of the upper extremity. A report of 13 cases.

    Science.gov (United States)

    Elbardouni, A; Kharmaz, M; Salah Berrada, M; Mahfoud, M; Elyaacoubi, M

    2011-04-01

    The purpose of this study is to determine if giant size is of bad prognosis in deep lipomas of the upper extremity. We report a retrospective study of 13 patients with deep-seated lipomas of the upper extremity treated during the period from April 1997 to April 2008. We evaluated the clinical and radiological characteristics, treatment and evolution profile of these patients. There were 10 women and three men, with an average age of 53 years (range 30-79 years). Seven of these lipomas were in the arm, one in the shoulder, and five in the forearm. Six lipomas were intramuscular, six intermuscular (three of them being attached to bone and labelled parosteal lipoma) and one epivaginal lipoma of the flexor tendon sheath. All patients presented a progressive slow-growing mass that was associated with radial paralysis in one case and carpal tunnel syndrome in one case. Plain radiographs showed a radiolucent soft-tissue image in all cases and an associated osteochondroma in one parosteal lipoma. Computer tomography (CT) or magnetic resonance imaging (MRI) suggested the lipomatous nature and benign characteristics of these deep lipomas that were giant in all cases (mean size: 7 cm). Lipoma marginal excision was performed and histopathological examination demonstrated features consistent with a benign lipoma. There was good function and no clinical recurrence was observed after a mean follow-up of three years. Giant deep-seated lipomas of the upper extremity are uncommon and can be intermuscular or intramuscular. A painless soft-tissue mass is the most frequent chief complaint. MRI with fat suppression suggests the diagnosis and studies the extension of deep lipoma. Marginal excision is the treatment of choice and histopathology eliminates diagnosis of well-differentiated liposarcoma. Appropriate evaluation of deep lipoma is to rule out malignancy by systematically performing MRI and biopsy. In contrast to deep-seated lipomas of the lower extremity or the retroperitoneal

  13. Sirenomelia sequence: early prenatal diagnosis of one rare case associated with acardiac malformation.

    Science.gov (United States)

    Zanforlin Filho, Sebastião M; Guimarães Filho, Hélio A; Araujo Júnior, Edward; Pires, Cláudio R; Mattar, Rosiane; Nardozza, Luciano M M

    2007-04-01

    Sirenomelia sequence is a very rare congenital malformation, with incidence of around 1.5-4.2 per 100,000 births. Prenatal diagnosis of sirenomelia in the first trimester is rare; there are only five cases reported for the present, and the association of sirenomelia with acardiac malformation is even rarer. We present a rare case of sirenomelia associated with acardiac malformation detected in the first trimester through combined two-dimensional, three-dimensional and color Doppler sonographies.

  14. Rare case of transverse testicular ectopia - Case report and review of literature.

    Science.gov (United States)

    Raj, Vinod; Redkar, Rajeev; Krishna, Swathi; Tewari, Shruti

    2017-01-01

    This case report has been reported in line with the SCARE criteria; Consensus-based surgical case report guidelines of International Journal of Surgery 2016. Transverse Testicular Ectopia (TTE) is a rare condition which manifests with unilateral undescended testis and contralateral hernia. Till now around 100 cases have been described in the literature. The management depends on the anatomy of the vas, vessels and testis found on surgical exploration. An algorithm exists for its management and we propose a modified algorithm for management of TTE. Five year male presented with complaints of unilateral undescended testis on the right and hernia on the left. Clinically the right testis was impalpable and left testis palpable in the left hemiscrotum and fluid hernia on the same side. Transverse testicular ectopia is a rare condition presenting with UDT and contralateral inguinal hernia. Although more than 100 cases have been described in the literature so far, those managed with a transeptal contralateral orchidopexy are two cases to the best of our knowledge. TTE was first described by Von Lenhossek in 1886. The mean age of presentation is around 4 years and most of the cases are diagnosed on surgical exploration. The management of TTE remains controversial even though an algorithm has been described for its management due to its varied presenting scenarios. TTE is a rare condition which requires high index of suspicion for diagnosis preoperatively. Whenever suspected we recommend an USG and/or MRI prior to diagnostic laparoscopy and proceed with orchidopexy. Diagnostic laparoscopy is both helpful in diagnosis and management. Transeptal contralateral orchidopexy gives good tension free fixation of testes in the scrotum.

  15. A rare case with synchronous gastric gastrointestinal stromal tumor, pancreatic neuroendocrine tumor, and uterine leiomyoma.

    Science.gov (United States)

    Arabadzhieva, Elena; Yonkov, Atanas; Bonev, Sasho; Bulanov, Dimitar; Taneva, Ivanka; Vlahova, Alexandrina; Dikov, Tihomir; Dimitrova, Violeta

    2016-11-15

    Although gastrointestinal stromal tumors (GISTs) are the most common mesenchymal tumors of the gastrointestinal tract, they comprise less than 1% of all gastrointestinal tumors. Neuroendocrine tumors (NET) of the gastro-enteropancreatic system are also rare, representing about 2% of all gastrointestinal neoplasms. Pancreatic localization of NET is extremely uncommon-these tumors are only 1-5% of all pancreatic cancers. We describe an unusual case with triple tumor localization-a gastric tumor, a formation in the pancreas, which involves the retroperitoneal space, and a uterine leiomyoma. The exact diagnosis was confirmed with immunohistochemical study after surgical treatment of the patient. Distal pancreatic resection, splenectomy, partial gastrectomy, omentectomy, and hysterectomy were performed. The histological examination proved an epithelioid type of gastric GIST. Immunostaining showed focal positive expression of c-kit and no mitotic figures per 50 HPF. Histology of the pancreatic and retroperitoneal formation proved a well-differentiated NET with origin from the islets of Langerhans. The immunohistochemical study demonstrated co-expression of chromogranin A and synaptophysin. This is the fourth case published so far of a patient with synchronous pancreatic NET and gastric GIST. The main objective of the study is to present a unique case because we have not found any reports for coexistence of the described three types of neoplasm, as in our patient, and we hope that it will be valuable in the future investigations about the genesis, diagnosis, and treatment of these types of tumors.

  16. Brown′s syndrome with ocular albinism: Case report of a rare presentation and literature review

    Directory of Open Access Journals (Sweden)

    Soumya Nambiar

    2015-01-01

    Full Text Available We report a rare case of Brown′s syndrome associated with ocular albinism in a 10-year-old boy. This report highlights the importance of further analysis of cases of Brown′s syndrome and throws some light on the etiology and association of this rare condition.

  17. A case of clear cell sarcoma-A rare malignancy

    DEFF Research Database (Denmark)

    Juel, Jacob; Ibrahim, Rami Mossad

    2017-01-01

    INTRODUCTION: Clear cell sarcoma (CCS) is a rare tumour of the soft tissue often misdiagnosed, as it shares characteristics with malignant melanoma (MM). Previously, CCS has been characterised, as malignant melanoma of the soft tissue, contemporary immunohistochemical techniques, however, have made...

  18. CASE REPORT Macrodystrophia lipomatosa – MR imaging of a rare ...

    African Journals Online (AJOL)

    This rare congenital abnormality occurs most frequently in the .... 1st, 2nd and 3rd digits owing to gross hypertrophy of fat and soft tissue in a striated pattern. ... a part of generalised hamartomatous disorder featuring fat in the soft tissues and ...

  19. Kindler′s syndrome: A rare case report

    Directory of Open Access Journals (Sweden)

    Neelam Suman

    2014-01-01

    Full Text Available Kindler syndrome is a rare hereditary disorder, associated with skin fragility. The syndrome involves the skin and mucous membrane with radiological changes. The genetic defect has been identified on the short arm of chromosome 20. This report describes a 16-year-old patient with classical features like blistering and photosensitivity in childhood and the subsequent development of poikiloderma.

  20. Obturator hernia: a rare case of acute mechanical intestinal obstruction.

    Science.gov (United States)

    Aydin, Ibrahim; Yucel, Ahmet Fikret; Pergel, Ahmet; Sahin, Dursun Ali

    2013-01-01

    Obturator hernia is a rare type of pelvic hernia which generally occurs in elderly patients with accompanying diseases. Because it is difficult to diagnose before surgery, the morbidity and mortality rates for obturator hernia are high. The most common symptom is strangulation combined with mechanical intestinal obstruction.

  1. Obturator Hernia: A Rare Case of Acute Mechanical Intestinal Obstruction

    Directory of Open Access Journals (Sweden)

    Ibrahim Aydin

    2013-01-01

    Full Text Available Obturator hernia is a rare type of pelvic hernia which generally occurs in elderly patients with accompanying diseases. Because it is difficult to diagnose before surgery, the morbidity and mortality rates for obturator hernia are high. The most common symptom is strangulation combined with mechanical intestinal obstruction.

  2. Congenital Cystic Adenomatoid Malformation of Lung-Rare Case Report

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    N. S. Kamakeri

    2016-10-01

    Full Text Available Congenital cystic adenomatoid malformation of lung associated with Cystic dysplasia of kidney, cystic disease of liver with mixed gonadal dysgenesis is rare and is not reported in literature so far. Hence an attempt is made to present this rarest entity.

  3. Case Report: Corneal Pyogenic Granuloma: Rare Complication of ...

    African Journals Online (AJOL)

    Slit lamp examination showed vascularized central corneal mass with surrounding stromal infiltrates. The mass was excised, and histopathological examination confirmed pyogenic granuloma of the cornea. Conclusion: Corneal pyogenic granuloma could be a rare complication of infectious keratitis. Therefore, it should be ...

  4. Syringomatous carcinoma: Case report of a rare tumor entity | El ...

    African Journals Online (AJOL)

    Syringomatous carcinoma is an extremely invasive tumor, locally destructive and slowly growing adnexal tumour, derived from eccrine sweat glands. It is often mistaken, both clinically and microscopically, for other benign and malignant entities. The tumour recurrence is high due to extensive perineural invasion, but

  5. Exertional Rhabdomyolysis after an Extreme Conditioning Competition: A Case Report

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    Ramires Alsamir Tibana

    2018-04-01

    Full Text Available This case report describes an instance of exercise-induced rhabdomyolysis caused by an extreme conditioning program (ECP competition. A 35-year-old female presented with abdominal pain and soreness, which began one day after she completed two days of ECPcompetition composed of five workouts. Three days after competition, creatine kinase (CK was 77,590 U/L accompanied by myalgia and abnormal liver function tests, while renal function was normal and this resulted in a diagnosis of rhabdomyolysis. A follow-up examination revealed that her serum level of CK was still elevated to 3034 U/L on day 10 and 1257 U/L on day 25 following the ECP competition. The subject reported myalgia even up to 25 days after the ECP competition. Exertional rhabdomyolysis can be observed in ECP athletes following competition and highlights a dangerous condition, which may be increasing in recent years due to the massive expansion of ECP popularity and a growing number of competitions. Future research should investigate the causes of rhabdomyolysis that occur as a result of ECP, especially training methods and/or tasks developed specifically for these competitions.

  6. A rare case of retained fourth molar teeth in maxilla and mandible. Case report

    Directory of Open Access Journals (Sweden)

    Rahnama Mansur

    2014-06-01

    Full Text Available The study presents a case of the rarely occurring totally retained fourth molar teeth simultaneously in maxilla and mandible. The appearance of supernumerary teeth is a relatively uncommon dental anomaly and it is rare for patients to have impacted fourth molars in two quadrant. The aim of this work is to describe the presence of unilateral (right fourth molars in the maxilla and the mandible in a young female patient aged 24 years. Orthopantomogram revealed impacted lower third molars but also unerupted unilateral (right upper and lower fourth molars. Before orthodontic treatment, the patient was subsequently admitted for removal of third and fourth impacted upper and lower molars under local anesthesia.

  7. Acute neonatal parotid abscess: A rare case report

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    Shreesh Kolekar

    2016-01-01

    Full Text Available Acute suppurative parotitis is uncommon in children and is very rare in neonates. Most common organism isolated is Staphylococcus aureus. We present a 15-day-old full-term breast-fed female neonate with left-sided acute parotid abscess. The baby presented with a left preauricular swelling, pain and redness. Pus was exuded from left Stensen's duct on compression of the gland externally. Early diagnosis and proper intravenous antibiotics are the keys to the treatment.

  8. Acute neonatal parotid abscess: A rare case report

    Science.gov (United States)

    Kolekar, Shreesh; Chincholi, Tejas S.; Kshirsagar, Ashok; Porwal, Narendra

    2016-01-01

    Acute suppurative parotitis is uncommon in children and is very rare in neonates. Most common organism isolated is Staphylococcus aureus. We present a 15-day-old full-term breast-fed female neonate with left-sided acute parotid abscess. The baby presented with a left preauricular swelling, pain and redness. Pus was exuded from left Stensen's duct on compression of the gland externally. Early diagnosis and proper intravenous antibiotics are the keys to the treatment. PMID:28051052

  9. A Rare Syndrome, Pentalogy of Cantrell: Case Report

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    Atilla Karateke

    2014-02-01

    Full Text Available Pentalogy of Cantrell is a rare syndrome; characterized by ectopia cordis with omphalocele and anterior wall defect of thoraco-abdominal. Prognosis of fetuses with this syndrome of which etiopathogenesis is unknown, is poor. The patient who admitted to our clinic at 25.th week of gestation, had all signs of pentalogy of Cantrell in ultrasonography. In this article, we discussed the pentalogy of Cantrell with review of the literature.

  10. Rare upper gastrointestinal hemorrhage of cetuximab: A case report.

    Science.gov (United States)

    Duan, Shi-Jie; Gao, Zi-Ming; Wang, Peng-Liang; Gong, Bao-Cheng; Huang, Han-Wei; Luo, Lei; Wang, Xin; Xing, Ya-Nan; Xu, Hui-Mian; Liu, Fu-Nan

    2017-12-01

    cetuximab, an epidermal growth factor receptor inhibitor, is a targeted therapeutic regimen of colorectal cancers. Several common adverse effects have been found, such as cutaneous or gastrointestinal toxicity. However, according to the articles had been published, upper gastrointestinal bleeding (UGIB) is considered to be rare and its mechanism remains unclear. In this report, we presented a 42-year-old male patient with advanced recto-sigmoid cancer. After palliative operation, the patient suffered from complete upper gastrointestinal (GI) obstruction, which was induced by extensive abdominal metastasis of the tumor. Considering his poor condition, we chose the targeted drug, cetuximab, as his further treatment. But after the application of cetuximab, the UGIB immediately happened twice in this patient. UGIB, as a rare complication of cetuximab, occured to the patient. We stopped the bleeding with thrombin, hemocoagulase and somatostatin and suspended the subsequent treatment plan of cetuximab. At the same time, anti-shock treatment was given immediately. He was died of respiratory and circulatory failure caused by UGIB and advanced tumor eventually. UGIB should be considered as a rare but severe complication of cetuximab. When cetuximab is applied for patients with advanced colon tumors, more cautions should be required if the patients are accompanied by upper gastrointestinal obstruction. In addition, for those patients who suffered from UGIB recently, cetuximab should be prohibited if the Rockall score ranged > 5 points. Copyright © 2017 The Authors. Published by Wolters Kluwer Health, Inc. All rights reserved.

  11. Report of a Case and Review of Literature of Internal Hernia through Peritoneal Defect in Pouch of Douglas: A Rare Occurrence.

    Science.gov (United States)

    Muthukumar, Vamseedharan; Venugopal, Sarveswaran; Subramaniam, Surees Kumar

    2017-01-01

    Intestinal obstruction attributable to internal hernia as a cause is a rare phenomenon with a reported incidence of 0.6%-5.8%. Internal hernias ensuing as a result of defect in the pouch of Douglas is extremely rare with only six such cases reported so far in the literature. We present a case of 74-year-old posthysterectomy status female who presented with features of intestinal obstruction. Intraoperatively, the site of obstruction was found to be a rent in the peritoneum of the pouch of Douglas through which a loop of ileum was found herniating. The viability of the bowel was confirmed, and the defect was closed. The postoperative course was uneventful. This report presents an extremely rare type of internal hernia caused by defect in the pouch of Douglas and review of the literature so far available.

  12. Isolated fracture of pisiform: case report of a rare injury of wrist

    African Journals Online (AJOL)

    Arun Kumar Agnihotri

    ABSTRACT: Isolated fracture of the pisiform is an extremely rare injury. Generally fractures of the pisiform are associated with fractures of other carpal bones or the distal radius. Fractures of the carpals and metacarpals account for roughly 6% of all fractures. The average incidence of pisiform fractures is 0.2% of all carpal ...

  13. Permanent neonatal diabetes mellitus - a case report of a rare cause ...

    African Journals Online (AJOL)

    Diabetes mellitus is a metabolic disease characterised by chronically high glucose levels. Genetic factors have been implicated in the aetiology following mutations in a single gene. An extremely rare form of diabetes mellitus is monogenic diabetes, a subset of which is permanent neonatal diabetes, and is usually ...

  14. Can pneumocephalus present as flashes of light? A rare case report

    Directory of Open Access Journals (Sweden)

    Santanu Ghosh

    2017-01-01

    Full Text Available Orbital emphysema and associated pneumocephalus usually result from trauma to orbital bones or due to sinus disease, allowing air to travel from paranasal sinus into orbit and brain. However, it is extremely rare to have orbital emphysema and pneumocephalus in the absence of orbital wall fracture. In our case, a young male was admitted with severe eye pain, diminution of vision due to sudden exposure of compressed air gun. Examination revealed proptosed, emphysematous right eye with conjunctival laceration. Computed tomography scan of the head and orbit revealed multiple radiolucencies with air in the right orbit and brain extending up to the spinal canal without any evidence of orbital sinus or cranial bone fracture. Visual acuity recovered completely on follow–up; however, 2 weeks following injury, the patient developed disabling flashes of light which the patient perceived as central in location and resolved finally over a period of 3 months. Flashes of light, in our case, could be attributable to unnoticed damage to cerebral vasculature or connective tissue surrounding the optic nerve due to pneumocephalus.

  15. A rare case of cervical epidural extramedullary plasmacytoma presenting with monoparesis

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    Turk Okan

    2017-03-01

    Full Text Available Multiple myeloma and other plasma cell disorders are characterized by production of a large number of plasma cells in the bone marrow. On the other hand, plasmacytoma results from proliferation of abnormal plasma cells in the soft tissue or skeletal system. Neurological complications are frequently observed in these diseases. The most commonly known complications among those complications are spine fractures, spinal cord compressions, and peripheral neuropathies. Although neurological involvements are common in plasmacytomas, extramedullary spinal epidural localizations have been reported very rarely. In this case report, we aimed to present a plasmacytoma case that presented with acute onset of upper extremity monoparesis. A 40-year-old woman was admitted to our clinic with complaints of sudden weakness and numbness in her left arm following neck and left arm pain. Emergency cervical magnetic resonance imaging (MRI revealed an epidural mass and the patient underwent emergency surgery. The patient showed improvement post-operatively and the pathology was reported as plasmacytoma. Following hematology consultation, systemic chemotherapy was initiated and radiotherapy was planned after wound healing.

  16. Rare variant association analysis in case-parents studies by allowing for missing parental genotypes.

    Science.gov (United States)

    Li, Yumei; Xiang, Yang; Xu, Chao; Shen, Hui; Deng, Hongwen

    2018-01-15

    The development of next-generation sequencing technologies has facilitated the identification of rare variants. Family-based design is commonly used to effectively control for population admixture and substructure, which is more prominent for rare variants. Case-parents studies, as typical strategies in family-based design, are widely used in rare variant-disease association analysis. Current methods in case-parents studies are based on complete case-parents data; however, parental genotypes may be missing in case-parents trios, and removing these data may lead to a loss in statistical power. The present study focuses on testing for rare variant-disease association in case-parents study by allowing for missing parental genotypes. In this report, we extended the collapsing method for rare variant association analysis in case-parents studies to allow for missing parental genotypes, and investigated the performance of two methods by using the difference of genotypes between affected offspring and their corresponding "complements" in case-parent trios and TDT framework. Using simulations, we showed that, compared with the methods just only using complete case-parents data, the proposed strategy allowing for missing parental genotypes, or even adding unrelated affected individuals, can greatly improve the statistical power and meanwhile is not affected by population stratification. We conclude that adding case-parents data with missing parental genotypes to complete case-parents data set can greatly improve the power of our strategy for rare variant-disease association.

  17. A Rare Case of Immature Ovarian Teratoma with Gliomatosis Peritonei

    African Journals Online (AJOL)

    KEY WORDS: Glial tissue, gliomatosis peritonei, immature teratoma, India, ovary. Case Report .... astrocytes in the fetal nerve tissue. GFAP immunostain confirms ... Immature teratoma of the ovary: A clinicopathology study of 28 cases. Indian.

  18. A rare case of adult scalp pyoderma gangrenosum with cranial osteolysis

    OpenAIRE

    L. Aljohmani; K. Abdul-Jalil; C. deBlacam; G.M. Murphy; J.B. O'Sullivan

    2018-01-01

    Pyoderma gangrenosum (PG) is a rare and painful idiopathic skin condition that has one or more areas of chronic ulceration with well demarcated and undermined borders. Bone osteolysis (the pathological destruction of bone tissue) secondary to PG is a rare phenomenon with limited cases reported in children only. This is the first case report of scalp PG with cranial osteolysis in an 80-year-old adult, with an initial presentation mimicking skin carcinoma. This case highlights the importance...

  19. Inverted papilloma of the cervix and vagina: report of 2 cases of a rare lesion associated with human papillomavirus 42.

    Science.gov (United States)

    Hennell, Claire; Jamison, Jackie; Wells, Michael; McCluggage, W Glenn

    2012-03-01

    We report 2 cases of a lesion that we term inverted papilloma of the lower female genital tract, occurring in the cervix and upper vagina of 60- and 50-year-old women, respectively. Microscopically, the features were similar to those of inverted transitional papilloma of the urinary bladder with interconnecting islands, trabeculae, and solid sheets of bland transitional epithelium with an inverted growth pattern. There were small foci of squamous and glandular differentiation in the cervical case. Linear array human papillomavirus genotyping revealed human papillomavirus type 42 in both cases. Inverted papilloma in the lower female genital tract is extremely rare with, as far as we are aware, only 3 previously reported similar cases in the cervix and none in the vagina. Our results suggest that these neoplasms when occurring in the lower female genital tract may be associated with low-risk human papillomavirus, perhaps specifically human papillomavirus 42. Copyright © 2012 Elsevier Inc. All rights reserved.

  20. Defining Political Extremism in the Balkans. The Case of Serbia

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    Babić Marko

    2015-12-01

    Full Text Available Political extremism (and particularly right wing political extremism remains relatively insufficiently explored due to the fact that the phenomenon is controversial and hard to define. Its ambiguity and variability depending on time and spatial point of view further complicates its definition. Its structure is amorphous and eclectic as it often includes elements from different ideologies and connects incompatible ideas. A multidimensional conceptualization and an interdisciplinary approach - sociological, social, psychological and historical, are the Author’s tools in explaining the phenomenon of political extremism in Serbia, hopefully contributing to its clarification and laying a foundation for its further explanatory theoretical studies.

  1. PRIMARY GIANT HYDATID DISEASE OF THE SPLEEN: A RARE CASE REPORT WITH REVIEW OF LITERATURE

    Directory of Open Access Journals (Sweden)

    Subramanyam

    2015-02-01

    Full Text Available The most common organ involved in hydatid disease is the liver, followed by the lungs. Hydatid disease of spleen is a rare clinical condition, as even in the endemic region the frequency is reported to be 0.5 – 4% of abdominal hydatid diseases. Most commonly splenic involvement is secondary i.e., along with other organs. Primary hydatid diseases in s pleen is rare, here we are reporting a rare case of primary splenic hydatid disease

  2. Thanatophoric Dysplasia; a Rare Case Report on a Congenital Anomaly

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    Maria Francis Yuvaraj

    2017-01-01

    Full Text Available The rare form of skeletal dysplasia is thanatophoric dysplasia. The meaning for thanatophoric dysplasia is death bearing which is derived from Greek word. It occurs 1in 20,000 to 50,000. It is mainly due to mutations in the fibroblast growth factor receptor 3gene. Features of thanatophoric dysplasia are frontal bossing, prominent eyes, narrow thorax, protruded abdomen and bowed legs. The knowledge about this condition is useful in the fields of Anatomy, Paediatrics, Obstetrics and Gynaecology, Ultrasonagraphy and Genetics, for future research purpose.

  3. Urinary Ascites in Newborn – A Rare Case Report

    Directory of Open Access Journals (Sweden)

    Suryakant Y. Ingale

    2014-01-01

    Full Text Available Urinary Ascites in the newborn is a very rare condition. It is commonly secondary to posterior urethral valves (PUV which are membranous folds extending from proximal urethra leading to obstruction to urine flow. Here we report a two days old male neonate delivered full term in a hospital who presented with gross, uniform tense distension of abdomen with massive scrotal oedema, secondary to massive urinary ascites as a result of rupture of renal calyces. In addition there was a peri-renal urinoma.

  4. A rare case of congenital insensitivity to pain with anhydrosiss

    Directory of Open Access Journals (Sweden)

    Govardhani Yanamadala

    2015-01-01

    Full Text Available Congenital insensitivity to pain syndrome with anhydrosis (CIPA is a rare inherited disorder. It is characterized by loss of pain and temperature sensation, lack of sweating and mild mental retardation. This disorder belongs to hereditary sensory and autonomic neuropathy family (type IV. Because of these abnormalities, patients require special anesthetic care. They include titration of intraoperative opioids, an anesthetic to ensure co-operation and immobility and intra-operative temperature monitoring. Here we report a 7-year-old female child with CIPA posted for restoration and cementation for dental caries along with sural nerve and skin biopsy.

  5. A rare case of lymphangioleiomyomatosis with recurrent pneumothorax

    Directory of Open Access Journals (Sweden)

    Vinay Mahishale

    2015-01-01

    Full Text Available Lymphangioleiomyomatosis (LAM is a rare disease of unknown etiology that traditionally affects young women of childbearing or premenopausal age. It is characterized by proliferation of atypical smooth muscle cells, preferentially along bronchovascular structures that cause progressive respiratory failure. Owing to its unusual and nonspecific presenting symptoms, patients often receive missed or delayed diagnosis. This disease occurs sporadically or in association with the genetic disease-tuberous sclerosis complex. Recurrent pneumothorax is the hallmark of LAM. We present a 16-year-old young female having recurrent pneumothorax with LAM.

  6. A rare case of unilateral diffuse melanocytic proliferation

    Directory of Open Access Journals (Sweden)

    Guruprasad Ayachit

    2018-01-01

    Full Text Available A 67-year-old woman presented with metamorphopsia in the right eye. Leopard mottling was seen temporal to the fovea oculus dexter with corresponding hyper- and hypo-autofluorescent lesions on fundus autofluorescence. Spectral domain-optical coherence tomography revealed hyperreflective dots in the retinal pigment epithelium and choroid with subretinal fluid (SRF. Intravitreal bevacizumab was administered with which SRF resolved, albeit with increase in the areas of mottling. The patient was diagnosed to have metastatic ductal carcinoma of the right breast. It is important to bear in mind that the well-known entity of bilateral diffuse uveal melanocytic proliferation can rarely present unilaterally.

  7. BILATERAL UNICONDYLAR HOFFA FRACTURE : A RARE CASE REPORT

    Directory of Open Access Journals (Sweden)

    Pardha Saradhi

    2015-02-01

    Full Text Available NTRODUCTION: Hoffa f racture was first described by FRIEDRICH BUSCH, a surgeon from Berlin in 1869, and always supposed by ALBERT HOFFA in 1904. It is a rare injury consisting of tangential (CORONAL SHEAR fracture of distal femoral condyles. These fractures are due to high energy trauma and typically seen in a motor bike accident in a young patient subjected to shear force in both sagittal and coronal plane. (1 These fractures are not easy to visualise on routine imaging and therefore could represent a diagnostic challenge t o the accident department and orthopaedic surgeons

  8. A rare case of familial reactive perforating collagenosis

    Directory of Open Access Journals (Sweden)

    Rajesh Verma

    2013-01-01

    Full Text Available A 4 year old boy presented with history of itchy raised lesions on body of 2 years duration. Though parental consanguinity was not present, his elder brother had similar complaints. Dermatological examination revealed multiple hyperpigmented papules with a central keratotic plug distributed mainly over face and extensors of upper and lower extremities. Koebnerisation was present. Skin biopsy revealed perforating collagen bundles in the upper dermis and epidermis which was confirmed by Van Gieson staining. Patient was being treated with topical retinoids and intralesional corticosteroids with minimal relief.

  9. Intimal aortic sarcoma mimicking ruptured thoracoabdominal type IV aneurysm. a rare case report and review of the literature

    Directory of Open Access Journals (Sweden)

    Dedeilias Panagiotis

    2011-12-01

    Full Text Available Abstract Primary intimal aortic sarcoma represents a very rare and highly lethal medical entity. Diagnosis is made either by embolic events caused by the tumor or by surrounding tissue symptoms such as pain. Herein we report an extremely rare case of a 51-year-old man previously operated for ascending aortic aneurysm, who presented with clinical and radiological findings suggestive of a ruptured thoracoabdominal type IV aneurysm. The patient underwent radical resection of the aorta and surrounding tissue with placement of a composite 4-branched graft. The diagnosis was made by frozen section and regular histopathologic examination of the specimen and the patient received adjuvant chemotherapy. Nine months after surgery the patient is still alive and has no signs of recurrence. We review the literature and discuss the option of postoperative chemotherapy.

  10. Uncommon presentation of a rare tumour - incidental finding in an asymptomatic patient: case report and comprehensive review of the literature on intrapericardial solitary fibrous tumours

    OpenAIRE

    Czimbalmos, Csilla; Csecs, Ibolya; Polos, Miklos; Bartha, Elektra; Szucs, Nikolette; Toth, Attila; Maurovich-Horvat, Pal; Becker, David; Sapi, Zoltan; Szabolcs, Zoltan; Merkely, Bela; Vago, Hajnalka

    2017-01-01

    Background A solitary fibrous tumour is a rare, mainly benign spindle cell mesenchymal tumour most commonly originating from the pleura. An intrapericardial location of a solitary fibrous tumour is extremely unusual. We present a case of an asymptomatic patient with a slow-growing massive benign cardiac solitary fibrous tumour. Case presentation A 37-year-old asymptomatic female patient was referred to our hospital with an enlarged cardiac silhouette found on her screening chest X-ray. The ec...

  11. Pyogenic Arthritis of the Ankle Joint Following a High-Voltage Electrical Burn in the Lower Extremity: A Case Report

    Energy Technology Data Exchange (ETDEWEB)

    Kim, Kuk Seon; Lee, Gyung Kyu; Kang, Ik Won; Hwang, Dae Hyun; Lee, Eil Seong; Min, Seon Jung; Han, You Mie [Dept. of Radiology, Hangang Scared Heart Hospital, Hallym University College of Medicine, Seoul (Korea, Republic of); Lee, Eil Seong [Dept.of Radiology, Gyeongju Hospital, Dongguk University College of Medicine, Gyeongju (Korea, Republic of)

    2011-04-15

    A high-voltage electrical burn caused extensive deep muscle injuries beneath a relatively small skin wound at the contact point. Hidden, undetected deep muscle injuries have a tendency for progressive tissue necrosis, which can lead to major amputations or sepsis. The radiologic features of this rare, sometimes life-threatening injury have occasionally been described in the literature. However, to the best of our knowledge, there have been no reports on a case of pyogenic arthritis of the ankle joint following a high-voltage electrical burn involving the lower extremity. We report a case of the pyogenic arthritis of the ankle joint following a high-voltage electrical burn involving the lower extremity.

  12. Modeling annual extreme temperature using generalized extreme value distribution: A case study in Malaysia

    Science.gov (United States)

    Hasan, Husna; Salam, Norfatin; Kassim, Suraiya

    2013-04-01

    Extreme temperature of several stations in Malaysia is modeled by fitting the annual maximum to the Generalized Extreme Value (GEV) distribution. The Augmented Dickey Fuller (ADF) and Phillips Perron (PP) tests are used to detect stochastic trends among the stations. The Mann-Kendall (MK) test suggests a non-stationary model. Three models are considered for stations with trend and the Likelihood Ratio test is used to determine the best-fitting model. The results show that Subang and Bayan Lepas stations favour a model which is linear for the location parameters while Kota Kinabalu and Sibu stations are suitable with a model in the logarithm of the scale parameters. The return level is the level of events (maximum temperature) which is expected to be exceeded once, on average, in a given number of years, is obtained.

  13. Ellis–Van Creveld syndrome in siblings: A rare case report

    Directory of Open Access Journals (Sweden)

    Sabitha Gokulraj

    2016-01-01

    Full Text Available Ellis–Van Creveld syndrome or chondroectodermal dysplasia is a rare autosomal recessive disorder presenting several skeletal manifestations and congenital heart malformations. Ellis–Van Creveld syndrome comprises of a tetrad of clinical manifestations of chondrodysplasia, polydactyly, ectodermal dysplasia, and cardiac defects. Here, we are presenting a very rare case of Ellis–Van Creveld syndrome in siblings.

  14. A giant occipital encephalocele with spontaneous hemorrhage into the sac: A rare case report

    Science.gov (United States)

    Nath, H. D.; Mahapatra, A. K.; Borkar, S. A.

    2014-01-01

    In giant encephalocele, head size is smaller than the encelphalocele. Occipital encephalocele is the commonest of all encephalocele. In our case, there was rare association with giant encephalocele with old hemorrhage in the sac. This was a unique presentation. In world literature, there was rare association with giant encephalocele with hemorrhage. PMID:25685207

  15. A giant occipital encephalocele with spontaneous hemorrhage into the sac: A rare case report

    OpenAIRE

    Nath, H. D.; Mahapatra, A. K.; Borkar, S. A.

    2014-01-01

    In giant encephalocele, head size is smaller than the encelphalocele. Occipital encephalocele is the commonest of all encephalocele. In our case, there was rare association with giant encephalocele with old hemorrhage in the sac. This was a unique presentation. In world literature, there was rare association with giant encephalocele with hemorrhage.

  16. A Rare Case Report of Bilateral Complex Macrocystic Adrenal Hemorrhage Mimicking Fetal Neuroblastoma

    OpenAIRE

    Sindhwani, Geetika; Patel, Viral; Jain, Abhinav

    2018-01-01

    Fetal and neonatal adrenal glands are large vascular organs, which make them vulnerable to frequent bleeding. Although neonatal adrenal hemorrhage is commonly reported, it is rarely diagnosed on antenatal sonography. We present a rare case of prenatally diagnosed bilateral adrenal hemorrhage, which mimicked antenatal neuroblastoma.

  17. Intestinal Duplication Cyst presenting as Volvulus: A rare case report with Review of Literatures

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    Prasan Kumar Hota

    2015-03-01

    Full Text Available Intestinal duplication cyst is a rare congenital anomaly occurring any where along the alimentary tract, from mouth to anus. They can present with numerous complications like perforation, bleeding or intestinal obstruction. We report a rare case  of intestinal duplication cyst of ileum with acute intestinal obstruction due to volvulus with review of literature.

  18. Hidradenocarcinoma eccrinale syringomatodes of the eyelid--case of a rare malignancy.

    Science.gov (United States)

    Karolina, Ziaja; Arkadiusz, Pogrzebielski; Jolanta, Orłowska-Heitzman; Bozena, Romanowska-Dixon

    2010-01-01

    Presentation of a case of very rare malignant tumor of eccrine sweat glands in the eyelid. A 44 years old man with a tumor of the upper left eyelid is presented. Sweat gland carcinomas are rare malignant tumors of the skin adnexa. Diagnosis of these carcinomas is difficult due to their infrequency.

  19. A rare case of a familial form of nonsyndromic trigonocephaly ...

    African Journals Online (AJOL)

    The psychomotor development of patients is usually normal and the majority of cases are mild. Most cases are sporadic but familial forms with apparently autosomal dominant transmission have been reported (7-8%). However, the concordance rate of isolated trigonocephaly in monozygotic twins is 43%, suggesting that ...

  20. A rare case of Osteogenesis Imperfecta Type III

    Directory of Open Access Journals (Sweden)

    Nagaraj MV, Jehangir HM

    2014-03-01

    Full Text Available Osteogenesis imperfecta (OI the most common genetic cause of osteoporosis is a generalized disorder of connective tissue, characterized by increased bone fragility, low bone mass, recurrent fractures & numerous extra-osseous features with unusual presentations. We report a case of 7 year old female child presenting with respiratory distress with bowing of limb. This case is presented for its rarity.

  1. Esophagitis due to dexketoprofen trometamol: a rare case report.

    Science.gov (United States)

    Olmez, Sehmus; Donmez, Salim; Aslan, Mehmet; Karadas, Sevdegul; Yavuz, Alpaslan

    2015-05-01

    Various drugs are known to cause pill esophagitis. Antimicrobial drugs and nonsteroidal anti-inflammatory drugs are the most common causes of pill-induced esophagitis. Most patients suffer only self-limiting pain, but serious complications can occur. A 21-year-old man was admitted to our outpatient clinic with retrosternal chest pain, dysphagia, and odynophagia complaints, which occurred within 2 weeks after starting dexketoprofen trometamol. An upper endoscopy system examination revealed three well-demarcated ulcers in the esophagus at 35 cm from the incisors. Dexketoprofen trometamol may cause esophageal lesions. This rare disorder should be considered in patients presenting with sudden-onset retrosternal pain in addition to dysphagia and odynophagia.

  2. A Rare Cause of Hypothalamic Obesity, Rohhad Syndrome: 2 Cases.

    Science.gov (United States)

    Şiraz, Ülkü Gül; Okdemir, Deniz; Direk, Gül; Akın, Leyla; Hatipoğlu, Nihal; Kendırcı, Mustafa; Kurtoğlu, Selim

    2018-03-19

    Rapid-onset obesity with hypoventilation, hypothalamic dysfunction and autonomic dysregulation (ROHHAD) syndrome is a rare disease that is difficult to diagnosis and distinguish from genetic obesity syndromes. The underlying causes of the disease has not been fully explained. Hypothalamic dysfunction causes endocrine problems, respiratory dysfunction and autonomic alterations. There are around 80 reported patients due to lack of recognition. We present two female patient suspected of ROHHAD due to weight gain since early childhood. The presented symptoms, respiratory and circulatory dysfunction, hypothalamic hypernatremia, hypothalamo-pituitary hormonal disorders such as santral hypothyrodism, hyperprolactinemia and santral early puberty are completely matched the criteria of ROHHAD syndrome. ROHHAD syndrome should be considered in differential diagnosis since it is difficult to distinguish from causes of monogenic obesity. Early identification of the disease reduces morbidity of the syndrome and patients require regular follow-up by a multidisciplinary approach.

  3. Associated rare anomalies in prune belly syndrome: A case report

    Directory of Open Access Journals (Sweden)

    Andreas Fette

    2015-02-01

    Full Text Available The triad of deficient abdominal wall musculature, undescended testes and urinary tract anomalies characterizes the Prune Belly Syndrome (PBS. PBS can be associated with other comorbid urological and non urological conditions. But the full pathogenesis and best treatment is still a matter of debate. A term newborn with a classical PBS (Woodhouse Group 2, Smith and Woodard Group 2 plus lung hypoplasia and funnel chest deformity, a megapenis with a tight phimosis and an obturated anterior urethra is presented. Unfortunately, the baby died in urosepsis and renal failure in his 3rd week of life, despite urine drainage surgery and peritoneal dialysis undertaken. According to the best of our knowledge, this is an unique combination of rare anomalies in PBS patients.

  4. Pancreatoblastoma, a Rare Childhood Tumor: A Case Report

    Directory of Open Access Journals (Sweden)

    Asuman ARGON

    2017-05-01

    Full Text Available Pancreatoblastoma, rarely encountered in the literature, is a malignant exocrine tumor seen in the pancreas. A 5-year-old boy suffering from abdominal pain was sent to our institute for further examination and treatment. Clinical examination was normal but for a palpable abdominal tumor mass. Abdominal Doppler ultrasonography showed a mass with well-defined margins within the body of the pancreas. Laboratory tests, including lactic dehydrogenase, alpha-fetoprotein and cancer antigen 125 were abnormal. The tumor invading the splenic vein and transverse colon was removed totally. We observed a hypercellular tumor in histopathological examination. The tumor had epithelial acinar cells and squamoid morules (corpuscles separated by stromal bands. Adjuvant chemotherapy was used after surgery. However, the patient died 14 months later. All data about pancreatoblastoma have to be collected in order to choose the treatment to elucidate the molecular pathogenesis of the tumor, to diagnose it early and to develop target-specific treatments.

  5. Morganella morganii in sinonasal region: A rare case report

    Directory of Open Access Journals (Sweden)

    Haşmet Yazıcı

    2013-09-01

    Full Text Available Morganella morganii is a gram negative pathogen andmay cause potentially lethal disease especially in patientswith underlying or immunosuppressive disease. It is commonlyfound in long-term urinary catheter used and immunesystem deficiency patients as nosocomial disease.Involving other systems such as skin, skeletal systemand central nervous system can be seen too. Sporadicoccurrence is rare and can be seen in any system by variouscauses like AIDS, snake bites and poisoning. In thiscase we present sporadic Morganella morganii infectionon sinonasal region with the presence of sinusitis, sinocutaneousfistula, preseptal cellulitis and hard palate defecton 58 year old male diabetic patient. Microbiologicalassessment from open wound and sinuses were reportedas Morganella morganii. To our knowledge, this is the firstcase of sino-nasal Morganella morganii infection with sino-cutaneous fistula, preseptal cellulitis and maxillofacialbone destruction. J Clin Exp Invest 2013; 4 (3: 383-386Key words: Morganella Morganii, sino-nasal fistula, preseptalcellulitis, bone destruction

  6. Drug abuse, a rare cause of stroke: Case report

    Directory of Open Access Journals (Sweden)

    Zeynep Özözen Ayas

    2017-04-01

    Full Text Available At the present time the incidence of illicit drug use increases worldwide among young adults. Abuse of these substances is a rare cause of stroke in young adults. Cocaine, heroin, cannabis, and amphetamines use increase the risk of stroke. Cannabis sativa induce main effects by delta-9-hydrocannabinol. The main mechanism of marijuana-related stroke in young patients is vasospazm. The other possible mechanisms are systemic hypotension, impaired cerebral autoregulation, alteration of cerebral blood flow, cardioembolism due to atrial fibrillation. In this article a 25-year-old young male patient with paresia and paresthesia of right side who had chronic abuse of marijuana is reported. Clinicians must be alert about marijuana can be seriously harmful to cerebrovascular system in chronic use.

  7. Recurrent thymoma in the retroperitoneal space: a rare case report

    Directory of Open Access Journals (Sweden)

    Jun Yang

    2015-06-01

    Full Text Available Thymoma is an epithelial neoplasm of the thymus, which commonly lies in the anterior mediastinum and recurrences of thymoma generally are locally, and retroperitoneal recurrence is considered to be rare. A 46-year old Asian woman with invasive thymoma had undergone thymectomy 10 years ago. Computed tomography demonstrated a wellcircumscribed mass in the left retroperitoneal space. The patient had not any symptom including myasthenia gravis. Because on the anterior mediastinum area shows no sign of tumor recurrence and the mass adjacent to the vertebral body, neurogenic tumor was suspected. Surgical resection was performed using a retroperitoneal approach, which revealed the tumor adhering neighboring diaphragm. The tumor was histologically diagnosed to be type B1 thymoma according to the World Health Organization classification. The retroperitoneal mass was an unusual local recurrence after thymectomy. The patients whose had under invasive thymectomy should be evaluated carefully when finding retroperitoneal mass during follow-up.

  8. Cystic Echinococcosis: A Rare Case of Brain Localization

    Directory of Open Access Journals (Sweden)

    Ali BARADAN BAGHERI

    2017-02-01

    Full Text Available Although Hydatid disease eradicated in many countries, it is still widespread in communities where agriculture is dominant. Cystic hydatidosis is significant public health problem in the regions with endemic echinococcosis. The hydatid cysts tend to form mostly in the liver or lung. Brain involvement is very rare. In the present report, we describe magnetic resonance imaging findings in an 18-yr-old male with cerebral echinococcosis, in Shahid Madani Hospital, Karaj, Iran in 2015. The patient, presented with headache, hemiparesis, impairment of speech, vomiting, and nausea. Computed tomography, magnetic resonance imaging, and surgical exploration proved a cyst in the superior portion of left temporal lobe. Pathological examination showed it to be a solitary primary cerebral hydatid cyst. 

  9. Rare case of malignant craniopharyngioma reactive to adjunctive stereotactic radiotherapy and chemotherapy; Case report and review.

    Science.gov (United States)

    Nomura, Shunsunke; Aihara, Yasuo; Amano, Kosaku; Eguchi, Seiichiro; Chiba, Kentaro; Komori, Takashi; Kawamata, Takakazu

    2018-06-19

    Malignant craniopharyngioma or anaplastic craniopharyngioma was first reported in 1987 by Akachi. It has a malignant clinical and histological feature; remarkably rapid progression, atypical pathology like squamous cell carcinoma and poor prognosis. To date seventeen cases of malignant craniopharyngioma have been reported and of these cases, most were of secondary malignant tumor in nature. With respect to traditional benign craniopharyngioma, adjunctive treatment after gross total removal is not necessary, but in the case of malignant types of the tumor, adjunctive treatment is important. This paper presents the first case of malignant craniopharyngioma reactive to adjunctive Gamma knife stereotactic radiosurgery and chemotherapy. Malignant craniopharyngioma is very rare, and we report Gamma knife stereotactic radiosurgery and chemotherapy (Carboplatine and etoposide chemotherapy), as well as Temozolomide chemotherapy were effective and could control progression of the tumor temporarily. Since adjunctive Gamma knife stereotactic radiosurgery and chemotherapy of malignant craniopharyngioma cases affects follow-up strategies, we propose supporting the need to a revision to the WHO classification regarding malignancy evaluation of craniopharyngioma. Copyright © 2018 Elsevier Inc. All rights reserved.

  10. A rare case of massive hepatosplenomegaly due to acute ...

    African Journals Online (AJOL)

    massive hepatosplenomegaly include chronic lymphoproliferative malignancies, infections (malaria, leishmaniasis) and glycogen storage diseases (Gaucher's disease).[4] In our case the probable causes of the massive hepatosplenomegaly were a combination of late presentation after symptom onset, leukaemic infiltration.

  11. A Rare Case of Chondromyxoid Fibroma Mimicking Spina Ventosa

    African Journals Online (AJOL)

    2012-10-10

    Journal of Surgical Technique and Case Report | Jan-Jun 2012 | Vol-4 | Issue-1. 43 ... on Wednesday, October 10, 2012, IP: 41.185.99.174] || Click here to download free Android application for this journal .... the metacarpophalangeal joint.

  12. Candidiasis, A Rare Cause of Gastric Perforation: A Case Report ...

    African Journals Online (AJOL)

    Most cases of gastric perforation occur as complications of peptic ulcer ... movement on respiration, marked generalized abdominal ... inflammatory cell infiltration consisting of mainly eosinophils, macrophages, plasma cells, and lymphocytes. No Helicobacter pylori like organisms or atypical cellular proliferations were seen.

  13. Rare Locations of Epidermoid Cyst: Case Reports and Review ...

    African Journals Online (AJOL)

    Hence, we aim at highlighting the differential diagnosis pertaining to the anatomical location. ... Another case in our report was localized in the upper lip. ... Epidermoid cyst, Dermoid cyst , Lower one third of face, Mandibular body, Upper lip.

  14. Enterovesical fistula, a rare complication of Meckel’s diverticulum: A case report

    Directory of Open Access Journals (Sweden)

    Bourguiba M.A.

    2017-01-01

    Conclusion: Vesico-diverticular fistula resulting from a perforated Meckel's diverticulum is a rare complication. To our knowledge, this is only the fourth reported case which is not associated to inflammatory bowel disease.

  15. A rare case of unifocal, unilateral pigmented paravenous retinochoroidal atrophy (PPRCA

    Directory of Open Access Journals (Sweden)

    Meleha Ahmad

    2016-12-01

    Conclusions and importance: PPRCA is typically bilateral and symmetric, affecting primarily the outer retina and choroid. However, in rare cases, this disease can present unilaterally and/or unifocally, with degeneration extending to the inner retinal layers.

  16. Report of a Rare Case of an Odontogenic Myxoma of the Maxilla ...

    African Journals Online (AJOL)

    tissue that replaces the cancellous bone and expands the cortex. OMs are locally ... homogenous hypodense lesion of the right side maxilla. Report of a Rare Case of an .... Clinical and radiographic differential diagnosis of OMs may include: ...

  17. Prenatal sonographic diagnosis of limb-body wall complex: case series of a rare congenital anomaly

    Directory of Open Access Journals (Sweden)

    Arshad Bhat, MBBS, DMRD

    2016-06-01

    Full Text Available Three case reports of a rare congenital anomaly “limb-body wall complex” also known as “body stalk syndrome” are presented with prenatal ultrasonographic diagnostic features, immediate after delivery evaluation, and histopathologic analysis.

  18. Rare case of primary spinal ependymomatosis occurring in a 26-year-old man: a case report.

    LENUS (Irish Health Repository)

    Kaliaperumal, Chandrasekaran

    2009-01-01

    The authors report a rare case of primary spinal ependymomatosis in a young adult man. Multiple primary ependymomatous lesions were seen on magnetic resonance imaging and no anaplasia was identified on the surgical-pathological analysis. The aetio-pathological mechanism and surgical significance of this rare occurrence is discussed.

  19. [Branchiogen cyst at unusual age and in rare localization. A case report].

    Science.gov (United States)

    Horvath, Dóra; Redl, Pál; Hegedűs, Csaba

    2015-12-01

    Branchiogen anomalies represent a heterogeneous group of developmental abnormalities, they arise from incomplete obliteration of branchial clefts and pouches during embriogenesis. Clinically they can present as a cyst, fistula or sinus. Second cleft lesions account for 95% of the branchial anomalies. Second branchial cleft cysts are usually located in the neck, along the anterior border of the stenocleidomastoid muscle, but they can be anywhere along the course of the second branchial fistula from the tonsillar fossa to the supraclavicular region. Their presence in the nasopharynx is extremely rare. Ultrasound, computed tomography (CT) or magnetic resonance imaging is recommended for diagnosis. Definitive treatment is surgical excision, these lesions do not regress spontaneously and often result recurrent infections. A 7 month old infant applied to a pediatrician with gastrointestinal viral infection. During examination a cystic mass was discovered in the right lateral nasopharyngeal wall, the lesion extended to the oropharynx. Marsupialisation was performed via transoral approach. In case of cystic lesion in the lateral epipharynx, branchial cleft cyst should be considered in the differential diagnosis.

  20. Adenoid cystic carcinoma of the nasal septum: A rare case report

    Directory of Open Access Journals (Sweden)

    Basavaraj P Belaldavar

    2013-01-01

    Full Text Available A 60-year-old male patient came to ENT OPD with complaints of left nasal obstruction from the last 5 years and moderate quantity of epistaxis from the last 4 months. It was associated with foul smelling mucopurulent rhinorrhea. On clinical examination, a fleshy mass was seen occupying the posterior part of left nasal cavity and displacing the septum on the right side. The mass was relatively painful, soft, and bleeding on touch. The provisional diagnosis of "vascular-tumor-like" angiofibroma was suspected. Diagnostic nasal endoscopy and CT scan PNS were done which revealed a mass occupying the left nasal cavity arising from the posterior part of septum along the choanae till the anterior part of sphenoid sinus. Biopsy of the same revealed an adenoid cystic carcinoma. Adenoid cystic carcinoma is uncommon and that too of the nasal cavity. The cases of the adenoid cystic carcinoma involving the nasal cavity usually involves the lateral wall and the involvement of the posterior part of nasal septum is extremely rare. Thus the presentation of this uncommon disease is discussed here.

  1. Hepaticocystic duct and a rare extra-hepatic "cruciate" arterial anastomosis: a case report

    Directory of Open Access Journals (Sweden)

    Abeysuriya Vasitha

    2008-02-01

    Full Text Available Abstract Introduction The variations in the morphological characteristics of the extra-hepatic biliary system are interesting. Case presentation During the dissection of cadavers to study the morphological characteristics of the extra-hepatic biliary system, a 46-year-old male cadaver was found to have drainage of the common hepatic duct drains directly into the gall bladder neck. The right and left hepatic ducts were not seen extra-hepatically. Further drainage of the bile away from the gallbladder and into the duodenum was provided by the cystic duct. Formation of the common bile duct by the union of the common hepatic duct and cystic duct was absent. Further more the right hepatic artery was found to be communicating with the left hepatic artery by a "bridging artery" after giving rise to the cystic artery. An accessory hepatic artery originated from the "bridging artery" forming a "cruciate" hepatic arterial anastomosis. Conclusion Combination of a Hepaticocystic duct and an aberrant variation in the extra-hepatic arterial system is extremely rare.

  2. Clinical and Pathological Findings on Intoxication by Yellow Phosphorus After Ingesting Firework Cracker: A Rare Case of Autopsy.

    Science.gov (United States)

    Türkmen Şamdanci, Emine; Çakir, Ebru; Şahin, Nurhan; Elmali, Candan; Sayin, Sadegül

    2016-01-01

    Yellow phosphorus is a toxic substance used in the production of firework cracker, fireworks, ammunition and agricultural dung. When ingested, it shows its effects mainly in the liver, the kidneys, and the brain. A four-year-old girl had died as a result of acute hepatic failure caused by ingesting a firework cracker. The case showed high levels of hepatic enzymes, along with non-specific signs such as nausea, vomiting and diarrhea. Autopsy revealed diffuse microvesicular steatosis in the liver and disseminated degeneration in the proximal tubules of the kidneys. In cases with concomitant hepatorenal failure and cardiovascular collapse, death is inevitable. However, when only hepatic failure develops, hepatic transplantation may be lifesaving. Although intoxication from ingesting yellow phosphorus has a very high rate of mortality, forensic cases are extremely rare in the literature.

  3. Mesenchymal Chondrosarcoma of Maxilla: A Rare Case Report and Review of Literature

    Directory of Open Access Journals (Sweden)

    R Thriveni

    2010-01-01

    Full Text Available Mesenchymal condrosarcomas are rare malignant neoplasms that can arise from both soft and hard tissues. They are distinct tumors arising in unicentric or multicentric locations. They reveal unusual clinical behavior, characteristic histopathological features, and poor prognosis with late recurrences. Here is a case report of a rare case of mesenchymal chondrosarcoma arising in a 19-year-old female patient′s right maxilla.

  4. Sirenomelia and ndash; Mermaid Syndrome with Oesophageal Atresia: A Rare Case Report

    Directory of Open Access Journals (Sweden)

    Veena Raja

    2015-09-01

    Full Text Available Sirenomelia or Mermaid syndrome is a very rare congenital deformity in which legs are fused together and commonly associated with abnormal kidney development, genital, and rectal abnormalities. In this present case, sirenomelia was associated with oesophageal atresia, which is a rare association and occurs in about 20-35% of cases. [J Interdiscipl Histopathol 2015; 3(3.000: 113-116

  5. Management of A Rare Case of Communicating Internal-External Inflammatory Resorption.

    Science.gov (United States)

    Arora, Suraj; Gill, Gurdeep Singh; Saluja, Priyanka; Setia, Vikas

    2015-05-01

    The present case describes the successful management of a rare case of communicating internal-external resorption in which both internal and external resorption seem to develop independent of each other. The case report highlights the importance of correct diagnosis and need of revision of classification system of resorptive defects.

  6. Zosteriform Lichen Planus: case report of a rare variant of Lichen Planus

    OpenAIRE

    Kanthilatha Pai; Sathish Pai

    2013-01-01

    Since its original description by Devergie in 1854, several cases of linear lichen planus have been described in the literature, but there have been notably few cases of the more rare zosteriform lichen planus. Zosteriform lichen planus needs to be differentiated from linear lichen planus and other linear dermatoses. We present a case of Zosteriform Lichen planus for its rarity and briefly review literature.

  7. Rare anatomical variation of the musculocutaneous nerve - case report

    Directory of Open Access Journals (Sweden)

    Sergio Ricardo Rios Nascimento

    2016-06-01

    Full Text Available ABSTRACT The clinical and surgical importance of anatomical knowledge of the musculocutaneous nerve and its variations is due to the fact that one of the complications in many upper-limb surgical procedures involves injury to this nerve. During routine dissection of the right upper limb of a male cadaver, we observed an anatomical variation of this nerve. The musculocutaneous nerve originated in the lateral cord and continued laterally, passing under the coracobrachialis muscle and then continuing until its first branch to the biceps brachialis muscle. Just after this, it supplied another two branches, i.e. the lateral cutaneous nerve of the forearm and a branch to the brachialis muscle, and then it joined the median nerve. The median nerve followed the arm medially to the region of the cubital fossa and then gave rise to the anterior intermediate nerve of the forearm. The union between the musculocutaneous nerve and the median nerve occurred approximately at the midpoint of the arm and the median nerve. Given that either our example is not covered by the classifications found in the literature or that it fits into more than one variation proposed, without us finding something truly similar, we consider this variation to be rare.

  8. A rare case of vestibular sinus tract: A periodontist enigma

    Directory of Open Access Journals (Sweden)

    Disha Nagpal

    2016-01-01

    Full Text Available Sinus tract occurs commonly in teeth with periapical/periodontal infection but the formation of a vestibular opening, causing esthetic compromise and food lodgement, is uncommon. Definitive treatment of a chronic sinus tract requires treatment of the original problem, that is, the necrotic pulp treated by endodontic therapy or by extraction of the tooth. However, at times endodontic therapy may not be adequate necessitating periodontal intervention. The present case had vestibular opening communicating with root canal of concerned tooth appearing ten years after trauma. The diagnosis of such cases can only be made after careful evaluation and the treatment plan has to be modified from the conventional. The success of such cases depends on the regular follow up as presented here and careful observation after each phase of treatment.

  9. A very rare case of a small bowel leiomyosarcoma leading to ileocaecal intussusception treated with a laparoscopic resection: a case report and a literature review.

    Science.gov (United States)

    Guzel, Tomasz; Mech, Katarzyna; Mazurkiewicz, Michał; Dąbrowski, Bohdan; Lech, Gustaw; Chaber, Andrzej; Słodkowski, Maciej

    2016-02-24

    Small bowel tumours are rare and comprise less than 2% of all primary gastrointestinal neoplasms. Among these tumours, a leiomyosarcoma belonging to soft tissue sarcomas is extremely rare and accounts for about 1 % of malignant mesenchymal lesions in the gastrointestinal tract. Due to its aggressive nature and slow growth, it is often diagnosed at the late stage when curative treatment is impossible. Authors report a first case of leiomyosarcoma with chronic recurrent ileocaecal intussusception and literature review to analyse diagnosis and treatment features of the ileum mesenchymal tumours. We present a case of an 87-year-old Caucasian man suffering from cramp-like abdominal pain for months. Due to lack of clinical signs and unspecific complaints, a diagnosis was delayed. Despite a detailed in-hospital examination, a proper diagnosis was established as late as during an operation. The patient was treated by surgery with good results. An uncommon laparoscopic resection of the small bowel with a tumour was performed. A histopathological investigation confirmed a very rare mesenchymal lesion of the distal ileum. The patient is under control with no recurrence for 1 year of the follow-up period. Reported case indicates that a usually asymptomatic tumour can cause uncommon chronic recurrent ileus signs. CT and MRI scans are investigation of choice in such cases, but they are sometimes inconclusive. It might be worth highlighting the good results of laparoscopic leiomyosarcoma lesion resection with a very good outcome.

  10. A rare case of acute poster ior reversible encephalopathy syndrome involving brainstem in a child

    Directory of Open Access Journals (Sweden)

    Olfa Chakroun-Walha

    2016-11-01

    Full Text Available Posterior reversible encephalopathy syndrome (PRES is a rare entity involving brainstem in very rare reported cases. We describe here the case of a boy who presented to the emergency department for headaches and strabismus. Diagnosis of PRES was retained by magnetic resonance imaging. The causes were blood pressure urgency and renal failure. Location of lesions was very rarely reported in literature and neurological troubles were persistent. Emergency physicians should evocate PRES each time there is a clinical context associated with neurological troubles by a normal brain CT scan. Early diagnosis is very important to treat its causes and improve prognosis.

  11. Linear verrucous hemangioma-a rare case and dermoscopic clues to diagnosis.

    Science.gov (United States)

    Dhanta, Aditi; Chauhan, Payal; Meena, Dilip; Hazarika, Neirita

    2018-01-01

    Verrucous hemangioma (VH) is a rare, congenital and localized vascular malformation, which usually presents as warty, bluish, vascular papules, plaques, or nodules, mainly on the lower extremities. Linear presentation of the disease is rare. A deep biopsy is necessary to confirm the clinical diagnosis by histopathological examination, with dermoscopy acting as a useful tool for evaluating the precise vascular structure. Here, we report on a 13-year-old female child with linear VH presenting over her foot since infancy and dermoscopic findings of VH along with the clinical-pathologic features.

  12. Merkel cell carcinoma with axillary metastasis; a case report of a rare disease

    Directory of Open Access Journals (Sweden)

    Serdar Culcu

    2018-04-01

    Full Text Available Merkel cell carcinoma is a rare primer neuroendocrine carcinoma of the skin. It is an extremely aggressive tumor. This rare carcinoma is seen with high local and regional recurrence ratios and distant metastasis. We report that a 64 years old female patient who had undergo an excision in another center because of a mass on 4 cm proximal of her right elbow had been diagnosed with Merkel cell carcinoma with positive surgical margins. She was treated with wide re-excision and axillary dissection at our clinic. Keywords: Merkel cell carcinoma, Skin, Axillary metastasis

  13. Effects of an extreme flood on river morphology (case study

    NARCIS (Netherlands)

    Yousefi, Saleh; Mirzaee, Somayeh; Keesstra, Saskia; Surian, Nicola; Pourghasemi, Hamid Reza; Zakizadeh, Hamid Reza; Tabibian, Sahar

    2018-01-01

    An extreme flood occurred on 14 April 2016 in the Karoon River, Iran. The occurred flood discharge was the highest discharge recorded over the last 60 years in the Karoon River. Using the OLI Landsat images taken on 8 April 2016 (before the flood) and 24 April 2016 (after the flood) the geomorphic

  14. Priapism Followed by Discontinuation of Methadone: A rare Case Report

    Directory of Open Access Journals (Sweden)

    Seyed-Ali Mostafavi

    2015-10-01

    Full Text Available Objective: Priapism is defined by persistent, painful penile erection which occurs without sexual stimulation. Methadone is used as an analgesic and is also used in detoxification and maintenance protocol for opioid dependence treatment. Here we will report a case of a male with priapism after rapid discontinuation doses of methadone.Case presentation: The case was a young married male who referred to a psychiatry clinic due to long-time spontaneous erections. The patient had no history of mental disorders, trauma or sickle cell anemia. He used to smoke opium for five years and used methadone for four years at a dose of 17 cc daily, which he abruptly discontinued. Then he often experienced spontaneous and painful erections without physical or mental stimulation that caused him shame and embarrassment .Conclusion: In this case, chronology indicates that rapid discontinuation of methadone was possibly responsible for the occurrence of priapism. This may have happened due to a compensatory reaction to methadone side effect of erectile dysfunction, followed by its rapid withdrawal.

  15. Addison’s Disease: A rare case report

    Directory of Open Access Journals (Sweden)

    Sanjay N. Agrawal

    2015-04-01

    Full Text Available A female patient presented with progressive weakness, asthenia and generalized hyperpigmentation. The characteristic hyperpimentation pointed towards possibility of Addison’s disease which was proved by markedly decreased plasma cortisol levels, hyponatremia and hyperkalemia. This could be one of the very few cases of Addison’s Disease reported.

  16. Diabetes insipidus - a rare complication of major flame burn: case ...

    African Journals Online (AJOL)

    Background: Diabetes insipidus is a syndrome characterised by polyuria which is associated with urinary frequency, enuresis, nocturia and polydipsia. We present this case to highlight the significant role of high degree of clinical suspicion and interdisciplinary management resulting in a rewarding outcome in a limited ...

  17. RARE CASE OF DESMOID TUMOR OF URINARY BLADDER

    Directory of Open Access Journals (Sweden)

    Chalapathy

    2015-08-01

    Full Text Available Desmoid tumor is a benign soft tissue tumor which belongs to a family of myofibroblastic fibromatoses. Occasionally, desmoid tumors have an unusual site of occurrence . We describe a case of incisional hernia in postmenopausal women with an intra operative incidental finding of a desmoid tumor from anterior wall of urinary bladder for which a wide excision was performed

  18. [Fournier's gangrene: Cervical and facial extension. A very rare case].

    Science.gov (United States)

    Carloni, R; Pechevy, L; Isola, N; Vidal, L; Goga, D; Watier, E; Bertheuil, N

    2016-02-01

    Fournier's gangrene is a fearsome disease with a bad prognosis and a mortality rate ranging between 10 and 80% according to the literature. It is extensive in 13 to 54% of cases. Up to date, cervico-facial extension has never been reported. We describe the case of a 51-year-old overweighed woman with a history of type 2 diabetes and a narrow lumbar canal who was referred to our institution for significant fatigue and increasingly painful legs. A diagnosis of Fournier's gangrene was made after correlating the physical findings with the results of a full body scan. Diffuse subcutaneous emphysema involving the face, neck, mediastinum, abdominal wall, right buttock, perineum and the right thigh was identified. Treatment included multiple surgical debridements, admission to intensive care unit, and an efficient antibiotic therapy that enabled preservation of the patient's life. To our knowledge, this is the first case of cervical and mediastinal extension of Fournier's gangrene to be reported. No clear guidelines exit on the management of this complication (cervico-facial and mediastinal drainage). We share our experience of this unusual case. Copyright © 2015 Elsevier Masson SAS. All rights reserved.

  19. Erupting complex odontoma: Report of a rare case

    Directory of Open Access Journals (Sweden)

    Pinakapani Ramakrishna

    2014-01-01

    Full Text Available Odontomas are the most frequent hamartomatous lesions involving the oral cavity. The complex variant is an agglomerate of all dental tissues characterized by abnormal morphodifferentiation despite normal histodifferentiation. These are usually asymptomatic and are frequently associated with eruption disturbances. We report an unusual case of erupting complex odontoma associated with an impacted maxillary second molar.

  20. Bilateral Paratesticular Liposarcoma ‑ A Rare Case Report

    African Journals Online (AJOL)

    Radiotherapy or chemotherapy is added for advanced disease or recurrences. CASE REPORT. A 65‑year‑old ... Intraoperative, a firm, well‑defined tumor, about 20 × 14 × 5 cms on the left and 16 × 14 × 4 ... patient was counseled on plans for further chemotherapy and radiotherapy. He, however, refused further treatment.

  1. A rare case report of 46XY mixed gonadal dysgenesis

    Directory of Open Access Journals (Sweden)

    Rakesh Arora

    2013-01-01

    Full Text Available A 16-year-old person, reared as female presented with complaints of genital ambiguity and primary amenorrhoea along with lack of secondary sexual characters, but without short stature and Turner′s stigmata. She was taking steroids after being misdiagnosed as congenital adrenal hyperplasia (CAH. Karyotype analysis revealed 46XY karyotype. There was no evidence of hypocortisolemia (cortisol 9.08 μg/dl, adrenocorticotropic hormone [ACTH] 82.5 pg/ml or elevated level of 17-OH-progesterone (0.16 ng/ml. Pooled luteinizing hormone (LH was 11.79 mIU/ml and follicle-stimulating hormone (FSH was 66.37 mIU/ml. Serum estradiol level was 25 pg/ml (21-251. Basal and 72 h post beta-human chorionic gonadotropin (hCG levels of androstenedione and testosterone levels were done (basal testosterone of 652 ng/dl and basal androstenedione of 1.17 ng/ml; 72 h post hCG testosterone of 896 ng/dl and androstenedione of 1.34 ng/ml. Magnetic resonance imaging (MRI pelvis (with ultrasonogrphy [USG] correlation revealed uterus didelphys with obstructed right moiety and bilateral ovarian-like structures. Right sided gonads and adjacent tubal structures were visualized laparoscopically and removed. Left sided gonads were not visualized and Mullerian remnants were adhered to sigmoid colon. Histopathological examination revealed presence of testicular tissue showing atrophic seminiferous tubules with hyperplasia of Leydig cells. No ovarian tissue was seen. Based on these results a diagnosis of 46XY mixed gonadal dysgenesis (MGD was made, which is rare and is difficult to distinguish from 46XY ovotesticular disorder of sexual differentiation (OT-DSD. The patient was managed with a multidisciplinary approach and fertility issues discussed with the patient′s caregivers.

  2. Preejaculatory illness syndrome: Two cases of a rare psychosomatic disorder

    Directory of Open Access Journals (Sweden)

    Adel Mohsen

    2016-01-01

    Full Text Available Human ejaculation happens in the orgasmic phase of the human sexual response cycle. Among psychosomatic ejaculatory disorders that may happen before ejaculation, we present two cases of preejaculatory illness syndrome. The two cases shared common symptoms of sympathetic over activity, the sensation of impending death, and muscle atonia with subsequent failure to ejaculate. Depression, anxiety disorders, and family histories of psychiatric problems were noticed as risk factors. Medical conditions that may lead to panic attack type symptoms were eliminated before the final diagnosis. After the failure of empirical medications, symptoms became controlled with fluoxetine. Patients reported a recurrence of the symptoms on trying to stop the prescribed medication. On the last follow-up, they still take fluoxetine on a regular base with satisfactory sexual life.

  3. Accidental intraoral formalin injection: a rare case report

    Directory of Open Access Journals (Sweden)

    Ramakant Dandriyal

    2014-12-01

    Full Text Available Formalin is a hazardous chemical that needs cautious handling and special storage. Owing to its disinfectant and fixative (i.e. for preserving pathologic tissue specimens in histopathology properties, it is widely used in dentistry. Although, the terms formaldehyde and formalin are often confused as being identical, these are different as to the concentrations of the primary component i.e. formaldehyde. In fact, the common fixative available as 10% neutral buffered formalin is actually a 4% solution of formaldehyde (i.e., a 10% solution made from a 37-40% commercially pure formaldehyde solution. This case report describes an unfortunate case of accidental injection instead of local anesthetic, of formalin into the pterygomandibular space in a 35-year old woman during inferior alveolar nerve block for surgical removal of impacted lower right third molar and its successful management by cautious debridement (under both local and general anesthesia and empirical drug therapy (utilizing analgesics and antibiotics.

  4. Infective Endocarditis with Uveitis: A Rare Case Report

    Directory of Open Access Journals (Sweden)

    Hsiang-Chun Lee

    2007-01-01

    Full Text Available We report a case of a 51-year-old diabetic male who presented with a complaint of intermittent chills and fever that he had experienced for 10 days. No obvious respiratory tract, genitourinary tract, gastrointestinal tract, or skin lesions were observed. Blood culture data were positive for group B β-streptococcus. Transthoracic and transesophageal echocardiography revealed vegetation in the anterior leaflet of the mitral valve. The patient was diagnosed with infective endocarditis (IE and prescribed a parenteral antibiotic. Three days after admission, the patient complained of progressively blurred vision. Slit lamp examination found fine keratic precipitates and aqueous cells in the anterior chambers in both eyes, implying that the patient had uveitis. He was then prescribed a topical steroid for 4 months, and his vision improved gradually. This case is an important reminder that uveitis, not only endophthalmitis, can occur with IE. Treatment for one condition, if misapplied, may worsen the other.

  5. Rare Case of Non Tuberculous Mycobacterial: A Diagnostic dilemma

    LENUS (Irish Health Repository)

    Marathe, N

    2017-02-01

    Non-Tuberculous Mycobacterial (NTM) infections occur in HIV-negative patients with or without underlying lung disease. It is generally felt that these organisms are acquired from the environment. Unlike tuberculosis, there are no convincing data demonstrating human-to-human OR animal-to-human transmission of NTM. We report a case of NTM infection in a 38 year old patient with underlying emphysematous lung disease. The case highlights the diagnostic dilemma which occurs when persistent sputum Acid- Fast Bacilli (AFB) smears are positive, but Nucleic acid amplification test is negative. To aid the diagnosis and rule out Pulmonary Tuberculosis as the other differential diagnosis, we applied American Thoracic Society\\/Infectious Disease Society of America (ATS\\/IDSA) guidelines & recommendations1. The decision to treat was taken on basis of CT findings, clinical, microbiologic criteria and expert consultation with Microbiology department at Waterford.

  6. Metachronous Testicular Cancer After Orchiectomy: A Rare Case.

    Science.gov (United States)

    Arda, Ersan; Cakiroglu, Basri; Cetin, Gizem; Yuksel, Ilkan

    2017-11-09

    Testicular cancer represents approximately 1% of all cancers diagnosed in males. The prevalence of bilateral testicular germ cell tumor cases varies from 1% to 5%. Intratubular germ cell neoplasia (ITGCN) is a precursor for almost all testicular germ cell tumors (TGCT) and is one of the highest risks of developing contralateral testicular cancer. The radical orchiectomy is still preferred for the treatment of testicular cancer. However, in some cases like solitary testis, bilateral cancer or if the tumor size is under 30% percent of the testicular extent, organ-sparing surgery can be an option. There are just a few published reports of metachronous contralateral testicular cancer, developed after orchiectomy with the histopathology of the intratubular germ cell neoplasia.

  7. Recurrent ovary cancer presenting with scleroderma - A rare case report

    OpenAIRE

    Sargin, Betul; Gurer, Gulcan; Bozbas, Gulnur; Noyan, Fatih; Barut, Kayra; Tataroglu, Canten

    2017-01-01

    Scleroderma is a chronic autoimmune multisystem disorder which is characterizedby progressive fibrosis of the skin and internal organs. Ovary cancers with sclerodermahave been reported in the literature. But recurrent ovary cancer with sclerodermahas not been reported before. Here, we report a 65 -year old female patient presentingwith recurrent ovary cancer and subsequently diagnosed with scleroderma. Due toliterature sources, this is the first case of presenting with recurrent ovary cancera...

  8. Oral manifestations of lamellar ichthyosis: A rare case report

    Directory of Open Access Journals (Sweden)

    Keerthi K Nair

    2016-01-01

    Full Text Available The ichthyoses are a heterogeneous group of disorders with both inherited and acquired forms. Autosomal recessive congenital ichthyosis (ARCI is a heterogeneous group of disorders that present at birth with the generalized involvement of skin without other systemic manifestations. Lamellar itchthyosis (LI is a nonsyndromic itchthyosis, which comes under the umbrella of ARCI. Little is only known about the oral manifestations of this disorder. We report a case of LI with oral manifestations.

  9. Spontaneous Bilateral Tubal Gestation: A Rare Case Report

    Directory of Open Access Journals (Sweden)

    Marwah Sheeba

    2016-01-01

    Full Text Available Here a case is presented where the woman after a positive pregnancy test underwent medical abortion for unwanted pregnancy without ultrasound confirmation of intrauterine pregnancy. On persistence of symptoms, a second opinion was procured, when examination and a transvaginal ultrasound scan revealed ruptured unilateral tubal ectopic pregnancy. However upon subsequent laparotomy (considering deteriorating hemodynamic status of the patient, intraoperatively it turned out to be a bilateral tubal ectopic gestation.

  10. ONE OF THE LONGEST APPENDIX: A RARE CASE REPORT

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    Venkat Rao

    2015-03-01

    Full Text Available The vermiform appendix is an organ that can have variable sizes. We are prompted to report here one of the longest appendix removed, measuring about 16cm in length. INTRODUCTION : The vermiform appendix is an organ that can vary in size, site, and presence, as well as in other clinical and functional aspects. We describe here one of the longest appendix removed, measuring about 16cm in length in a case of acute appendicitis

  11. Gingival Fibromatosis with Chronic Periodontitis – A Rare Case Report

    OpenAIRE

    Sanadi Rizwan M

    2009-01-01

    Gingival fibromatosis is frequently an isolatedcondition of little consequence apart from a cosmeticproblem and occasional associations with uncommonsyndromes. This case report of a 38 year old femalepatient describes the gingival enlargement associatedwith chronic periodontitis. The enlargement extendedbeyond the middle thirds of the clinical crowns of theteeth. It was associated with generalized mobility,pocket formation and bone loss. The patient alsocomplained of bleeding gums and difficu...

  12. Primary gastric melanoma: case report of a rare malignancy

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    Alexander Augustyn

    2015-03-01

    Full Text Available We report the case of a 64-year-old white male who presented to his primary care physician with complaints of fatigue. Physical exam was unremarkable and laboratory studies revealed profound anemia, for which the patient received a transfusion. Esophagogastroduodenoscopy revealed a bleeding mass in the proximal stomach that was histologically determined to be malignant melanoma, with immunohistochemical staining demonstrating positivity for SOX10, S100, MART-1, and HMG-45. After an extensive dermatological exam no other primary lesion was identified. Whole body positron emission tomography (18-FDG-PET/CT demonstrated pathologic uptake only in the area of the proximal stomach. For this reason, primary gastric melanoma was suspected in this patient. The patient underwent subtotal gastrectomy with mass excision followed by Roux-en-Y reconstruction. Very few cases of primary gastric melanoma have been reported. We report this case and present diagnostic criteria for primary non-cutaneous melanoma and discuss potential non-surgical therapies.

  13. Rare neonatal diabetes insipidus and associated late risks: Case report

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    Rivas-Crespo Maximiliano

    2012-05-01

    Full Text Available Abstract Background Most cases of neonatal central diabetes insipidus are caused by an injury, which often results in other handicaps in the patient. The infant’s prognosis will be determined by his or her own early age and disability as well as by the physician’s skill. However, the rarity of this condition prevents the acquisition of personal experience dealing with it. Case Presentation A neonatal hemorrhagic stroke, caused by an aortic coarctation, caused right lower limb paresis, swallowing disability, and central diabetes insipidus in a term infant. The scant oral intake, as a consequence of his disability, caused progressive undernutrition which closed a vicious circle, delaying his development and his ability to overcome the swallowing handicap. On the other hand, nasal desmopressin absorption was blocked by several common colds, resulting in brain bleeding because of severe dehydration. This even greater brain damage hampered the improvement of swallowing, closing a second harmful circle. Moreover, a devastating central myelinolysis with quadriplegia, caused by an uncontrolled intravenous infusion, consummated a pernicious sequence, possibly unreported. Conclusions The child’s overall development advanced rapidly when his nutrition was improved by gastrostomy: This was a key effect of nutrition on his highly sensitive neurodevelopment. Besides, this case shows potential risks related to intranasal desmopressin treatment in young children.

  14. A case study of occipital outgrowth: a rare suboccipital abnormality.

    Science.gov (United States)

    Mushkin, A Y; Gubin, A V; Ulrich, E V; Snischuk, V P

    2016-05-01

    To describe the clinical and radiological characteristics of uncommon upper cervical spine abnormality in children. Clinical and diagnostic characteristics of three patients aged 6-12 years with a similar uncommon type of occipital anomaly are described. The patients were admitted in 2007, 2009, and 2014, respectively. All patients were clinically and radiologically examined. In each case the massive, additional unilateral outgrowth of the occipital bone (os occipitale) was visualized. The signs and symptoms included torticollis, acute brain ischemia, and limited head motion. Two of the three patients underwent surgical treatment: an occipital-cervical fusion was performed in the first patient, and the outgrowth was removed in the second patient. After 1 year of follow-up the results were estimated as good for both patients, with better functional outcome for the second patient. The parents of the third patient did not consent for the surgical treatment. The unique features of this abnormality distinguish it from previous descriptions of the manifestation of pro-atlas, atlas, or atlanto-occipital synostosis. The presented abnormality had different manifestation of various severity in each case, from torticollis to acute vascular disorder. Clinical case series. IV.

  15. A rare case of hidebound disease with dental implications

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    Vikram Bali

    2013-01-01

    Full Text Available Systemic sclerosis (also called as Scleroderma or hidebound disease is a chronic sclerotic disease of unknown etiology which causes diffuse, increased deposition of extra cellular matrix in connective tissue with vascular abnormalities, resulting in tissue hypoxia. The disease is characterized by diffuse fibrosis; degenerative changes; and vascular abnormalities in the skin (scleroderma, articular structures, and internal organs. Aesthetic and facial dysfunctions are followed by important oral and facial manifestations. Most oral manifestations begin with tongue rigidity and facial skin changes. Bone resorption of mandibular angle and widening of periodontal ligament space on periapical radiographs are important radiological findings. Other systemic changes include the involvement of internal organs, which lead to serious complications as well as disorders in the cardiac muscle and Raynaud΄s phenomenon. This is a case report of 30-year-old female patient with the classical features of this disease. This case is reported for its rarity and variable expressivity. The main aim of this article is to describe thorough presentation of the case report, various forms of scleroderma, pathogenesis, oral, extraoral, periodontal manifestations of scleroderma, and its treatment options. A brief review of the literature, focusing on dental alterations is also presented.

  16. A rare case of hidebound disease with dental implications.

    Science.gov (United States)

    Bali, Vikram; Dabra, Sarita; Behl, Ashima Bali; Bali, Rajiv

    2013-07-01

    Systemic sclerosis (also called as Scleroderma or hidebound disease) is a chronic sclerotic disease of unknown etiology which causes diffuse, increased deposition of extra cellular matrix in connective tissue with vascular abnormalities, resulting in tissue hypoxia. The disease is characterized by diffuse fibrosis; degenerative changes; and vascular abnormalities in the skin (scleroderma), articular structures, and internal organs. Aesthetic and facial dysfunctions are followed by important oral and facial manifestations. Most oral manifestations begin with tongue rigidity and facial skin changes. Bone resorption of mandibular angle and widening of periodontal ligament space on periapical radiographs are important radiological findings. Other systemic changes include the involvement of internal organs, which lead to serious complications as well as disorders in the cardiac muscle and Raynaud΄s phenomenon. This is a case report of 30-year-old female patient with the classical features of this disease. This case is reported for its rarity and variable expressivity. The main aim of this article is to describe thorough presentation of the case report, various forms of scleroderma, pathogenesis, oral, extraoral, periodontal manifestations of scleroderma, and its treatment options. A brief review of the literature, focusing on dental alterations is also presented.

  17. Bilateral Breast Abscess Caused by E. coli in a Non-lactating Woman: A Rare Case.

    Science.gov (United States)

    Şimşek, Gürcan; Gündeş, Ebubekir; Tekin, Şakir; Tavlı, Şakir

    2014-07-01

    Breast abscess usually occurs during lactation and the responsible organism is often S. Aureus . Breast abscess in non-lactating women is extremely rare and limited data is available in the literature regarding this entity. In our study, a 36-year-old non-lactating female patient who developed bilateral breast abscess due to E. coli infection without any predisposing factors has been discussed in light of the literature.

  18. A Rare Case Report of a Collodion Baby with Severe Hypernatremia

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    Ahmad Farhat

    2017-06-01

    Full Text Available Background: Collodion baby is a rare condition (i.e., 1:300000 birth, which is referred to a neonate covered with a tight and shiny membrane desquamating within two weeks. This condition takes place as a result of the epidermal cornification disorder. Given the impairment of the skin barrier function, these neonates are at the risk of several complications, including hypernatremic dehydration, hypothermia, skin infections, fissures, conjunctivitis, sepsis, dehydration, and constrictive bands of the extremities resulting in vascular compromise and edema. This condition has a high mortality rate; accordingly, the majority of the collodion babies die within the first few weeks of birth due to the secondary complications depending on the type of mutations. Although the collodion membrane is a transient condition, it can lead to a number of complications in 45% of the newborns and result in 11% mortality rate in the first few weeks of the neonatal life. However, in the recent years, the mortality rates have been declined owing to the introduction of systemic retinoids to the clinical practice and the advanced care methods, which are used in the intensive care units. Harlequin ichthyosis has been associated with the mutation in the ABCA12 gene; therefore, genetic counseling and mutation screening of this gene should be considered.Case report: Herein, we reported a case of a collodion baby with favorable evolution, who developed hypernatremia and suffered from some complications having aroused from the condition.Conclusion: Infection prevention and supportive care of collodion babies mainly with incubator, serum therapy, and feeding support can be effective in preventing complications.

  19. Occupational Maxillofacial Trauma: Report of a Rare Case

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    Dervisoglou Theodoros

    2015-03-01

    Full Text Available Maxillofacial trauma, any physical trauma in the face, can involve soft tissues (lacerations, avulsions, bruises etc, bone injuries (fractures and dislocation, avulsed or fractured teeth (dental issues and special regions (nerves, eyes, salivary glands etc. As the most exposed part of the human body, the face can be susceptible to injuries in work-related accidents. Occupational accident in the maxillofacial region rates 0.9-5% and, in some cases, can reach 9%. Based on their occupation, patients are classified as farm and forestry workers, construction workers, factory workers, craftsmen, service workers, and office workers.

  20. Eagle′s syndrome: A rare case of young female

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    Mohammad Abdul Baseer

    2013-01-01

    Full Text Available Eagle′s syndrome is a condition that causes pain in the Craniofacial and cervical region of the neck. Symptoms related to the Eagle′s syndrome may be confused with the variety of neuralgias, oral, dental and temporomandibular joint (TMJ conditions. In this paper, a case of the very young female suffering with the difficulty in swallowing and recurrent dull pain in the throat with restriction of the movement of head to the left side was presented. A thorough past medical and dental history, extra oral and intra oral examination coupled with the panoramic radiographic interpretation were used to diagnose Eagle′s syndrome.

  1. A rare case of ileal metastasis from cervical cancer.

    Science.gov (United States)

    Iliescu, L; David, L; Orban, C; Herlea, V; Toma, L

    2014-01-01

    We present the case of a 70-year-old woman, with a history of radiation-treated and surgically- resected cervical cancer, who was admitted to our clinic for intermittent sub occlusive symptoms. CT scan revealed a liver nodule and intestinal obstruction. The patient underwent surgery for excision of suspected liver metastasis and resolution of intestinal obstruction.Intraoperatively an ileal tumour was found to be the cause of the obstruction. Anatomo-pathological findings were consistent with an ileal metastasis from the cervical cancer.The liver nodule was only an area of focal steatosis. Celsius.

  2. Partial prune belly syndrome: A rare case report

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    Aditya Pratap Singh

    2017-01-01

    Full Text Available Prune belly syndrome (PBS is characterized by deficient development of abdominal muscles that causes the skin of the abdomen to wrinkle like a prune, bilateral cryptorchidism, abnormalities of the urinary tract. The etiology of PBS is unclear and possible familial genetic inheritance was reported in some of the studies. We are presenting here a case with the absence of the muscle in the right side of the abdomen as hernia, thinning of the muscle on left side with bilateral cryptorchidism, and abnormalities of the urinary tract. It is the partial presentation of the PBS.

  3. How to assess extreme weather impacts - case European transport network

    Science.gov (United States)

    Leviäkangas, P.

    2010-09-01

    To assess the impacts of climate change and preparing for impacts is a process. This process we must understand and learn to apply. EWENT (Extreme Weather impacts on European Networks of Transport) will be a test bench for one prospective approach. It has the following main components: 1) identifying what is "extreme", 2) assessing the change in the probabilities, 3) constructing the causal impact models, 4) finding appropriate methods of pricing and costing, 5) finding alternative strategy option, 6) assessing the efficiency of strategy option. This process follows actually the steps of standardized risk management process. Each step is challenging, but if EWENT project succeeds to assess the extreme weather impacts on European transport networks, it is one possible benchmark how to carry out similar analyses in other regions and on country level. EWENT approach could particularly useful for weather and climate information service providers, offering tools for transport authorities and financiers to assess weather risks, and then rationally managing the risks. EWENT project is financed by the European Commission and participated by met-service organisations and transport research institutes from different parts of Europe. The presentation will explain EWENT approach in detail and bring forth the findings of the first work packages.

  4. Rare pathological findings in cases of complicated migraine

    International Nuclear Information System (INIS)

    Wessely, P.; Zeiler, K.; Holzner, F.; Kristoferitsch, W.

    1986-01-01

    137 patients suffering from classical or complicated migraine were investigated in the Neurology Department of the University of Vienna between 1971 and 1984. 13 of these patients were found to have pathological alterations and their case histories are presented. Clinically, 11 patients suffered from migraine accompagnee (in 2 cases accompanied by epileptic seizures), 1 patient had ophthalmoplegic migraine and 1 had a subarachnoid haemorrhage imitating migraine. The underlying pathological findings were: 1 tumour, 4 arteriovenous malformations, 4 aneurysms, 1 arterio-venous shunt, 1 pathological vascular network, 1 Moya-Moya syndrome and 1 intracerebral haemorrhage without detectable source of bleeding. 8 of the patients underwent successful surgery and most of them showed subsequent clinical improvement. The family history was positive in only 2 patients. The time interval between the occurence of the first symptoms and the establishment of the final diagnosis was up to 25 years. The neurologist should undertake extensive investigation of the patient, including cerebral angiography, if the following criteria apply: hemicrania consistently on the same side; change in type of headache after a number of years; uniform complicating neurological symptoms; additional occurence of epileptic seizures; manifestation of neurological symptoms after the prodromal phase; persistent neurological signs without remission; negative family history; persisting diffuse or locally accentuated EEG changes; pathological CAT results. (Author)

  5. A Rare Case of Medulloblastoma with Excessive Nodularity: Imagistic Features

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    Tascu A.

    2014-10-01

    Full Text Available Medulloblastoma is the most common malignant tumor of childhood. Neuroimaging can play a role in the diagnosis of medulloblastoma, however atypical features do exist [2]. We report the case of a 1 year and 10- month-old infant diagnosed with a medulloblastoma with what we term “excessive” nodularity based on neuroimaging features and confirmed by neuropathology. CT-scan (CT and magnetic resonance imaging (MRI examination of the brain revealed a very large posterior fossa tumor attached to tentorium. On T2-weighted and post-gadolinium sequences, the tumor shows an extensive nodular grape-like appearance. Initial the patient was underwent a ventriculo-peritoneal shunt. The second operative procedure was tumour resection. Histology examen revealed a diagnosis of medulloblastoma desmoplastic with extensive nodularity. The neuroradiographic features of this medulloblastoma with what we describe as “excessive” nodularity are important to recognize as these children may be cured with chemotherapy alone.

  6. A rare case of cytomegalovirus papillitis in patient with immunodeficiency

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    Dinda A. Devona

    2016-10-01

    Full Text Available A 26-year-old male diagnosed with AIDS came with sudden blurred vision and central sco-toma in left eye since 2 weeks before admission. His visual acuity was counting finger at 5 meters with normal IOP and anterior segment. The posterior segment revealed edematous optic nerve covered by exudates and hemorrhages. Due to low CD4+ count and serological test result, we considered a HIV-related opportunistic ocular infection, specifically HSV infection. As visual acuity worsened during treatment with acyclovir, we performed PCR ex-amination from aqueous tap which revealed positive CMV DNA. Unfortunately, the visual acuity had worsened to no light perception before he received any specific anti-CMV agent. CMV papillitis is an unusual presentation of CMV retinitis. PCR examination from aqueous or vitreous tap should be performed while waiting for serological test result, especially in doubtful cases. Therefore, appropriate diagnosis and management can be established early to prevent irreversible visual loss.

  7. A Rare Case of Mediterranean Spotted Fever and Encephalitis

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    Raquel Sousa Almeida

    2016-01-01

    Full Text Available Mediterranean spotted fever is a tick-borne zoonotic disease caused by Rickettsia conorii. It is transmitted by the dog tick Rhipicephalus sanguineus. It usually presents as a benign self-limited disease characterized by a skin rash, high fever, and, sometimes, a characteristic ulcer at the tick bite site called tache noir. The course of this disease is usually benign, although severe manifestations have been previously described, mainly in adults. Neurological manifestations are very unusual. We present a case of Mediterranean spotted fever with encephalitis to highlight the importance of clinical suspicion, mainly in endemic areas, the potential severity of this disease, and the need of early initiation of therapy in order to prevent severe complications.

  8. GITELMAN SYNDROME AND PREGNANCY- A RARE CASE REPORT

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    Venkatachalam Sibiya

    2017-09-01

    Full Text Available PRESENTATION OF CASE A 22 years old G2 P1 D1 came with complaints of shivering of both upper limb and lower limb. She was diagnosed with Gitelman syndrome in previous pregnancy. Previous was a preterm delivery due to polyhydramnios and the baby died at 12 days of life due to some congenital malformation of heart. In previous pregnancy, patient presented with paralysis at 6 months of gestation and was treated conservatively by correcting the electrolytes level. In the present pregnancy, patient had persistent hypokalaemia and hypomagnesaemia, which was treated. Anomaly scan was done. No gross anomaly was detected. Patient is symptomatically better and she is continuing her pregnancy, hope better outcome since GS has no adverse effect on pregnancy.

  9. Rare Abdominal Wall Malformation: Case Report of Umbilical Cord Hernia

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    Andro Gliha

    2018-02-01

    Full Text Available The umbilical cord hernia is the rarest form of abdominal wall malformations, anatomically completely different from gastroschisis and omphalocele. It occurs due to the permanent physiological evisceration of abdominal organs into umbilical celom and persistence of a patent umbilical ring. The umbilical cord hernia is often mistaken for omphalocele and called “small omphalocele”. Here we present a case of a female newborn with umbilical cord hernia treated in our Hospital. After preoperative examinations surgery was done on the second day of life. The abdominal wall was closed without tension. The aim of this article is to present the importance of the proper diagnose of these three entities and to stimulate academic community for the answer, is this umbilical cord hernia or small omphalocele.

  10. Rare Abdominal Wall Malformation: Case Report of Umbilical Cord Hernia.

    Science.gov (United States)

    Gliha, Andro; Car, Andrija; Višnjić, Stjepan; Zupancic, Bozidar; Kondza, Karmen; Petracic, Ivan

    The umbilical cord hernia is the rarest form of abdominal wall malformations, anatomically completely different from gastroschisis and omphalocele. It occurs due to the permanent physiological evisceration of abdominal organs into umbilical celom and persistence of a patent umbilical ring. The umbilical cord hernia is often mistaken for omphalocele and called "small omphalocele". Here we present a case of a female newborn with umbilical cord hernia treated in our Hospital. After preoperative examinations surgery was done on the second day of life. The abdominal wall was closed without tension. The aim of this article is to present the importance of the proper diagnose of these three entities and to stimulate academic community for the answer, is this umbilical cord hernia or small omphalocele.

  11. Polycythemia vera presenting with bilateral papilledema: A rare case report

    Directory of Open Access Journals (Sweden)

    Parija S

    2008-01-01

    Full Text Available A 45-year-old male patient presented with gradual onset of headache, vomiting and blurring of vision of 28 days duration. Ophthalmological examination revealed normal anterior segment and pupillary reflex. No abnormality was detected in the vitreous. Optic disc showed features of advanced papilledema with normal macula and retinal periphery in both eyes. Visual acuity was 20/200 in the right eye and counting fingers close range in the left eye. Non-contrast computed tomography of brain was normal and magnetic resonance imaging showed sagittal sinus thrombosis without any evidence of venous infarction or intracranial mass. Routine hematological investigations revealed increased hemoglobin level, packed cell volume and leucocytosis. Further investigation revealed increased Vitamin B12 and decreased serum erythropoietin. A diagnosis of polycythemia vera was made from the above findings. This case is being presented for the rarity of association of polycythemia vera with bilateral advanced papilledema due to sagittal sinus thrombosis.

  12. Malign Fibrous Histiocytoma of the Bladder: A Rare Case Report

    Directory of Open Access Journals (Sweden)

    Ižbrahim Bozkurt

    2014-06-01

    Full Text Available Malignant fibrous histocytoma is a mesenchimal tumor, which was described in 1964. It is the most common soft tissue sarcoma in patients over the age of 40 years. There were very few reports about malignant fibrous histocytoma in urinary tract especially in bladder with 30 patients. Patients usually present with gross hematuria. Because of its agressive characteristics; recurrences, progressions and metastasis are likely. Tumor grade, tumor size, amount of invasion and histological type are the risk factors for metastasis. Early radical cystectomy is the first treatment option because of poor prognosis of these tumors but usually can not be sufficient. Chemotherapy and radiotherapy are used to as an alternative treatment or adjuvant treatment with surgery. We would like to present a bladder malignant fibrous histocytoma case to contribute to the lirature.

  13. Ascher′s syndrome: A rare case report

    Directory of Open Access Journals (Sweden)

    Shivcharan Lal Chandravanshi

    2015-01-01

    Full Text Available An 18-year-old Indian girl with upper lip deformity presented with on and off painless swelling of her both upper eyelids for 3 years. Clinical evaluation revealed bilateral blepharochalasis, narrowing of horizontal palpebral fissure, decreased outer intercanthal distance, iris coloboma, cleft soft palate, bifid uvula, sensorineural deafness and double upper lip. Clinical examination of the thyroid, thyroid hormone assay and ultrasonography revealed normal thyroid gland structure and function. Ascher′s syndrome was diagnosed. To our knowledge, this is the first reported case of Ascher′s syndrome associated with iris coloboma, heterochromia iridum, and narrowing of horizontal palpebral fissure and decreased outer intercanthal distance secondary to lengthening of lateral canthal ligament.

  14. Systemic Lupus Erythematosus with Hepatosplenic Granuloma: A Rare Case

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    Anju Bharti

    2014-01-01

    Full Text Available Background. Systemic lupus erythematosus (SLE is an autoimmune disease which is known to present with a wide variety of clinical manifestations. Case Report. A 15-year-old male presented with complaints of moderate grade fever and generalized body swelling. There was no history of cough, weight loss, joint pain, oral ulcerations, skin rash, photosensitivity, loss of hair, pain abdomen, jaundice, or any significant illness in the past. Contrast enhanced computerized tomography of the abdomen revealed hypodense lesions in both liver and spleen (without contrast enhancement, suggestive of granulomas along with few retroperitoneal and mesenteric lymph nodes. On the basis of immunological tests and renal biopsy report, SLE with hepatosplenic granulomatosis diagnosis was made. He was given pulse methylprednisolone 500 mg, for 3 days and he showed dramatic improvement clinically. Conclusion. Hepatic and splenic granulomas are not common in SLE, but this should be kept in differential diagnosis.

  15. Cavernous hemangioma of the tongue: A rare case report

    Directory of Open Access Journals (Sweden)

    K A Kamala

    2014-01-01

    Full Text Available Hemangiomas are developmental vascular abnormalities and more than 50% of these lesions occur in the head and neck region, with the lips, tongue, buccal mucosa, and palate most commonly involved. They are considered as hamartomas rather than true neoplasms. Here we report a case of hemangioma of the body of the tongue, discussing the diagnostic aspects and treatment modalities of such lesion and emphasizing the role of the color Doppler ultrasonography, especially in the diagnosis and treatment. Factors such as patient′s age, size and site of lesion and the proximity of lesion to vital structure are paramount in the determination of the therapeutic approach and surgical excision. Even though radiotherapy, cryotherapy, laser therapy, medical treatment, injection of sclerosing substances and the selective embolization of the lingual artery seem to have some efficacy, the author conclude that surgery is the therapy of choice in the isolated vascular lesions of the body of the tongue.

  16. A rare case of ceftriaxone induced anaphylaxis in anaethesia practice

    Directory of Open Access Journals (Sweden)

    Anita Kumari

    2015-01-01

    Full Text Available Medications are among the second most common cause of anaphylaxis and the primary cause of anaphylaxis in adults. The most common classes of drugs causing anaphylaxis are antibiotics especially β-lactam antibiotics and nonsteroidal anti-inflammatory drugs (NSAIDs. Diagnosis of anaphylaxis is clinically based and usually straight forward. However data on epidemiology of anaphylaxis, particularly the most profound and life threatening form such as anaphylactic shock is limited and thought to be under-reported. In spite of negative skin testing, our patient had severe reaction resulting in anaphylactic shock after antibiotic administration but was managed successfully without any residual compromise. This case reflects the limitations of screening test done preoperatively for the diagnosis of sensitization to the drugs.

  17. A rare case of a retroperitoneal enterogenous cyst with in-situ adenocarcinoma

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    Woodland James G

    2007-10-01

    Full Text Available Abstract Background Retroperitoneal enterogenous cysts are uncommon and adenocarcinoma within such cysts is a rare complication. Case presentation We present the third described case of a retroperitoneal enterogenous cyst with adenocarcinomatous changes and only the second reported case whereby the cyst was not arising from any anatomical structure. Conclusion This case demonstrates the difficulties in making a diagnosis as well as the importance of a multi-disciplinary approach, and raises further questions regarding post-operative treatment with chemotherapy.

  18. Ultrasound, CT and MRI Appearances of a Rare Symptomatic Laryngeal Chondrometaplasia: A Case Report

    International Nuclear Information System (INIS)

    Lee, Ryan Ka Lok; Hok Yuen, Edmond Yuen; Abdullah, Victor James; Ping Lee, Yolanda Yim; Ahuja, Anil Tejbhan

    2015-01-01

    Symptomatic laryngeal chondrometaplasia is rare. To the best of our knowledge, there are only few case reports on laryngeal chondrometaplasia. The imaging appearance of this uncommon disease is even more rarely described. There are only two case reports describing its appearances in computed tomography (CT) and magnetic resonance imaging (MRI). Ultrasound (US) features have not been reported so far. This case report is to show the US, CT and MRI features of this disease entity to stress the role of imaging in this disease

  19. [A rare case of diencephalic cachexia in an adult female with cranio-pharyngioma].

    Science.gov (United States)

    Klochkova, I S; Astaf'eva, L I; Konovalov, A N; Kadashev, B A; Kalinin, P L; Sharipov, O I; Kutin, M A; Sidneva, Yu G; Shishkina, L V; Pronin, I N

    Diencephalic cachexia (DС) is progressive weight loss despite a normal caloric intake and a satisfactory state of health, which is caused by hypothalamic lesions. This is a rare (about 100 cases were reported) and potentially fatal disorder of unknown pathogenesis. At present, there is no effective pharmacological therapy for the disorder. Cachexia may regress only if the tumor reduces in size, therefore the timely diagnosis and treatment are of vital importance for the patient. DС is typical of early childhood, and only a few cases have been reported in adults. We present a rare case of DС in a 24-year-old female with papillary craniopharyngioma.

  20. Systemic Mastocytosis: A Rare Case of Increased Liver Stiffness

    Directory of Open Access Journals (Sweden)

    Stefanie Adolf

    2012-01-01

    Full Text Available Assessment of liver stiffness (LS by transient elastography (Fibroscan has significantly improved the noninvasive diagnosis of liver fibrosis. We here report on a 55-year-old patient with drastically increased LS due to previously unknown systemic mastocytosis. The patient initially presented with increased weight loss, nocturnal pruritus, increased transaminases, bilirubinemia, and thrombocytopenia. Abdominal ultrasound showed ascites, hepatomegaly, and splenomegaly. In addition, LS was 75 kPa (IQR 0 kPa clearly exceeding the cut-off value for F4 cirrhosis of 12.5 kPa. However, histological analysis of the liver specimen indicated liver involvement by systemic mastocytosis and excluded liver cirrhosis. An additional CT scan detected disseminated bone lesions. After three months of treatment with Midostaurin, LS slightly decreased down to 31.9 kPa (IQR 8.3 kPa. This case illustrates that diffused sinusoidal neoplastic infiltrates are a pitfall in the non-invasive diagnosis of liver cirrhosis. In conclusion, refined clinical algorithms for increased LS should also include mastocytosis in addition to inflammation, congestion, and biliary obstruction.