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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. Extremely rare borderline phyllodes tumor in the male breast: a case report.

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    Kim, Jung Gyu; Kim, Shin Young; Jung, Hae Yoen; Lee, Deuk Young; Lee, Jong Eun

    2015-01-01

    Phyllodes tumor of the male breast is an extremely rare disease, and far fewer cases of borderline phyllodes tumors than benign or malignant tumors in the male breast have been reported. We report a case of borderline phyllodes tumor in the male breast with imaging findings of the tumor and pathologic correlation. Copyright © 2015 Elsevier Inc. All rights reserved.

  3. 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

    2018-02-19

    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.

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

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    Aline Lariessy Campos Paiva

    2017-11-01

    Full Text Available ABSTRACT CONTEXT: 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. CASE REPORT: 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. CONCLUSIONS: 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.

  5. Synovial sarcoma of the maxillary sinus: an extremely rare case with excellent response to chemotherapy

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    Saito S

    2018-01-01

    Full Text Available Shin Saito,1 Hiroyuki Ozawa,1 Yuuichi Ikari,1 Nana Nakahara,1 Fumihiro Ito,2 Mariko Sekimizu,1 Junichi Fukada,3 Kaori Kameyama,4 Kaoru Ogawa1 1Department of Otorhinolaryngology – Head and Neck Surgery, Keio University, School of Medicine, 2Department of Otorhinolaryngology – Head and Neck Surgery, NHO Tokyo Medical Center, 3Department of Radiology, 4Department of Pathology, Keio University, School of Medicine, Tokyo, Japan Abstract: This paper presents an extremely rare case of synovial sarcoma arising from the maxillary sinus, which resulted in a clinically complete response to chemotherapy. Synovial sarcoma is a rare soft tissue malignant tumor, most commonly affecting the extremities. While ~10% occur in the head and neck region, synovial sarcoma of the sinonasal tract is extremely rare, with only 11 cases having been reported previously. As with other sarcomas, the standard treatment is complete resection while allowing for a safe margin, but this is often difficult in the head and neck area due to the complicated anatomy there. This makes the treatment of head and neck sarcoma challenging and leads to the need for a multimodal approach in advanced cases. However, the exact efficacy of chemotherapy is not well understood. In this report, we present a case of unresectable maxillary sinus synovial sarcoma that was successfully treated by chemotherapy followed by radiation therapy. A 53-year-old Japanese man was referred to our hospital with a history of left nose obstruction over the previous couple of years. Computed tomography/magnetic resonance imaging revealed a tumor arising from the maxillary sinus that extended to adjacent tissues. A biopsy was performed, and the tumor was diagnosed as synovial sarcoma. Since the tumor was unresectable, neoadjuvant chemotherapy was administered. The response was excellent, and the tumor became undetectable under endoscopy and radiological imaging. This provided us with a clinical evaluation of

  6. Malignant mesothelioma of tunica vaginalis: an extremely rare case presenting without risk factors

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    Akin, Yigit; Bassorgun, Ibrahim; Basara, Isil; Yucel, Selcuk

    2015-01-01

    Testicular tumours have many different manifestations, including hydrocele formation. Herein, we present an extremely rare case of testicular mesothelioma presenting with left hydrocele, but without risk factors. Left radical inguinal orchidectomy was performed, and pathological examination revealed a malignant mesothelioma of the tunica vaginalis of the testis. No infiltration of the spermatic cord was evident, and upon advanced radiological evaluation, no sign of metastasis was detected. Follow-up was still ongoing in our urology outpatient clinic at the time of this report. Although hydrocele is a simple and common condition that is easy to diagnose, a detailed investigation should be performed. Thus, when encountering a patient with hydrocele, the clinician should evaluate the possibility of the presence of an underlying testicular/paratesticular tumour, including a rare one such as mesothelioma of the tunica vaginalis. PMID:25820862

  7. 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.

  8. 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.

  9. Three's a Crowd - An Extremely Rare Case of Cor Triatriatum Dexter.

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    Elagizi, A; Marvin, R; O'Bryan, G; Vyas, V; Arcement, L

    2017-01-01

    Cor triatriatum is a congenital cardiac anomaly in which the left (sinister) or right (dexter) atrium is divided into two compartments by residual embryonic tissue, resulting in a tri-atrial heart. As cor triatriatum dextrum can present clinically in various ways and have multiple associated cardiac anomalies, this report attempts to contribute to what is known about this exceedingly rare disorder. A 40 year old Hispanic man with a medical history of gastritis presented with complaints of palpitations, dizziness and bilateral lower extremity edema. He was found to have atrial fibrillation and new onset heart failure. The patient was admitted for rate control and further evaluation, which revealed several cardiac anomalies. Initial 2D echocardiography demonstrated severe right atrial enlargement, right ventricular hypertrophy and an engorged coronary sinus, which prompted further assessment of the patient's cardiovascular anatomy. Transesophageal echocardiography (TEE) revealed a severely enlarged, septated right atrium with a possible unroofed coronary sinus and a small patent foramen ovale (PFO). Left- and right-heart catheterization established a coronary-cameral fistula between the right coronary artery (RCA) and right atrium, as well as left-to-right shunt. The patient improved clinically with conservative management including diet modification, furosemide and digoxin for fluid and rate control, and was referred to cardiothoracic surgery for further evaluation. Cor triatriatum dextrum is an extremely rare cardiac condition: In high-volume echocardiographic laboratories, prevalence is less than 0.01 percent. This case highlights the association between cor triatriatum and other congenital cardiac anomalies, including persistent left superior vena cava with an unroofed coronary sinus, PFO and left-to-right shunt; all of which were found in this patient. While cases of cor-triatriatum sinistrum often require correction in infancy (due to left sided heart failure

  10. 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.

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

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    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.

  12. 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.

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

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    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.

  14. [Osteoid osteoma in the lower extremity of the radius: about a case, rare location and review of the literature].

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    Abdelhafid, Derfoufi; Moncef, Erraji; Abdessamad, Kharraji; Najib, Abdeljaouad; Hicham, Yacoubi

    2016-01-01

    Osteoid osteoma is a benign but painful bone tumor whose treatment involves complete surgical resection. We report the case of a young patient with osteoid osteoma in the lower extremity of the radius.

  15. A Rare Case of Aggressive Digital Adenocarcinoma of the Lower Extremity, Masquerading as an Ulcerative Lesion that Clinically Favored Benignancy

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    Ryan Vazales

    2014-08-01

    Full Text Available A rare case report of Aggressive Digital Adenocarcinoma (ADPCa is presented complete with a literature review encompassing lesions that pose potential diagnostic challenges. Similarities between basal cell carcinoma (BCC, marjolin’s ulceration/squamous cell carcinoma (MSCC and ADPCa are discussed. This article discusses potential treatment options for ADPCa and the need for early biopsy when faced with any challenging lesion. An algorithmic approach to ADPCa treatment based on the most current research is recommended.

  16. Neurofibromatosis and caroli's disease: an extremely rare association

    International Nuclear Information System (INIS)

    Arfan ul Bari; Mehmood, T.; Hussain, S.; Yousaf, R.; Majeed, S.; Simeen ber Rahman

    2004-01-01

    Neurofibromatosis type-1 (NF-1) is a rare genetic disorder with an extremely variable phenotype. A broad spectrum of associations have also been reported with it. We present a florid case of NF, presenting with unusual symptoms, which was found to have an associated Caroli's disease, a rare congenital disorder of the intrahepatic bile ducts. The case is reported along with a brief review of both the disorders. (author)

  17. 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.

  18. Case studies in rare aneurysm localisations

    International Nuclear Information System (INIS)

    Kleinsorge, F.; Berg-Schlosser, V.; Riester, K.P.; Dombrowski, H.

    1987-01-01

    We describe four cases of rare aneurysm localisations, one case of an aneurysm of the hepatic artery, two cases of aneurysms of the superior mesenteric artery and its branches and, as an extreme rarity, one case of an aneurysm of the superior gluteal artery (only three known cases up to now). All cases were documented by arteriography/DSA, ultrasonography and computed tomography. We refer to incidence, aetiology, symptomatology, and possible complications as indicated by literature. (orig.) [de

  19. Case studies in rare aneurysm localisations

    Energy Technology Data Exchange (ETDEWEB)

    Kleinsorge, F.; Berg-Schlosser, V.; Riester, K.P.; Dombrowski, H.

    1987-07-01

    We describe four cases of rare aneurysm localisations, one case of an aneurysm of the hepatic artery, two cases of aneurysms of the superior mesenteric artery and its branches and, as an extreme rarity, one case of an aneurysm of the superior gluteal artery (only three known cases up to now). All cases were documented by arteriography/DSA, ultrasonography and computed tomography. We refer to incidence, aetiology, symptomatology, and possible complications as indicated by literature.

  20. 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.

  1. Myocarditis, pancreatitis, polyarthritis, mononeuritis multiplex and vasculitis with symmetrical peripheral gangrene of the lower extremities as a rare presentation of leptospirosis: a case report and review of the literature

    OpenAIRE

    Panagopoulos, Periklis; Terzi, Irene; Karanikas, Michail; Galanopoulos, Nikolaos; Maltezos, Efstratios

    2014-01-01

    Introduction Leptospirosis is a zoonosis caused by the spirochete, Leptospira interrogans. While most cases of leptospirosis are mild to moderate, the course may be complicated by multiorgan dysfunction. We present a rare case of leptospirosis with acute myocarditis, pancreatitis, polyarthritis, mononeuritis multiplex and severe vasculitis with necrosis of the extremities. Case presentation A 32-year-old man from Congo presented with high-grade fever, confusion and headache. He developed tach...

  2. 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

  3. 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.

  4. Agenesis of permanent canines: Rare case report

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    Sumit Kumar Yadav

    2017-01-01

    Full Text Available Agenesis of permanent canines is a rare condition, and that of both maxillary and mandibular permanent canines is extremely rare. It may occur either isolated or in association with other dental anomalies. Reports of such cases are very scarce in the literature. Need for early diagnosis of such conditions should be emphasized because of functional, esthetic, and psychological problems which should be evaluated and treated appropriately. The present paper presents a report of bilaterally missing permanent maxillary and mandibular canines. This case might contribute in the future studies of incidence of agenesis of permanent canines.

  5. Agenesis of permanent canines: Rare case report.

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    Yadav, Sumit Kumar; Yadav, Achla Bharti; Kedia, Neal Bharat; Singh, Abhinav Kumar

    2017-01-01

    Agenesis of permanent canines is a rare condition, and that of both maxillary and mandibular permanent canines is extremely rare. It may occur either isolated or in association with other dental anomalies. Reports of such cases are very scarce in the literature. Need for early diagnosis of such conditions should be emphasized because of functional, esthetic, and psychological problems which should be evaluated and treated appropriately. The present paper presents a report of bilaterally missing permanent maxillary and mandibular canines. This case might contribute in the future studies of incidence of agenesis of permanent canines.

  6. A rare case of biventricular myxoma.

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    Ermek, Tangsakar; Aybek, Naibi; Zhang, Wei-Min; Guo, Yong-Zhong; Guo, Sheng; Mamataly, Azze; Chang, Dong-Qing; Liu, Jun; Zhang, Zong-Gang

    2017-03-27

    Cardiac myxoma is the most common primary cardiac tumor. Approximately 75-80% of myxomas are located in the left atrium. Occurrence of multiple myxomas is extremely rare. We describe a rare case of biventricular myxomas resulting in right ventricular inflow and tricuspid valve obstruction. The lesions were detected by echocardiography and thoracic computerized tomography (CT) and confirmed on positron emission tomography-computed tomography. The patient underwent successful surgical resection of the multiple cardiac myxomas. This kind of biventricular case has not been previously reported. The patient is asymptomatic as of the 10-month follow-up.

  7. 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...

  8. Primary Malignant Fibrous Histiocytoma: A Rare Case

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    Anastasios Katsourakis

    2011-01-01

    Full Text Available Malignant fibrous histiocytoma (MFH of the small intestine is an extremely rare condition. It occurs most commonly in the extremities and the trunk. We report a case of a 67-year-old woman who admitted with fever, myalgia, and altered status. After thorough investigation, a tumor of the jejunum was found. The patient underwent complete surgical removal of the tumor. A diagnosis of MFN (undifferentiated high-grade pleomorphic sarcoma was made. The patient received adjuvant chemotherapy with Gemcitabine. Two years after the operation, the patient died due to recurrence of the disease. MFH of the small intestine is an extremely rare neoplasm with an aggressive biological behaviour. In this paper, pathogenesis, natural history, and treatment are reviewed.

  9. Bilateral extracorporeal testicular ectopia: An extremely rare ...

    African Journals Online (AJOL)

    B.M. Abubakar

    The baby was delivered at home without any complications, as reported ... the time of delivery. There was no history suggestive of other congenital anomalies and no history of maternal febrile illness. The boy's mother is a 20-year-old housewife, P4+0. A3. ... a few cases of iatrogenic scrotoschisis following Cesarean section.

  10. 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.

  11. 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...

  12. 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

  13. GAINT RETROPERITONEAL LIPOSARCOMA: A RARE CASE REPORT

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    Jakkula

    2015-03-01

    Full Text Available INTRODUCTION: Retroperitoneal liposarcoma is a rare malignancy arises from perirenal fat comprises of 0.02 to 0.2% of all malignancies and 10 - 20% of all soft tissue sarcomas . [1,2,3 ] Usual age presentation is 5 - 6 th decade of age with slight male predominance . [4 ] These tumors are usually large , in 20% of patients the tumor is more than 10cms size . [5,6 ] The liposarcoma may have weight and dimension variable ; those over 20 kg are called “giant liposarcomas” and are extremely rare . [ 1,5 ] Com p lete surgical resection [R0] is the only way for survival advantage . [7,8,9 ] We report a case of huge retroperitoneal liposarcoma , which of size of 30><20cms and 8kg weight. Few cases have been reported of this size in literature

  14. A rare case modafinil dependence

    OpenAIRE

    Krishnan, Raman; Chary, Krishnan Vengadaragava

    2015-01-01

    Modafinil, a non-amphetamine psychostimulant, is indicated for narcolepsy, shift work sleep disorder and severe obstructive sleep apnea syndrome. Modafinil is prescribed at the dose of 100 mg once in a day or as two doses, 12 h apart in a day. It has also been found that it reduces cocaine dependence and withdrawal phenomenon. Modafinil is claimed to have very low liability for abuse and dependence. Here we report a rare case of modafinil dependence.

  15. 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.

  16. 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.

  17. Papillary cystadenocarcinoma of submandibular salivary gland: A rare case report

    OpenAIRE

    Mardi Kavita; Sharma Sudarshan; Gupta Neelam

    2010-01-01

    Papillary cystadenocarcinoma is an extremely rare malignant neoplasm characterized by cysts and papillary endophytic projections. It was first defined in 1991 by World Health organization as a separate entity. Major locations of this neoplasm are the parotid gland, the sublingual gland, and minor salivary glands, while occurrence in the submandibular gland is extremely rare. We present a case of papillary cystadenocarinoma arising from the submandibular gland in a 67-year-old male patient. Fu...

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

    Energy Technology Data Exchange (ETDEWEB)

    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.)

  19. Retroperitoneal Schwannoma: A Rare Case

    Directory of Open Access Journals (Sweden)

    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.

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

    International Nuclear Information System (INIS)

    Kim, Man Deuk; Lee, Gun; Shin, Sung Wook

    2005-01-01

    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

  1. 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.

  2. Myocarditis, pancreatitis, polyarthritis, mononeuritis multiplex and vasculitis with symmetrical peripheral gangrene of the lower extremities as a rare presentation of leptospirosis: a case report and review of the literature.

    Science.gov (United States)

    Panagopoulos, Periklis; Terzi, Irene; Karanikas, Michail; Galanopoulos, Nikolaos; Maltezos, Efstratios

    2014-05-14

    Leptospirosis is a zoonosis caused by the spirochete, Leptospira interrogans. While most cases of leptospirosis are mild to moderate, the course may be complicated by multiorgan dysfunction. We present a rare case of leptospirosis with acute myocarditis, pancreatitis, polyarthritis, mononeuritis multiplex and severe vasculitis with necrosis of the extremities. A 32-year-old man from Congo presented with high-grade fever, confusion and headache. He developed tachycardia and hypotension followed by electrocardiogram changes and elevation of troponin I levels suggesting myocarditis. A physical examination revealed conjunctival suffusion, polyarthritis of his lower extremities and cutaneous necrosis of his feet due to vasculitis. Laboratory findings included amylase levels 10-fold the upper normal serum levels and thrombocytopenia. The diagnosis was confirmed by a positive leptospira immunoglobulin M, negative immunoglobulin G and a positive rapid agglutination test. Our patient recovered progressively with antimicrobials and supportive care. Because the clinical features and diagnostic findings of leptospirosis are not specific, a high index of suspicion must be maintained for the diagnosis. Serology is the most important tool for accurate and quick diagnosis in order to administer the appropriate therapy.

  3. Moebius Syndrome: A Rare Case Report

    Directory of Open Access Journals (Sweden)

    M Srinivasa Raju

    2011-01-01

    Full Text Available Moebius syndrome is an extremely rare disorder. Only approximately few hundred cases have been reported in literature. In a nationwide survey reported in 2003, the prevalence of this syndrome was at least 0.002% of births for the years 1996 to 1998. The definition and diagnostic criteria for Moebius syndrome vary among authors. In most studies, it is defined as congenital facial weakness combined with abnormal ocular abduction. The list of signs and symptoms mentioned in various sources for Moebius syndrome includes more than 20 peculiar features. Here, we report an interesting case of Moebius syndrome in a 12-year-old male patient with all the important peculiar signs and symptoms.

  4. 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.

  5. Intraoral hydatid cyst: A rare case report

    Directory of Open Access Journals (Sweden)

    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.

  6. Papillary cystadenocarcinoma of submandibular salivary gland: A rare case report

    Directory of Open Access Journals (Sweden)

    Mardi Kavita

    2010-01-01

    Full Text Available Papillary cystadenocarcinoma is an extremely rare malignant neoplasm characterized by cysts and papillary endophytic projections. It was first defined in 1991 by World Health organization as a separate entity. Major locations of this neoplasm are the parotid gland, the sublingual gland, and minor salivary glands, while occurrence in the submandibular gland is extremely rare. We present a case of papillary cystadenocarinoma arising from the submandibular gland in a 67-year-old male patient. Further, we have discussed the cytological and histopathological features of this rare entity and reviewed the current literature.

  7. 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.

  8. 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.

  9. Primary laryngeal leishmaniasis: A rare case report

    Directory of Open Access Journals (Sweden)

    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.

  10. Cutaneous collagenous vasculopathy: A rare case report

    Directory of Open Access Journals (Sweden)

    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.

  11. Cutaneous collagenous vasculopathy: A rare case report

    Science.gov (United States)

    Rambhia, Kinjal Deepak; Hadawale, Snehal D.; Khopkar, Uday S.

    2016-01-01

    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. PMID:26955587

  12. Intraoral capillary haemangioma: A rare case report

    Directory of Open Access Journals (Sweden)

    Sushma Parimi

    2016-01-01

    Full Text Available Hemangiomas are common benign vascular tumors of the head and neck region which account for 7% of all benign tumors of infancy and childhood. Adults are rarely affected, and they have a female predilection. Based on the microscopic appearance, they are classified into capillary, cavernous, mixed, and sclerosing variety. Incidence of intraoral capillary hemangioma (CH is infrequent, and its topographical presentation on the palatal mucosa and gingiva marks extreme rarity. They are uncommonly encountered by the dentists. The aim of this article is to present a case of CH in a 46-year-old male who presented with a swelling on the posterior hard palate on the left side involving the palatal gingiva and palatal mucosa.

  13. Enterobius vermicularis infestation leading to Meckel's diverticulitis in an adolescent boy: An extremely rare presentation.

    Science.gov (United States)

    Sharma, Manupriya; Kaul, Rashmi; Chander, Bal

    2018-01-01

    Enterobius vermicularis is an intestinal nematode commonly affecting children worldwide. Its transmission is by feco-oral route. Meckel's diverticulitis due to E. vermicularis infestation is an extremely rare presentation. An 11-year-old boy presented with acute abdomen. During surgery inflamed Meckel's diverticulum (M.D) was seen. Histopathology examination of specimen revealed E. vermicularis . Till date, only one case of E. vermicularis infestation of M.D is reported around five decades ago. This histopathological confirmation is extremely important as the required treatment (Mebendazole) of the infected case along with household contacts can prevent the spread of infection and may avoid surgery in known contacts.

  14. OSTEO ARTICULAR TUBERCULOSIS: RARE CASE REPORTS

    Directory of Open Access Journals (Sweden)

    Kumar

    2015-11-01

    Full Text Available Osteoarticular tuberculosis is one of the rare forms of extrapulmonary tuberculosis especially when it involve the foot bones. This article highlights rare case reports of 3 cases of osteoarticular tuberculosis, 2 affecting the bones of foot and the third, the distal femur. Out of three cases 2 were treated as category 3 of RNTCP (Revised national tuberculosis control programme and for the last case we added Inj. streptomycin during the first 2 months of treatment. It was found that the last case recovered quickly when compared to the other 2 cases. All three cases were very rare presentations amongst osteoarticular tuberculosis.

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

    Directory of Open Access Journals (Sweden)

    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.

  16. Cervical intramural pregnancy: Report of a rare case | Sharma ...

    African Journals Online (AJOL)

    Cervical pregnancy is an extremely rare condition of ectopic pregnancy with potential grave consequences if not diagnosed and treated early. A case of intramural pregnancy in the cervix is being reported for its rarity. Early diagnosis is imperative to preserve fertility, otherwise if the pregnancy is disturbed, it may precipitate ...

  17. 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.

  18. Onychomycosis: A Rare Presentation of Fungal Urinary Tract Infection (UTI in Extremely Preterm Neonate

    Directory of Open Access Journals (Sweden)

    Shilpa Kalane

    2015-04-01

    Full Text Available Onychomycosis refers to nail infections caused by any fungus, including yeasts and nondermatophyte molds. Fungal infection has emerged as an important cause of neonatal infections with significant morbidity and mortality, especially in extremely low and very low birth weight infants. We report a 24-days-old boy who presented with onychomycosis on left ring finger nail associated with fungal urine tract infection. Nail finding helped us in detecting fungal urinary tract infection (UTI. Further studies are needed to evaluate the etiologies and treatment of neonatal onychomycosis, and dermatologists should pay attention to this rare event. Hence we are reporting this rare case.

  19. 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.

  20. Oral malignant melanoma: A rare case report and review

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    Mangala Rakaraddi

    2011-01-01

    Full Text Available Primary malignant melanoma is only rarely found in oral cavity (estimated between 0.2 and 8-0% of all melanomas and occurs approximately four times more frequently in oral mucosa of the upper jaw, usually on the palate, alveolar gingiva and melanoma of mandibular gingiva is extremely rare. The peak age of diagnosis of melanoma is between 55 and 65 years. A biopsy is required to confirm the diagnosis. Here, we present a rare case of malignant melanoma of mandibular gingiva in a 64-year-old female patient which is confirmed by histopathology.

  1. Primary mediastinal synovial sarcoma: A rare case report

    OpenAIRE

    Ershadi, Reza; Rahim, Mohamadbagher; Davari, Hamidreza

    2016-01-01

    Introduction: Synovial sarcomas commonly occur in the extremities of young adults. A primary occurrence in the mediastinum is very rare with only a few reported cases in the world literature. We report a case of mediastinal synovial sarcoma. This paper is about a 47-year-old male who presented with retrosternal chest pain and shortness of breath on exertion. Imaging showed an anterior mediastinal mass. Pathological examination of the resected mass showed a biphasic neoplasm with a spindle ...

  2. 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.

  3. Rhinoentomophthoromycosis: A rare case report

    Directory of Open Access Journals (Sweden)

    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.

  4. A Rare Case: Atypical Measles

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    Ümmü Sena Sarı

    2016-03-01

    Full Text Available Atypical measles has been described in persons who were exposed to wild measles virus several years after they were immunized with killed measles vaccine. Occasionally, it can be caused by live measles vaccines also. It is a clinical picture different from typical measles. In this report, an adult patient with a history of immunization, who presented with high fever, maculopapular rash starting at the palms and soles, and pneumonia, is presented. Atypical measles that was first reported in the 1970s in mostly kids should be considered for differential diagnosis in adult cases presenting with high fever, atypical rash and pneumonia even if patients have a history of immunization

  5. 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.

  6. 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.

  7. Floral double mesiodentes: A rare case report

    Science.gov (United States)

    Singaraju, Gowri Sankar; Reddy, B. Rama Mohan; Supraja, G.; Reddy, K. Narayana

    2015-01-01

    Mesiodens is the most commonly erupting supernumerary in the midline between two maxillary central incisors. Mesiodentes is a rare condition when mesiodens erupt in multiples, which may present as either labial or palatal to permanent central incisors. We report a rare case of double mesiodentes (non-syndromic mesiodentes) with floret like appearance. The presence of mesiodens has functional and esthetic implications. Careful radiographic evaluation should be done to prevent complications during their extraction. PMID:25810670

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

    Directory of Open Access Journals (Sweden)

    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.

  9. Pediatric oral leiomyosarcoma: rare case report.

    Science.gov (United States)

    Divyambika, C V; Sathasivasubramanian, S; Krithika, C L; Malathi, N; Prathiba, D

    2012-01-01

    Soft tissue sarcomas comprise a group of histologically diverse malignant neoplasms arising from mesenchymal cell lines. Among these, leiomyosarcomas are sarcomas exhibiting smooth muscle differentiation. Occurrence of this neoplasm in the oral cavity is exceedingly rare and its presentation is unusual in children. We present a case report of leiomyosarcoma of the oral cavity in an eight-year old child. Primary oral leiomyosarcoma, being a rare entity in children, this case report emphasizes the prompt recognition of this tumor to institute appropriate multimodality treatment.

  10. Pediatric oral leiomyosarcoma: Rare case report

    Directory of Open Access Journals (Sweden)

    C V Divyambika

    2012-01-01

    Full Text Available Soft tissue sarcomas comprise a group of histologically diverse malignant neoplasms arising from mesenchymal cell lines. Among these, leiomyosarcomas are sarcomas exhibiting smooth muscle differentiation. Occurrence of this neoplasm in the oral cavity is exceedingly rare and its presentation is unusual in children. We present a case report of leiomyosarcoma of the oral cavity in an eight-year old child. Primary oral leiomyosarcoma, being a rare entity in children, this case report emphasizes the prompt recognition of this tumor to institute appropriate multimodality treatment.

  11. Idiopathic nodular glomerulosclerosis- a rare case

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    Jazmin I. Yepez Kuri

    2016-10-01

    Full Text Available Nodular glomerulosclerosis is considered a signature lesion of diabetes nephropathy, but when it occurs in the absence of diabetes, it is called Idiopathic Nodular Glomerulosclerosis (ING. Idiopathic nodular glomerulosclerosis is a very rare disease and has been associated with obesity, hypertension, and smoking. We report a case of ING presenting as end-stage renal failure requiring hemodialysis.

  12. Congenital Agenesis of the Internal Jugular Vein: An Extremely Rare Anomaly

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    Oguz Kayiran

    2015-01-01

    Full Text Available Vascular anomalies of major venous vessels are rarely seen. Moreover, congenital absence of internal jugular vein is extremely uncommon. In our case, a female patient presented with primary unknown left cervical mass. Cervical ultrasonography demonstrated absence of right internal jugular vein. In addition, computed tomography and dynamic magnetic resonance imaging scans confirmed this diagnosis. Compensatory left internal jugular vein enlargement mimicked sort of cervical mass. Venous magnetic resonance imaging images revealed the absence of right internal jugular vein with compensatory left internal jugular vein dominance. In the literature, the agenesis of IJV was mentioned in a case with concomitant multiple problems. Here, an asymptomatic case is reported with an incident diagnosis. No interventions were planned upon the patient's request. It should be kept in mind that any kind of anomalies can be seen during venous access and neck surgery.

  13. 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.

  14. 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.

  15. Bilateral kidney matrix stones: a rare case.

    Science.gov (United States)

    Lahyani, Mounir; Rhannam, Yassine; Slaoui, Amine; Touzani, Alae; Karmouni, Tarik; Elkhader, Khalid; Koutani, Abdellatif; Andaloussi, Ahmed Ibn Attya

    2016-01-01

    Kedney matrix stones are a rare form of calculi. Flank pain and urinary tract infections (UTI) are the most common presentations of matrix calculi. The diagnosis is usually made at surgery, but some preoperative radiographic findings might be suggestive. Open surgery was the method of choice for treatment. However, combination of ureterorenoscopy and percutaneous nephrolithotomy (PCNL) was found to be safe and effective. We report a rare case of renal and ureteral matrix stones that were diagnosed and treated by open surgery. We also describe its clinical, radiological and therapeutic features through a review of the literature.

  16. Adult-onset Idiopathic Focal Lower Extremity Dystonia: A Rare Task-Specific Dystonia

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    Ritesh Ramdhani

    2013-03-01

    Full Text Available Background:Adult-onset focal lower extremity (LE dystonia is rare, but there have recently been a number of case series that have reported an idiopathic variant triggered during ambulation.Methods:We describe nine patients with idiopathic, focal task-specific LE dystonia. We conducted a comparative analysis that included our cohort and several recently published case series to further characterize the disorder.Results:A total of 48 patients (37 female, 11 male were compared. The average age of onset was 48 years; 36 patients had distal extremity involvement (75%, 5 proximal (10%, and 7 both proximal and distal (15%. Among 33 patients in which the dystonic side was known, 20 were affected on the left (61%. Inversion of the foot with flexion of one or more toes was the most prevalent pattern in those with distal extremity involvement. Discussion:This is a novel task-specific dystonia triggered during ambulation that is often misdiagnosed as an orthopedic or psychogenic issue.

  17. A rare case of abnormal uterine bleeding caused by cavernous hemangioma: a case report

    OpenAIRE

    Benjamin, Mridula A; Yaakub, Hjh Roselina; Telesinghe, PU; Kafeel, Gazala

    2010-01-01

    Abstract Introduction Cavernous hemangiomas of the uterus are extremely rare, benign lesions. A survey of the current literature identified fewer than 50 cases of hemangioma of the uterus. Case presentation We report a case of cavernous hemangioma of the uterus in a 27-year-old Malay, para 1 woman who presented at our hospital with torrential vaginal bleeding having been transferred by land ambulance from a district hospital 30 minutes away. 11 weeks previously she had an urgent cesarean sect...

  18. A rare case: Hallucination associated with pazopanib.

    Science.gov (United States)

    Demirci, Nebi Serkan; Erdem, Gokmen Umut; Dogan, Mutlu; Ozdemir, Nuriye Yıldırım; Zengin, Nurullah

    2015-01-01

    Diarrhea, hyperglycemia, anemia, depigmentation of the hair, and rash are common side effects of tyrosine kinase inhibitors. Neurological side effect like hallucination due to pazopanib is exceptionally rare in literature cases. Herein, we reported a case of hallucination related to pazopanib in a patient with renal cell carcinoma. A 47-year-old male patient with renal cell carcinoma developed repetitive hallucinations on the following days of pazopanib initiation. There was no other significant finding in the differential diagnosis of hallucination. Neurological symptoms disappeared after termination of pazopanib. We aimed to emphasize that neurological side effect like hallucination may rarely occur during the treatment of pazopanib and take note that physicians should be aware of this infrequent side effect in the patients treated with pazopanib.

  19. 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.

  20. 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.

  1. Prune Belly syndrome: A rare case report

    OpenAIRE

    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 megauret...

  2. Duodenal lymphangitis carcinomatosa: A rare case

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    Ilanchezhian Subramanian, MBBS, MD

    2014-01-01

    Full Text Available Duodenal lymphangitis carcinomatosa has been sporadically described, and little attention has been paid so far. To our knowledge, no data on radiological findings for this rare entity has been published. We report a case of duodenal lymphangitis carcinomatosa secondary to gallbladder mass in a 44-year-old Indian man to focus on the radiological diagnosis, which was further confirmed by endoscopic-guided biopsy and immunohistochemical analysis.

  3. Albright hereditary osteodystrophy: A rare case report

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    Goswami M

    2009-09-01

    Full Text Available Albright hereditary osteodystrophy (AHO is a rare hereditary metabolic disorder that may be associated with or without resistant to parathyroid hormone (pseudohypoparathyroidism. It is commonly characterized by a constellation of physical features of short stature, round face, short neck, and small metacarpals and metatarsals, mild mental retardation, osteoporosis, subcutaneous calcification, and sometimes olfactory and hearing functional defect. Hypocalcaemia and hyperphosphatemia are the most important manifestations of the case. We report a clinical case of siblings with AHO with reduced Gs-alpha activity and we discuss their clinical features with oral manifestations, radiographic findings, laboratory tests along with treatment.

  4. A Rare Cause of Lower Extremity Ulcers: Prolidase Deficiency.

    Science.gov (United States)

    Adışen, Esra; Erduran, Funda Bapur; Ezgü, Fatih Süheyl; Kasapkara, Çiğdem Seher; Besio, Roberta; Forlino, Antonella; Gürer, Mehmet Ali

    2016-03-01

    Prolidase deficiency is an autosomal recessive disorder, which is associated with chronic skin ulcers, a characteristic facial appearance, mental retardation, and recurrent infections. This study describes 4 patients with recurrent leg ulcerations and abnormal facies who were first clinically suspected of prolidase deficiency and then biochemically confirmed. Two siblings and 2 other patients were admitted to our clinic at different times, and they had some common features such as chronic leg and foot ulcers recalcitrant to treatment, consanguineous parents, facial dysmorphism, mental retardation, and widespread telangiectasias. Physical examination and detection of low prolidase level in blood finally led us to the diagnose of ulcers secondary to prolidase deficiency. Prolidase deficiency is a rare genodermatosis and must be considered in the differential diagnosis of recurrent leg and foot ulcers that develop at an early age. © The Author(s) 2015.

  5. 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.

  6. 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.

  7. A rare case of malignant hydrocele in a young patient.

    Science.gov (United States)

    de Lima, George Rafael Martins; de Oliveira, Vladmir Pinheiro; Reis, Paulo Henrique de Moura; Pinheiro, Fábio Gurgel do Amaral; Lima, Marcos Venício Alves; Gonzaga-Silva, Lúcio Flávio

    2009-06-01

    Malignant mesotheliomas of the tunica vaginalis are extremely rare tumors, especially in young patients. We report a case of a 15-year old patient presenting with clinical signs of hydrocele. Preoperative ultrasound scanning revealed two papillary lesions. Initially, before mesothelioma was suspected, the patient was submitted to focal resection of the macroscopic lesions. Following diagnosis by histopathology, the patient was submitted to radical orchiectomy and hemiscrotectomy. The patient presents no signs of local or systemic recurrence at the time of writing. A review of the literature on the subject has been included.

  8. Nonexpansile Unicystic Ameloblastoma: A Rare Case Report

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    Supreet Jain

    2017-01-01

    Full Text Available Ameloblastoma is the most common benign odontogenic tumor that has the potential to grow into a large size. Unicystic ameloblastoma (UA is a variant of the solid or multicystic ameloblastoma. It is a rare (relative frequency 5–22% benign, locally invasive odontogenic tumor of young age that mimics clinically and radiographically as an odontogenic cyst. Radiographically it may present as unilocualr or multilocular with cortical plate expansion. Conventional radiograph has less advantage to determine their extension into soft tissue. Hence, conventional tomography, cone-beam computed tomography, and magnetic resonance imaging may be used to determine expansion, root resorption and relation with adjacent structures. It has low recurrence rate after conservative therapy. Here, we present a rare case of nonexpansile UA with an unusual presentation in a young male patient.

  9. 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.

  10. 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.

  11. Surgical approach to a rare case of double-outlet right ventricle and aortopulmonary window.

    Science.gov (United States)

    Tunks, Robert D; Steed, R Dennis; Lodge, Andrew J

    2016-01-01

    Although the spectrum of physiology seen in infants and children with double-outlet right ventricle is broad, this anatomy in combination with an aortopulmonary window is extremely rare. We present an interesting case of an infant prenatally diagnosed with this rare CHD. To our knowledge, this is the first report of complete repair in the neonatal period for such a combination of defects.

  12. Metastatic Basal Cell Adenocarcinoma of Submandibular Gland to the Spine: An Extremely Rare Condition.

    Science.gov (United States)

    Pluemvitayaporn, Tinnakorn; Kunakornsawat, Sombat; Piyaskulkaew, Chaiwat; Pruttikul, Pritsanai; Pongpinyopap, Warongporn

    2017-12-01

    Basal cell adenocarcinomas are rare malignant neoplasms of salivary glands, accounting for gland tumors. Few cases of distant metastases have been reported. A 50-year-old Thai man was diagnosed with basal cell adenocarcinoma of the submandibular gland with pulmonary and cervical spine metastases with progressive myelopathy. He was treated with wide surgical resection of the soft tissue tumor and modified radical neck dissection, anterior cervical total corpectomy with fusion combined with posterior decompression and fusion of the cervical spine, and surgical wound coverage by anterolateral thigh free tissue transfer, followed by adjuvant radiotherapy. At 18-month follow-up, the patient remained in good condition, and no signs of local recurrence or contiguous spreading were detected. Postoperative radiographs showed solid osseous fusion without loss of correction or implant failure. This case highlighted an extremely rare condition of metastatic basal cell adenocarcinoma of the submandibular gland to the lung and spine, which, to our knowledge, has not been previously reported in the literature. Copyright © 2017 Elsevier Inc. All rights reserved.

  13. 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.

  14. A rare case of dens invaginatus in a mandibular canine.

    Science.gov (United States)

    George, Roy; Moule, Alexander J; Walsh, Laurence J

    2010-08-01

    Dens invaginatus (dens in dente) is a common dental anomaly with a reported prevalence of between 0.04% and 10%. It typically affects permanent maxillary lateral incisors, central incisors and premolars. These developmental lesions are less common in mandibular teeth and are extremely rare in canines and molars. This report describes a rare case of dens invaginatus (Oehlers type II) in a permanent mandibular canine. The tooth was mature with a closed apex and showed apical pathosis. The tooth was treated endodontically using a non-surgical technique with hand endodontic files, and then followed up after a period of 8 months. A follow-up radiograph showed some healing of the lesion.

  15. 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.

  16. Nonsyandromic oligodontia: A rare case report

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    Mayank Chaturvedi

    2015-01-01

    Full Text Available Dental agenesis is the most common developmental anomaly in humans, which can be either in the form of anodontia, oligodontia, or hypodontia. Oligodontia can occur either as an isolated finding or as part of a syndrome. Nonsyndromic oligodontia is rare. Management requires integrated multidisciplinary approach. Prompt intervention improves quality of life. Here, we report a case of isolated, nonsyndromic oligodontia in a 13-year-old female who allegedly had complete set of primary teeth but failed to develop complete permanent dentition. No other clinical feature, except oligodontia, was present that could suggest any syndromic association.

  17. 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.

  18. Three rare cases of cutaneous phaeohyphomycosis

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    Aditi Chhonkar

    2016-01-01

    Full Text Available Phaeohyphomycosis is a chronic infectious condition caused by dematiaceous fungi which usually involve the skin and subcutaneous tissue. Subcutaneous phaeohyphomycosis is characterised by papulonodules, verrucous, hyperkeratotic or ulcerated plaques, cysts, abscesses, pyogranuloma, non-healing ulcers or sinuses. In India, commonly associated genera are Exophiala, Phialophora, Cladosporium, Curvularia, Fonsecaea and Alternaria. This condition involves the presence of brown-walled hyphal structures in the dermis and epidermis. Here, we are reporting a rare case series of three patients of phaeohyphomycosis with lesions on finger and dorsum of the hand.

  19. Extremely rare cause of congenital diarrhea: enteric anendocrinosis.

    Science.gov (United States)

    Sayar, Ersin; Islek, Ali; Yilmaz, Aygen; Akcam, Mustafa; Flanagan, Sarah E; Artan, Reha

    2013-10-01

    Congenital diarrheal disorders consist of a variety of chronic enteropathies. There are approximately 30 different diseases that can be classified into four groups according to the mechanisms involved in pathogenesis: (i) absorption and transport of nutrients and electrolytes; (ii) enterocyte differentiation and polarization; (iii) enteroendocrine cell differentiation; and (iv) modulation of the intestinal immune response. Affected patients often present with life-threatening diarrhea, in the first few weeks of life. A new disorder, enteric anendocrinosis, which is characterized by severe malabsorptive diarrhea and a lack of intestinal enteroendocrine cells has recently been described in six patients with recessively inherited mutations in the Neurogenin-3 gene. In this report we describe a seventh case with a review of the literature. © 2013 The Authors. Pediatrics International © 2013 Japan Pediatric Society.

  20. 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.

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

    Science.gov (United States)

    Sharma, Gaurav; Mutneja, Anudeep R; Nagpal, Archna; Mutneja, Puneet

    2015-01-01

    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.

  2. 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.

  3. Extreme oncoplastic breast surgery: A case report

    OpenAIRE

    Bordoni, Daniele; Cadenelli, Pierfrancesco; Falco, Giuseppe; Rocco, Nicola; Manna, Paola; Tessone, Ariel; Ornelli, Matteo; Magalotti, Cesare

    2016-01-01

    Introduction: So called “extreme oncoplastic surgery” is emerging as a new promising concept in breast cancer surgery allowing successful breast conservation in selected patients with multicentric tumors. Presentation of case: We report the case of a 48-year-old woman presenting with a multicentric breast cancer and successfully treated with an oncoplastic technique consisting in three radical lumpectomies followed by breast reshaping and simultaneous contralateral symmetrization. Discu...

  4. Rare imaging of a known entity: fat embolism seen on CT in lower extremity vein after trauma

    Directory of Open Access Journals (Sweden)

    Varun Chowdhary, MD

    2017-09-01

    Full Text Available Fat embolism occurs in the vast majority of patients who have had trauma (approximately 90%. The most common occurrence is after long bone fracture. It has also been noted in cases after orthopedic surgery. Fat embolism is most often diagnosed when the clinical manifestations of fat embolism syndrome become apparent. Reported cases of fat emboli in transit are unusual. In our case, we present the rare finding of fat embolism seen on computed tomography in the lower extremity after a trauma.

  5. Case Report: A Rare Case Report of Spontaneous Resolution of ...

    African Journals Online (AJOL)

    Hepatic portal venous gas (HPVG) is a rare disease entity. It is an ominous finding, usually, associated with intraabdominal ischemic necrosis. It frequently requires emergent surgical intervention. Herein, we present a case of a patient who complained of intense abdominal pain, with radiographic findings of HPVG as a ...

  6. Permanent Maxillary Canine Agenesis: A Rare Case Report.

    Science.gov (United States)

    Kambalimath, Halaswamy V; Jain, Somya; Patil, Raju Umaji; Asokan, Alexander; Kambalimath, Deepashri

    2015-01-01

    Congenitally missing teeth (CMT) are among one of the commonly known dental anomalies. The most frequently missing teeth in the permanent dentition, excluding the third molars, are mandibular second premolars and maxillary lateral incisors. Exclusive agenesis of both maxillary canines is an extremely rare occurrence and only a few cases have been reported. Previous studies showed that the prevalence of maxillary canine agenesis varies between 0.07 and 0.13%. In recent studies on Indian population, no cases of maxillary canine agenesis have been documented. This paper reports a case of non-syndromic bilateral agenesis of permanent maxillary canines, along with agenesis of both mandibular central incisors in a healthy 13-year-old Indian female patient; and a brief literature review on prevalence, etiology and treatment modalities of the condition. How to cite this article: Kambalimath HV, Jain S, Patil RU, Asokan A, Kambalimath D. Permanent Maxillary Canine Agenesis: A Rare Case Report. Int J Clin Pediatr Dent 2015; 8(3):242-246.

  7. 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.

  8. 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.

  9. A rare case of rynopharyngeal melanoma

    Directory of Open Access Journals (Sweden)

    Francesco Grecchi

    2012-01-01

    Full Text Available Primary mucosal melanomas (MM of the head and neck region constitute 0.5-2% of all malignant melanomas. The rynopharynx is a region that is less often involved by malignant melanomas. Because most of mucosal melanotic lesions are painless in their early stages, the diagnosis is unfortunately often delayed until symptoms resulting from ulceration, growth, and/or bleeding are noted. Here, we document the rare case of a malignant rynopharynx melanoma of a 43 year old woman. Its treatment and the pertinent literature are discussed. No complication was recorded in the post-operative period and no further surgery was performed. The follow up showed no recurrence in the same position and with the same characteristics, even after six years. Mucosal melanomas are aggressive tumours and the prognosis in these patients is poor. Clinicians must use treatment strategies that provide functional benefit, so as to maintain quality of life without excessive toxicity.

  10. A rare case of Moebius sequence

    Directory of Open Access Journals (Sweden)

    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.

  11. 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.

  12. Rare Case of Methemoglobinemia Complicating Pregnancy

    Directory of Open Access Journals (Sweden)

    S. Verma

    2016-01-01

    Full Text Available A patient at 38 weeks of gestation when taken for emergency cesarean section had her oxygen saturation of 66 to 70% (by saturation probe on monitor in operation theatre. She was otherwise asymptomatic but her oxygen saturation was persistently low on the monitors. Her arterial blood gas analysis showed all parameters to be normal. Her electrocardiography was normal. Her surgery was imperative but due to her reduced oxygen saturation she became a high-risk case. In presence of senior consultants of anesthesia and gynecology and under high-risk consent she had an uneventful cesarean delivery. Physician and cardiologist opinions were sought thereafter. The outcomes and the results of our efforts to find the etiology of her reduced saturation on monitors despite being clinically asymptomatic lead to the disclosure of this rare hemoglobinopathy. Mother and baby had uneventful course after delivery and were discharged well.

  13. Osteosarcoma of breast: A rare case of extraskeletal osteosarcoma

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    Ashwin A Kallianpur

    2013-01-01

    Full Text Available Primary osteogenic sarcomas of the breast are exceptionally uncommon. We describe such a case occurring in a 50 year-old woman who presented with a large painful mass in her left breast. Simple mastectomy of the left breast was performed. Microscopical and immunohistochemical findings established the diagnosis of primary osteogenic sarcoma. Similar to extremity osteosarcoma, adjuvant adriamycin and cisplatin based chemotherapy and external beam radiotherapy was given to the present case. She remained well 57 months later, without tumor recurrence. The current article made a literature search proving the rarity of this lesion type and discusses in detail the diagnostic implications and the treatment of this rare site tumor entity.

  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. A rare case of primary amenorrhea

    Directory of Open Access Journals (Sweden)

    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.

  16. 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.

  17. 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.)

  18. Gingival complex odontoma: a rare case report with a review of the literature

    Science.gov (United States)

    Araujo, Juliane Pirágine; Gallo, Camila de Barros; Trierveiler, Marília

    2018-01-01

    Odontomas are odontogenic tumors composed of a mixture of dental tissues. They are very common hamartomas of the jaws. However, their peripheral or gingival counterparts are extremely uncommon. The objective of this article is to report a rare case of gingival complex odontoma in an 11-year-old patient, and also to review all published cases of this type of lesion. PMID:29515984

  19. 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.)

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

    Directory of Open Access Journals (Sweden)

    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.

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

    Directory of Open Access Journals (Sweden)

    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.

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

    Directory of Open Access Journals (Sweden)

    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

  3. Bilateral Maxillary Paramolars and Endodontic Therapy: A Rare Case Report

    Science.gov (United States)

    Parolia, A.; Kundabala, M.

    2010-01-01

    Supernumerary paramolars are a rare anomaly of the maxillofacial complex. They are more common in the maxilla than the mandible. This article reports a rare case of bilateral maxillary paramolars, their complications and management. PMID:21998783

  4. 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

  5. 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.

  6. 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.

  7. 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

  8. Unilateral Aplasia of Mandibular Condyle: A Rare Case Entity

    Directory of Open Access Journals (Sweden)

    Anil G Ghom

    2011-01-01

    Full Text Available Aplasia of the mandibular condyle is extremely rare when not seen in association with or as a part of any syndrome. The incidence is estimated to be 1 in 5.600 births. Growth disturbances in the development of the mandibular condyle may occur in utero late in the first trimester and may result in disorders, such as aplasia or hypoplasia of the mandibular condyle. We report a case of aplasia of left mandibular condyle along with hypoplasia of right condylar head in a 20-year-old female patent. The patient reported to the clinic with the complaint of proclined upper front teeth, wanting to improve her esthetics. Clinical, conventional radiography and computer tomographic studies revealed the complete absence of condyle on the left and hypoplasia of the head of mandibular condyle on the right side. The etiology was unknown and on the basis of history, clinical study and radiological examination it was suggested to be of developmental origin.

  9. 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.

  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 Mesenteric Gastrointestinal Stromal Tumor ...

    African Journals Online (AJOL)

    Gastrointestinal stromal tumours (GIST) are rare tumours arising from mesenchyme of gastrointestinal tract and overexpress C-kit protein. Mainly seen in stomach and small bowel. Mesenteric GIST are rarely reported as they constitute less than 1% of total GIST. We here report such a rare case of GIST arising from ...

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

    OpenAIRE

    Shahshahani, Hayedeh Javadzadeh; Vahidfar, Mohamad Reza; Khodaie, Seyed Ali

    2013-01-01

    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 ...

  13. 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.

  14. A RARE CASE REPORT OF A LARGE PLACENTAL CHORIOANGIOMA

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    Alaka Banerjee

    2017-02-01

    Full Text Available BACKGROUND Chorioangioma is a relatively common benign tumour of placenta arising from chorionic tissue. The incidence of it is very rare around 0.6%. These often remain unnoticed as they are rarely more than 5 cm. CASE REPORT We describe a case with large chorioangioma of placenta that had a poor pregnancy outcome. CONCLUSION Chorioangioma of placenta is a rare problem associated with potentially serious foetal risk, hence constant surveillance during pregnancy is required.

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

    Directory of Open Access Journals (Sweden)

    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.

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

    Science.gov (United States)

    Balaji, Preetha; Balaji, S M

    2017-01-01

    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.

  17. A Rare Case of Jejunal Diverticulosis Causing Mesenteric Volvulus

    OpenAIRE

    Reddi, Bhavani Rao; Konkena, Janardhan Rao; Bogarapu, Chaitanya Babu; Kollu, Srinu Babu; Neelam, Prasad

    2015-01-01

    Jejunal diverticulosis is a rare asymptomatic entity [1]. In literature, there are only few cases of jejunal diverticulosis where small gut volvulus is reported. The disease entity is important as it may masquerade as hemorrhage, obstruction, or perforation which are life threatening. We report a rare case of small bowel volvulus secondary to jejunal diverticulosis.

  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. 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 ...

  20. 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 ...

  1. 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

  2. Histoid leprosy: a rare exuberant case*

    Science.gov (United States)

    de Andrade, Pedro Jose Secchin; Messias, Sulamita dos Santos Nascimento Dutra; Ferreira, Paola Cristina Brandão Oliveira; Sales, Anna Maria; Machado, Alice de Miranda; Nery, José Augusto da Costa

    2015-01-01

    Leprosy is a neglected disease. We point up the need of recognizing the unusual clinical presentations of the disease in order to make early diagnosis and proper treatment possible, and break the transmission chain. The authors report a rare type of multibacillary leprosy: histoid leprosy and present images of numerous well-circumscribed indurated papules and nodules distributed throughout the entire body. PMID:26560226

  3. Histoid leprosy: a rare exuberant case*

    OpenAIRE

    de Andrade, Pedro Jose Secchin; Messias, Sulamita dos Santos Nascimento Dutra; Ferreira, Paola Cristina Brand?o Oliveira; Sales, Anna Maria; Machado, Alice de Miranda; Nery, Jos? Augusto da Costa

    2015-01-01

    AbstractLeprosy is a neglected disease. We point up the need of recognizing the unusual clinical presentations of the disease in order to make early diagnosis and proper treatment possible, and break the transmission chain. The authors report a rare type of multibacillary leprosy: histoid leprosy and present images of numerous well-circumscribed indurated papules and nodules distributed throughout the entire body.

  4. Talon cusps in mandibular incisors: Report of eight rare cases

    Directory of Open Access Journals (Sweden)

    Shiu-yin Cho

    2014-01-01

    Full Text Available Talon cusps in mandibular anterior teeth are very rare. Talon cusps in mandibular anterior teeth associated with other anomalies are even rarer and that a bilateral case in the mandible has not been reported before. In this report, eight such rare cases of talon cusps in permanent mandibular incisors are presented. It includes a bilateral case that in the author′s knowledge is the first case reported in the English literatures.

  5. Extreme oncoplastic breast surgery: A case report.

    Science.gov (United States)

    Bordoni, Daniele; Cadenelli, Pierfrancesco; Falco, Giuseppe; Rocco, Nicola; Manna, Paola; Tessone, Ariel; Ornelli, Matteo; Magalotti, Cesare

    2016-01-01

    So called "extreme oncoplastic surgery" is emerging as a new promising concept in breast cancer surgery allowing successful breast conservation in selected patients with multicentric tumors. We report the case of a 48-year-old woman presenting with a multicentric breast cancer and successfully treated with an oncoplastic technique consisting in three radical lumpectomies followed by breast reshaping and simultaneous contralateral symmetrization. According to our experience, oncoplastic conserving breast surgery could represent a better option than the combination of mastectomy, reconstruction and radiation therapy, in terms of quality of life for selected patients affected by multicentric breast cancer. The surgical treatment for multicentric breast cancers remains controversial even though emerging evidences show good oncological and aesthetic outcomes following oncoplastic conserving breast surgery. Copyright © 2016 The Authors. Published by Elsevier Ltd.. All rights reserved.

  6. Intralobar pulmonary sequestration in elderly woman: a rare case report with emphasis on imaging findings

    Directory of Open Access Journals (Sweden)

    Qays Ahmed Hassan Al-Timimy, MBCHB, DMRD, CABMS (RAD

    2016-09-01

    Full Text Available Intralobar pulmonary sequestration is a rare malformation that predisposes to recurrent respiratory infections. It is difficult to diagnose unless a more extensive directed investigation (to the vasculature and pulmonary parenchyma is take on. Failure to diagnose and treat this condition can lead to recurrent pneumonia and fatal hemoptysis. Most cases are diagnosed before the age of 20 years. In this report, we present an extremely rare case of elderly woman with initial diagnosis of intralobar sequestration, and to our knowledge, this case represents the oldest diagnosed patient in the literature.

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

    Directory of Open Access Journals (Sweden)

    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.

  8. Revisiting Cementoblastoma with a Rare Case Presentation

    Directory of Open Access Journals (Sweden)

    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.

  9. 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.

  10. 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.

  11. A rare case of abnormal uterine bleeding caused by cavernous hemangioma: a case report

    Directory of Open Access Journals (Sweden)

    Telesinghe PU

    2010-05-01

    Full Text Available Abstract Introduction Cavernous hemangiomas of the uterus are extremely rare, benign lesions. A survey of the current literature identified fewer than 50 cases of hemangioma of the uterus. Case presentation We report a case of cavernous hemangioma of the uterus in a 27-year-old Malay, para 1 woman who presented at our hospital with torrential vaginal bleeding having been transferred by land ambulance from a district hospital 30 minutes away. 11 weeks previously she had an urgent cesarean section at our hospital. She had to undergo a hysterectomy to control her bleeding after other measures were unsuccessful. A histopathological report confirmed a diffuse ramifying hemangioma of the cervix and uterus with left hematosalpinx. Conclusion Most ramifying hemangioma lesions are asymptomatic and are found incidentally, but sometimes they may cause abnormal vaginal bleeding and hence should be included in the differential diagnosis of patients with vaginal bleeding. Hysterectomy is the primary mode of treatment in most symptomatic cases.

  12. Primary mediastinal synovial sarcoma: A rare case report.

    Science.gov (United States)

    Ershadi, Reza; Rahim, Mohamadbagher; Davari, Hamidreza

    2016-01-01

    Synovial sarcomas commonly occur in the extremities of young adults. A primary occurrence in the mediastinum is very rare with only a few reported cases in the world literature. We report a case of mediastinal synovial sarcoma. This paper is about a 47-year-old male who presented with retrosternal chest pain and shortness of breath on exertion. Imaging showed an anterior mediastinal mass. Pathological examination of the resected mass showed a biphasic neoplasm with a spindle cell component admixed with gland-like elements. The tumor showed positive staining with cytokeratin, epithelial membrane antigen and vimentin confirming the diagnosis of a biphasic synovial sarcoma. A wide range of neoplasms, both primary and metastatic, occur in the mediastinum, which pose considerable diagnostic difficulties. A synovial sarcoma should always be considered in the differential diagnosis, and immunohistochemistry is an important adjuvant tool in this situation. This paper highlights the importance of recognizing an unusual presentation of this aggressive neoplasm to aid appropriate clinical management. Copyright © 2016 The Author(s). Published by Elsevier Ltd.. All rights reserved.

  13. 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.

  14. A rare case of metastatic basal cell carcinoma

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    Dai Wee Lee

    2017-12-01

    Full Text Available Background: Basal cell carcinoma (BCC is relatively uncommon in Malaysia with non-melanomatous skin cancer being the 12th most common malignancy in Malaysia.Metastatic basal cell carcinoma (MBCC is extremely rare and the incidence is estimated to be 0.0028% to 0.55% among all BCC. We report a case of MBCC with a summary of clinical and histopathology findings; and the management. Case presentation: A 69-year-old Chinese man, presented with a left wrist mass for 4 years. The mass was on the dorsal surface of the wrist, ulcerated, and measured 15 x 10cm. The tumour size was 2.5 × 8.6 × 10cm on MRI, extending into the extensor digitorum tendons and possible invasion into the lumbricals. He underwent wide excision of the mass, decorticotomy of left 3rd to 5th metacarpal bones and amputation of 2nd metacarpal bone. Histopathological examination showed BCC involving whole skin thickness invading the subcutaneous fat but sparing the underlying tissues, resections margins were clear, maximal diameter was 11.5 cm. The tumour was staged as pT2N0M0. After 18 months, he developed an 8 × 10cm left axillary mass which was fungating and bleeding. CT scan also showed multiple lung metastases. Repeat biopsy of the left axillary mass showed BCC with similar appearance as the previous histopathology examination. Radiotherapy to the left axilla was given (65 Gray in 30 fractions for local control as the tumour was bleeding profusely. Discussion: A review by Wysong et al. for 194 cases of MBCC suggests that its prognosis remains poor over the past 30 years, with the median overall survival of 10 months from diagnosis despite the introduction of systemic chemotherapy and radiotherapy. Most common metastatic sites are the regional lymph nodes, lungs and bone. Large lesions (particularly those over 10 cm2 and tumours that invade deep structures, such as cartilage, skeletal muscle, or bone, are most likely to metastasize. Perineural invasion, aggressive histologic

  15. Norwegian scabies - rare case of atypical manifestation.

    Science.gov (United States)

    Ebrahim, Karina Corrêa; Alves, Júlia Barazetti; Tomé, Lísias de Araújo; Moraes, Carlos Floriano de; Gaspar, Arianne Ditzel; Franck, Karin Fernanda; Hussein, Mohamad Ali; Cruz, Lucas Raiser da; Ebrahim, Leonardo Duque; Sidney, Luis Felipe de Oliveira

    2016-01-01

    Human scabies affects all social classes and different races around the world. It is highly contagious, but the exact figures on its prevalence are unknown. A 19-year-old male patient was admitted to the emergency room reporting fever (38°C) and multiple lesions throughout the body, except face, soles, and palms. Lesions were non-pruritic, which hampered the initial diagnostic suspicion. Skin biopsy was performed, and the final diagnosis was crusted scabies (Norwegian). It was concluded that human scabies is a significant epidemic disease, due to its different clinical manifestations, and because it is extremely contagious.

  16. 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.

  17. 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.

  18. 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…

  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 abnormal uterine bleeding caused by cavernous hemangioma: a case report.

    Science.gov (United States)

    Benjamin, Mridula A; Yaakub, Hjh Roselina; Telesinghe, Pu; Kafeel, Gazala

    2010-05-17

    Cavernous hemangiomas of the uterus are extremely rare, benign lesions. A survey of the current literature identified fewer than 50 cases of hemangioma of the uterus. We report a case of cavernous hemangioma of the uterus in a 27-year-old Malay, para 1 woman who presented at our hospital with torrential vaginal bleeding having been transferred by land ambulance from a district hospital 30 minutes away. 11 weeks previously she had an urgent cesarean section at our hospital. She had to undergo a hysterectomy to control her bleeding after other measures were unsuccessful. A histopathological report confirmed a diffuse ramifying hemangioma of the cervix and uterus with left hematosalpinx. Most ramifying hemangioma lesions are asymptomatic and are found incidentally, but sometimes they may cause abnormal vaginal bleeding and hence should be included in the differential diagnosis of patients with vaginal bleeding. Hysterectomy is the primary mode of treatment in most symptomatic cases.

  1. 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

  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. Paraphilic infantilism. A rare case of fetishistic behaviour.

    Science.gov (United States)

    Pandita-Gunawardena, R

    1990-11-01

    A rare case of infantile behaviour of a fetishistic nature in an 80-year-old man was possibly related to a head injury at the age of six, when his regression first began. Treatment was deemed unwarranted.

  5. A Rare Case of Central Nervous System Tuberculosis

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    Ravish Parekh

    2014-01-01

    Full Text Available Intracranial abscess is an extremely rare form of central nervous system (CNS tuberculosis (TB. We describe a case of central nervous system tuberculous abscess in absence of human immunodeficiency virus (HIV infection. A 82-year-old Middle Eastern male from Yemen was initially brought to the emergency room due to altered mental status and acute renal failure. Cross-sectional imaging revealed multiple ring enhancing lesions located in the left cerebellum and in bilateral frontal lobe as well as in the inferior parietal lobe on the left. The patient was placed on an empiric antibiotic regimen. Preliminary testing for infectious causes was negative. Chest radiography and CT of chest showed no positive findings. He was not on any immunosuppressive medications and human immunodeficiency virus (HIV enzyme immunoassay (EIA test was negative. A subsequent MRI one month later showed profound worsening of the lesions with increasing vasogenic edema and newly found mass effect impinging on the fourth ventricle. Brain biopsy showed focal exudative cerebellitis and inflamed granulation tissue consistent with formation of abscesses. The diagnosis of CNS TB was finally confirmed by positive acid-fast bacilli (AFB cultures. The patient was started on standard tuberculosis therapy but expired due to renal failure and cardiac arrest.

  6. Rare case of metaplastic breast cancer in a man.

    Science.gov (United States)

    Katz, Heather; Jafri, Hassaan; Dougherty, Thomas; Lebowicz, Yehuda

    2018-03-07

    Metaplastic breast cancer (MBC) in men is an extremely rare entity. MBC is typically very aggressive with a poor prognosis. In men, it has only been reported three times in the literature. We report a 47-year-old man who presented with right-sided breast erythema and nipple inversion. Mammogram revealed a 2.4 cm spiculated mass. Initial pathology was inconclusive; however, right-sided simple mastectomy showed invasive metaplastic carcinoma with adenosquamous histology. He received adjuvant chemotherapy with 4 cycles of dose dense Adriamycin and cyclophosphamide followed by 12 weeks of paclitaxel and chest wall radiation. Although oestrogen receptor status was 1%, tamoxifen was not given due to recent diagnosis of pulmonary embolism. Two years after treatment, he is currently living with no signs of recurrence. This case will serve as a useful addition to the current literature discussing successful diagnosis, treatment and prognosis of a man with MBC. © BMJ Publishing Group Ltd (unless otherwise stated in the text of the article) 2018. All rights reserved. No commercial use is permitted unless otherwise expressly granted.

  7. The Inside Mystery of Jejunal Gastrointestinal Stromal Tumor: A Rare Case Report and Review of the Literature

    OpenAIRE

    Dhull, A. K.; Kaushal, V.; Dhankhar, R.; Atri, R.; Singh, H.; Marwah, N.

    2011-01-01

    Gastrointestinal stromal tumors (GISTs) are malignant and rare form of soft tissue sarcoma of the digestive tract. The incidence of gastrointestinal stromal tumors is very low Kramer et al. 2005 Jejunal GISTs are extremely rare. Here we present a rare case of jejunal GIST with unusually large size at presentation. The patient presented with severe abdomen pain, exophytic growth, and dimorphic anemia. Surgical resection of the tumor was carried out, and operative findings revealed a 15 × 10 cm...

  8. Fracture of the proximal extremity of the tibia after anterior cruciate ligament reconstruction: case report

    Directory of Open Access Journals (Sweden)

    Márcio de Oliveira Carneiro

    2015-06-01

    Full Text Available We report a rare condition that has been little described in the literature: a fracture of the proximal extremity of the tibia after anterior cruciate ligament reconstruction using an autologous patellar bone-tendon graft. In this report, we discuss the factors that predisposed toward this episode, the treatment and the evolution of the case after the surgical treatment.

  9. Osteoblastoma of the scaphoid bone – A rare case report

    Directory of Open Access Journals (Sweden)

    Khodamorad Jamshidi

    2009-04-01

    Full Text Available

    • Osteoblastoma is rarely found in the hand or wrist. Review of literature  evealed that only five cases of osteoblastoma have been reported in scaphoid bone. This paper presents a rare case of scaphoid steoblastoma with unusual course ina young patient and its treatment process.
    • KEYWORDS: Hand, osteoblastoma, scaphoid, wrist.

  10. Carcinosarcoma of ovary with its various immunohistochemical expression: Report of a rare case

    Directory of Open Access Journals (Sweden)

    Senjuti Dasgupta

    2015-01-01

    Full Text Available Ovarian carcinosarcoma is an extremely rare tumor with an incidence of <2%. A report of such a rare case in a 40--year-old multiparous woman is being presented here. The patient complained of abdominal pain and distension. Abdominal computed tomography revealed a variegated mass in the right adnexal region. The CA-125 level was 1635 U/ml. The patient underwent laparotomy and the tumor was removed. Microscopic examination of the tumor showed presence of both carcinomatous and sarcomatous elements. Immunohistochemical analysis revealed that the carcinomatous component was positive for cytokeratin and estrogen receptor and negative for progesterone receptor. The sarcomatous component was positive for vimentin. Ki-67 was positive in 60% cells. A final diagnosis of ovarian carcinosarcoma was rendered. This case is significant owing to extreme rarity of the tumor. Records of similar cases must be maintained for future reference with regard to impact of treatment protocol followed on prognosis.

  11. A Rare Case of Aplasia Cutis Congenita

    Directory of Open Access Journals (Sweden)

    Sumita Mehta

    2017-10-01

    Full Text Available Aplasia cutis congenita is a heterogenous group of disorders characterized by the absence of a portion of skin either localized or widespread at birth. Most commonly seen on the scalp (84%, it can affect any part of the body, including the trunk and limbs. We report a case of a baby born with aplasia cutis congenital managed conservatively.

  12. A Rare Case of Popliteal Venous Aneurysm

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    Roberto Fiori

    2010-01-01

    aneurysm was identified. In the same time open tangential aneurysmectomy and lateral vein reconstruction were realised. This case is interesting because the Angio Computed Tomography study, in delayed acquisition, has allowed a correct diagnostic assessment of PVA and the surgical treatment.

  13. 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.

  14. 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.

  15. Unusual Gingival Enlargement: A Rare Case Report

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    Ashutosh Dixit

    2014-01-01

    Full Text Available This is an atypical case report of a 20-year-old male patient who suffered from unusual unilateral, gingival enlargement together with rapidly progressive alveolar bone loss. The enlarged gingiva completely covered his left posterior teeth in both arches. The patient was diagnosed with gingival fibromatosis and aggressive periodontitis based on the clinical, histological, and radiographic findings. The gingival enlargement was treated by conventional gingivectomy under local anaesthesia. The postoperative result was uneventful.

  16. 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.

  17. Gingival cyst of adult: A rare case

    OpenAIRE

    Malali, Vijayalaxmi V.; Satisha, T. S.; Jha, A. K.; Rath, S. K.

    2012-01-01

    Gingival cyst of adult is an uncommon cyst of gingival soft tissue occurring in either the free or attached gingiva. This odontogenic epithelial cyst is most frequently seen near mandibular canine and premolar region, believed to represent the soft tissue counter part of the lateral periodontal cyst. This article presents a case of gingival cyst treated with exicisional biopsy followed by histopathological confirmation and an emphasis on the clinical aspects of this lesion.

  18. 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.

  19. Scrotal abscess, a rare case of extra intestinal amoebiasis.

    Science.gov (United States)

    Prasetyo, R H

    2015-09-01

    The majority of amoeba infection are asymptomatic, but clinically intestinal amoebiasis or extra intestinal amoebiasis may result. Genital amoebiasis is very rare manifestation of extra intestial amoebiasis, but a case of amoebic scrotal abscess, seen in Surabaya. The invasive form of Entamoeba histolytica trophozoite was seen in Giemsa stained aspirate of the abscess. In case of an abscess bacteria are primarly considered, but the case presented here shows that amoeba can be the cause, although very rarely. Thus when bacteriological diagnostics are negative amoeba should be considered, especially in case of brown-reddish colored and foul smelling pus.

  20. Paraneoplastic edematous dermatomyositis: A rare syndrome observed in a case of small cell lung cancer

    Directory of Open Access Journals (Sweden)

    Kevin Zarrabi

    2017-10-01

    Full Text Available Dermatomyositis with subcutaneous edema is a rare process with few reported cases. We report a 63-year-old with lung cancer who presented with an erythematous skin rash and was found to have biopsyproven dermatomyositis. Her course was complicated by generalized edema, myalgias, muscle weakness, dysphagia, and laryngeal edema. The edema was severe and caused respiratory distress requiring intubation. The patient underwent therapy with high-dose glucocorticoids and intravenous immunoglobulin but failed treatment. Altogether, she presented as an extreme case and rare variant of dermatomyositis, known as edematous dermatomyositis. Diagnostic and treatment guidelines do not account for this variant and literature pertaining to edematous dermatomyositis is sparse. Moreover, this disease was a paraneoplastic manifestation of her small cell lung cancer, which is rarely observed. There are no cases reporting edematous dermatomyositis as a paraneoplastic manifestation of small cell lung cancer, and we highlight the high rate of morbidity and mortality in such patients.

  1. A RARE CASE OF CAVERNOUS HAEMANGIOMA FACE

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    Tarun Guha

    2017-12-01

    Full Text Available PRESENTATION OF CASE A 60-year-old male presented to ENT OPD with a swelling on the left side of face for past 30 years. This was insidious in onset, slowly progressive, not associated with pain, visual disturbance, bleeding from the swelling, facial deformity or impairment of hearing. He had a small ulcer over the swelling for past 2 months and hunchback for past 20 years. He had no history of diabetes mellitus, hypertension, tuberculosis, bronchial asthma, radiation exposure, trauma, surgery in the past or any other intervention.

  2. Poland syndrome: rare presentation in two cases.

    Science.gov (United States)

    Gocmen, Hayrettin; Akkas, Yucel; Doganay, Selim

    2010-08-27

    Poland syndrome was first described in 1840 by Alfred Poland while still a medical student and the other components of the syndrome were described at London Guy's Hospital following the dissection of a cadaver's hand, which had hypoplasia and syndactyly. The incidence of Poland syndrome has been reported to be 1 in 30,000 live births. In the present case report, two Poland syndrome patients with ipsilateral hypomastia and a reduction in the axillary/pectoral hairs diagnosed during adulthood are presented; one patient was affected on the left side and had widespread cafe au lait spots, and the other patient had respiratory dysfunction due to multiple rib anomalies.

  3. A RARE CASE OF THORACIC ACTINOMYCOSIS

    Directory of Open Access Journals (Sweden)

    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

  4. Ascher's syndrome: A rare case report

    Science.gov (United States)

    Chandravanshi, Shivcharan Lal; Mishra, Vinay

    2015-01-01

    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. PMID:25971175

  5. 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.

  6. 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...

  7. 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.

  8. 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.

  9. 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.

  10. 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.

  11. 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.

  12. Kluth type IV3 membranous esophageal atresia at middle one-third of esophagus: An extremely rare entity

    Directory of Open Access Journals (Sweden)

    Rahul Gupta

    2017-01-01

    Full Text Available We herein present an extremely rare case of an isolated membranous atresia causing near-complete obstruction of the esophagus. The neonate presented with drooling of saliva and frothing from the mouth. A red rubber catheter met with an obstruction at 12 cm from the gum margins. Radiograph showed paucity of gas in the abdomen. Thoracotomy revealed external continuity of esophagus; dilated and elongated proximal segment constituting upper half of esophagus, and a membrane with tiny opening in the center, at its junction with narrow distal segment. Excision was performed. A high index of suspicion for membrane should be kept in dealing with esophageal obstruction beyond 10 cm from the gum margins, particularly those with paucity of abdominal gas pattern or gasless abdomen.

  13. Candidiasis, A Rare Cause of Gastric Perforation: A Case Report ...

    African Journals Online (AJOL)

    Fungi are unusually rare causes of gastric perforation, with most cases of gastric perforation occurring as complications of peptic ulcer disease (PUD), nonsteroidal anti‑inflammatory drugs (NSAIDs) and gastric neoplasms. Here, we report the case of a 70‑year‑old Nigerian male who presented with severe epigastric pain, ...

  14. 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 ...

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

    African Journals Online (AJOL)

    Arun Kumar Agnihotri

    ABSTRACT: 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 ...

  16. 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 ...

  17. Case Report:Triceps Tendon Avulsion: A Rare Injury | Sharma ...

    African Journals Online (AJOL)

    Background: Triceps tendon avulsion is one of the rare tendinous injuries. Such injuries can easily be missed, and should be kept as a differential diagnosis in all patients who present with pain and swelling at the back of the elbow after a traumatic event. Case Details: We present a case of triceps tendon avulsion which ...

  18. Rare case of urinary tract infections and the "forgotten" solution

    NARCIS (Netherlands)

    Ten Donkelaar, Celine S.|info:eu-repo/dai/nl/413646343; Wortel, Ruud C.; Lock, M. T W T|info:eu-repo/dai/nl/148539769

    2015-01-01

    This report presents a rare case of recurrent urinary tract infections in a kidney transplant patient. Analysis revealed a ureteral stump calculus 31 years after bilateral nephrectomy which was disintegrated by means of flexible ureteroscopy and holmium laser. In case of unexplained recurrent

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

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    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.

  20. 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

  1. Transitional Cell Carcinoma of Kidney- Report of a Rare Case

    Directory of Open Access Journals (Sweden)

    Priyesh Halgaonkar

    2015-01-01

    Full Text Available Hematuria is a common presentation in the surgical outpatient department. The most common causes being urinary tract infection or renal calculi that causes hematuria. Few of them are being diagnosed as Renal or Bladder mass. Transitional cell carcinoma affecting urogenital tract accounts for 5-10% of the primary renal malignancies which is relatively rare. Here we report such rare case in an elderly female who presented with painless hematuria.

  2. Benign cementoblastoma: A rare case report with review of literature

    Directory of Open Access Journals (Sweden)

    Neeta Sharma

    2014-01-01

    Full Text Available We present a rare case of radiopaque lesion with radiolucent rim in the right body of mandible, with history of extraction of involved tooth, which made diagnosis confusing unless pre-extraction intraoral periapical radiography radiograph was recovered, finally diagnosed as benign cementoblastoma. It was surgically managed, with no recurrence of the lesion more than 2 years of follow-up. Benign cementoblastoma is a rare, benign tumor of odontogenic ectomesenchymal origin, usually associated with roots of first mandibular molar.

  3. A RARE CASE OF CONGENITAL ABNORMALITIES OF THE MAXILLOFACIAL REGION

    Directory of Open Access Journals (Sweden)

    Юлия Владимировна Степанова

    2014-09-01

    Full Text Available This article describes a rare case of congenital abnormality: a median facial cleft in combination with congenital complete bilateral cleft lip and palate. An integrated approach to the treatment of children with severe pathology of the maxillofacial area provides a good aesthetic and functional result. Surgical treatment in combination with orthodontic treatment, massage, myogymnastics, dynamic observation of the child contributes to the normal development of the maxillofacial area in a child with a rare congenital facial cleft.

  4. Asymptomatic Osteoblastoma of the Mandible: A Rare Case Report

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    Soudabeh Sargolzaei

    2018-06-01

    Full Text Available Osteoblastoma is a rare solitary osteoblastic bone neoplasm. It is characterized by proliferation of osteoblasts forming trabeculae within a vascular fibrous stroma. There is a variety of jaw bone lesions with very close clinical, radiological and microscopic interrelations, which make diagnosis more challenging. Familiarity with these rare bony lesions is vital for oral pathologists. This report presents a new case of asymptomatic mandibular osteoblastoma occurring in a 43-year-old male

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

    OpenAIRE

    Vanga, Rohini R.; Bal, Bikram; Olden, Kevin W.

    2013-01-01

    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...

  6. HUGE SYNOVIAL SARCOMA ARISING FROM CHEST WALL: A RARE CASE REPORT

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    Waddi Sudhakar

    2015-02-01

    Full Text Available Synovial sarcomas are the fourth most common malignant soft - tissue tumors, and typically develop in para - articular locations of the extremities. However, the occurrence of these tumors in the chest wall is rare. In this article, we report the interesting case of a 27 - year - old male with spindle cell variant of synovial sarcoma arising in the anterior chest wall with a brief review of the literature. KEYWORDS:Synovial sarcoma;chest wall;spindle cell variant

  7. 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 ...

  8. Deadliest tumor of oral cavity: A rare case of intra oral malignant melanoma

    Directory of Open Access Journals (Sweden)

    Nunsavathu Purnachandrarao Naik

    2014-01-01

    Full Text Available Malignant melanoma is a rare tumor arising from the uncontrolled growth of melanocytes found in the basal layer of the epithelium. Although cutaneous malignant melanoma is the third most common malignancy of the skin, it accounts only for 3-5% of all dermal malignancies. Primary malignant melanoma of the oral mucosa is extremely rare, accounting for only 0.2-8% of all malignant melanomas. The incidence is slightly higher in males while others report a higher incidence in females. In the oral cavity, 80% of the cases occur in the maxilla with the prevalence of the hard palate or combined with the gingival or alveolar ridge. In this paper, we present a rare case of a 47-year-old female patient who reported with completely asymptomatic, pigmented growth in the maxillary anterior region.

  9. Radicular Dens Invaginatus: Report of a Rare Case

    Science.gov (United States)

    Beena, V. T.; Sivakumar, R.; Heera, R.; Rajeev, R.; Choudhary, Kanaram; Panda, Swagatika

    2012-01-01

    Dens invaginatus is a developmental anomaly resulting from invagination of a portion of crown forming within the enamel organ during odontogenesis. The invagination ranges from a slight pitting (coronal type) to an anomaly occupying most of the crown and root (radicular type). Although a clinical examination may reveal a deep fissure or pit on the surface of an anterior tooth, radiographic examination is the most realistic way to diagnose the invagination. The objective of this case presentation is to report a rare case of radicular dens in dente, which is a rare dental anomaly. PMID:22900211

  10. Radicular Dens Invaginatus: Report of a Rare Case

    Directory of Open Access Journals (Sweden)

    V. T. Beena

    2012-01-01

    Full Text Available Dens invaginatus is a developmental anomaly resulting from invagination of a portion of crown forming within the enamel organ during odontogenesis. The invagination ranges from a slight pitting (coronal type to an anomaly occupying most of the crown and root (radicular type. Although a clinical examination may reveal a deep fissure or pit on the surface of an anterior tooth, radiographic examination is the most realistic way to diagnose the invagination. The objective of this case presentation is to report a rare case of radicular dens in dente, which is a rare dental anomaly.

  11. CASE REPORT Radiographic diagnosis of a rare case of ...

    African Journals Online (AJOL)

    ODDD is a rare autosomal dominant congenital disorder mainly affecting the development of the face, eyes, skeletal system, heart and dentition. ODDD has been mapped to chromosome 6q22-q24 and germline mutations have been identified in the connexin 43 gene, GJA1. To date, over 70 reports in the literature describe ...

  12. 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.

  13. 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.

  14. ACCESSORY CYSTIC DUCT IDENTIFIED IN LAPAROSCOPIC CHOLECYSTECTOMY: A RARE CASE

    OpenAIRE

    Kumar; Kumar; Shaleen; Wasif; Amit

    2014-01-01

    We report the case of a 53 year old lady who underwent laparoscopic cholecystectomy and was found to have an accessory cystic duct close to the fundus. Careful dissection of the liver bed was done and the duct clipped preventing a bile leak. The presence of such ducts though rare should be identified during surgery to prevent potential complications.

  15. Primary neuroendocrine carcinoma of breast: a rare case report ...

    African Journals Online (AJOL)

    Primary neuroendocrine carcinoma (PNEC) of breast was an unknown pathologic entity till recently due its rare incidence and lack of definitive criteria for diagnosis. We present a case of PNEC of breast in a middle aged lady. A 34 years lady presented with a breast lump since 1 month, who underwent modified radical ...

  16. Hepatic pulmonary fusion: A rare case | Taide | African Journal of ...

    African Journals Online (AJOL)

    This paper describes a rare case of a seven-monthold infant with right-sided congenital diaphragmatic hernia, who was found to have fusion of his herniated part of liver with lower lobe of right lung. The report describes clinical presentation and successful surgical management along with a review of the world literature.

  17. 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.

  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. Candidiasis, A Rare Cause of Gastric Perforation: A Case Report ...

    African Journals Online (AJOL)

    Candidiasis, A Rare Cause of Gastric Perforation: A Case Report and Review of Literature. Ukekwe FI, Nwajiobi C1, Agbo MO2, Ebede SO3, Eni AO. Department of Morbid Anatomy, University of Nigeria, Enugu Campus, Enugu/University of Nigeria Teaching Hospital. Ituku‑Ozalla, 1Division of General Surgery, Department ...

  20. 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

  1. Case Study: Telangiectatic osteosarcoma, a rare complication of ...

    African Journals Online (AJOL)

    Case Study: Telangiectatic osteosarcoma, a rare complication of Paget's disease of bone. FH Van Zyl, M Conradie, K Barnard, FS Hough, BH Ascott-Evans. Abstract. No Abstract. Full Text: EMAIL FREE FULL TEXT EMAIL FREE FULL TEXT · DOWNLOAD FULL TEXT DOWNLOAD FULL TEXT.

  2. Hypomelanosis of Ito: Case report of a rare neurocutaneous ...

    African Journals Online (AJOL)

    Hypomelanosis of Ito (HI) though said to be the third most common neurocutaneus disorder, is rarely reported in paediatric practice in Africa. A high index of suspicion must be maintained in children with cutaneous lesions as a seizure may be the first symptom that may bring the child to attention. A case of HI in a neonate is ...

  3. Primary Renal Synovial Sarcoma: A Rare Case Report

    Directory of Open Access Journals (Sweden)

    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. A Rare Case of Synovial Sarcoma of the Prostate

    African Journals Online (AJOL)

    Department of Urology, KEM Hospital, Mumbai, India. AbStRACt. Prostatic synovial sarcomas are exceedingly rare. To our knowledge, only six primary cases have been reported so far. We herein describe a primary synovial sarcoma of the prostate seen in a 25- year-old male patient, the youngest patient seen with this ...

  5. A Rare Case of Male‐to‐Eunuch Gender Dysphoria

    Directory of Open Access Journals (Sweden)

    Maria Hermann, MD

    2015-12-01

    Conclusion: Healthcare professionals must be aware of this group of patients as they have a greater risk of self‐harm in order to self‐castrate, which could be acute and life threatening if not performed in the right way. Hermann M and Thorstenson A. A rare case of male‐to‐eunuch gender dysphoria. Sex Med 2015;3:331–333.

  6. 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 ...

  7. 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 ...

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

    Directory of Open Access Journals (Sweden)

    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.

  9. 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

  10. A Rare Case of Mesenteric Gastrointestinal Stromal Tumor ...

    African Journals Online (AJOL)

    We here report such a rare case of GIST arising from mesentery of small bowel and presenting as acute abdomen. Good surgical clearance ensures good survival whereas incomplete resection results in a high incidence of recurrences with distant metastasis. Keywords: Gastrointestinal stromal tumors, imatinib, mesenteric ...

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

    African Journals Online (AJOL)

    2011-07-29

    Jul 29, 2011 ... Nigerian Journal of Clinical Practice • Jul-Sep 2012 • Vol 15 • Issue 3. Case Report. Abstract. Lymphadenopathy is a rare mode of presentation of cysticercus infestation. Hence, in endemic areas, cysticercosis must be included in the differential diagnosis of superficial palpable swellings in the neck region.

  12. Bilateral breast fibromatosis: a rare case report | Fewou | Revue de ...

    African Journals Online (AJOL)

    Introduction: Breast fibromatosis is a rare benign tumor of soft tissues with local spread, often frequently recurrent but never results to metastasis. Methodology: We present a case diagnosed in a 41 years old patient who was referred to a Douala private medical center in February 2012 presenting bilateral breast tumors.

  13. A rare case report: mucopoly-saccharidosis (hurler syndrome) with ...

    African Journals Online (AJOL)

    A rare case report: mucopoly-saccharidosis (hurler syndrome) with rachitic changes in a Nigerian. Egbuna Obidike, Ngozi R Njeze, AC Ude, GO Ossi. Abstract. No Abstract. West African Journal of Radiology Vol. 11(1) 2004: 14-18. Full Text: EMAIL FULL TEXT EMAIL FULL TEXT · DOWNLOAD FULL TEXT DOWNLOAD ...

  14. 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 ...

  15. Tree Species Traits but Not Diversity Mitigate Stem Breakage in a Subtropical Forest following a Rare and Extreme Ice Storm

    OpenAIRE

    Nadrowski, Karin; Pietsch, Katherina; Baruffol, Martin; Both, Sabine; Gutknecht, Jessica; Bruelheide, Helge; Heklau, Heike; Kahl, Anja; Kahl, Tiemo; Niklaus, Pascal; Kröber, Wenzel; Liu, Xiaojuan; Mi, Xiangcheng; Michalski, Stefan; von Oheimb, Goddert

    2014-01-01

    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 o...

  16. Aortic Endograft Infection by Pasteurella multocida: A Rare Case.

    Science.gov (United States)

    Jayakrishnan, Thejus T; Keyashian, Brian; Amene, Juliet; Malinowski, Michael

    2016-08-01

    Infection of an aortic endograft is a rare complication following endovascular aneurysm repair. These patients have been treated with explantation of the graft to obtain source control followed by an extra-anatomic bypass to restore circulation. The present case study describes an interesting case of Pasteurella infection involving an aortic endograft managed nonoperatively by percutaneous drainage and graft preservation. © The Author(s) 2016.

  17. A Rare Case of Inguinal Hernia with Complete Bladder Herniation

    Directory of Open Access Journals (Sweden)

    Ayaaz Habib

    2017-01-01

    Full Text Available Involvement of the bladder in inguinal hernias is rare and occurs in less than 5% of the cases. The diagnosis and management of this condition may present a challenge to the surgeon. We present a case of an elderly gentleman who presented with a large left-sided inguinoscrotal hernia causing an obstructive uropathy which was surgically repaired. The patient made a quick postoperative recovery with complete resolution of renal function.

  18. A Rare Case of Inguinal Hernia with Complete Bladder Herniation

    OpenAIRE

    Habib, Ayaaz

    2017-01-01

    Involvement of the bladder in inguinal hernias is rare and occurs in less than 5% of the cases. The diagnosis and management of this condition may present a challenge to the surgeon. We present a case of an elderly gentleman who presented with a large left-sided inguinoscrotal hernia causing an obstructive uropathy which was surgically repaired. The patient made a quick postoperative recovery with complete resolution of renal function.

  19. 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...

  20. 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...

  1. A Rare Case of Lipomatous Hypertrophy of the Interventricular Septum

    OpenAIRE

    Papadopoulos, Christodoulos E.; Matsiras, Sotirios; Vassilikos, Vassilios

    2016-01-01

    Asymmetrical left ventricular hypertrophy secondary to interventricular septum hypertrophy is usually considered a typical phenotype of hypertrophic cardiomyopathy. In rare cases other conditions such as tumors or lipomatous hypertrophy of the interventricular septum may have a similar presentation. We present a case of a male patient who presented for routine cardiology work up and was diagnosed of having ventricular septal hypertrophy secondary to localized lipomatous hypertrophy.

  2. Pan-brachial plexus neuropraxia following lightning: A rare case report.

    Science.gov (United States)

    Patnaik, Ashis; Mahapatra, Ashok Kumar; Jha, Menka

    2015-01-01

    Neurological complications following lightning are rare and occur in form of temporary neurological deficits of central origin. Involvement of peripheral nervous system is extremely rare and only a few cases have been described in the literature. Isolated unilateral pan-brachial plexus neuropraxia has never been reported in the literature. Steroids have long been used for treatment of neuropraxia. However, their use in lightning neural injury is unique and requires special mention. We report a rare case of lightning-induced unilateral complete flaccid paralysis along with sensory loss in a young patient. Lightning typically causes central nervous involvement in various types of motor and sensory deficit. Surprisingly, the nerve conduction study showed the involvement of peripheral nervous system involvement. Steroids were administered and there was significant improvement in neurological functions within a short span of days. Patients' functions in the affected limb were normal in one month. Our case was interesting since it is the first such case in the literature where lightning has caused such a rare instance of unilateral pan-brachial plexus lesion. Such cases when seen, raises the possibility of more common central nervous system pathology rather than peripheral involvement. However, such lesions can be purely benign forms of peripheral nerve neuropraxia, which can be managed by steroid treatment without leaving any long-term neurological deficits.

  3. Structural safety in case of extreme actions

    DEFF Research Database (Denmark)

    Giuliani, Luisa

    2012-01-01

    The behaviour of buildings and other constructions under critical events is widely recognised to be an important part of structural design. Still, the problem is often addressed in a qualitative way by many current codes and regulations and designers cannot refer to specific methodologies...... for the assessment or the achievement of the structural integrity of constructions. The term structural integrity is intended here as the ability of the structure to sustain extreme actions that directly affects its system without developing a major collapse. In this paper two main methods are outlined....... The use of the first approach is proposed for assessing, by means of a set of nonlinear static analyses, the robustness of structural systems, intended as the ability of a structure to sustain local failure (Starossek, 2009) without developing a major collapse....

  4. 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.)

  5. Extensive squamous metaplasia in a benign phyllodes tumor: A rare case report

    Directory of Open Access Journals (Sweden)

    Harsh Kumar

    2015-01-01

    Full Text Available Phyllodes tumors (PTs with extensive squamous metaplasia is an uncommon biphasic fibroepithelial neoplasms of breast, comprising of <1% of all breast neoplasm′s. In this article, we report a case of a 55-year-old female patient admitted to the General Surgery Department with a rapidly enlarging, palpable mass in right breast. After histopathological examination, it was diagnosed to be as benign PT with extensive squamous metaplasia. Metaplastic changes are known, but infrequent in these tumors. Extensive squamous metaplasia within PT is rare and may occur in benign, borderline and malignant subtypes. There are only a few cases reported in the literature. We therefore, aimed to present this case in view of its extremely rare nature.

  6. 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.

  7. 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.

  8. Multiple intracranial cavernous angiomas: A rare case series

    Directory of Open Access Journals (Sweden)

    Pradeepgoud H Patil

    2012-01-01

    Full Text Available Cavernous angiomas are cerebral cavernous malformations and they are relatively rare lesions. Two forms of cavernous angiomas have been described: a sporadic form, in which patients usually have a single lesion, and a familial form, the hallmarks of which are multiple lesions and autosomal dominant transmission. The familial form appears to be very uncommon and has mainly been described in the Hispanic population. We report two cases of multiple intracranial cavernous angiomas which is an autosomal dominant pattern of inheritance. It is very rare to find this in non Hispanic population.

  9. Acoustic Effects Accurately Predict an Extreme Case of Biological Morphology

    Science.gov (United States)

    Zhang, Zhiwei; Truong, Son Nguyen; Müller, Rolf

    2009-07-01

    The biosonar system of bats utilizes physical baffle shapes around the sites of ultrasound emission for diffraction-based beam forming. Among these shapes, some extreme cases have evolved that include a long noseleaf protrusion (sella) in a species of horseshoe bat. We have evaluated the acoustic cost function associated with sella length with a computational physics approach and found that the extreme length can be predicted accurately from a fiducial point on this function. This suggests that some extreme cases of biological morphology can be explained from their physical function alone.

  10. 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.

  11. 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.

  12. 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.

  13. A child presented with bilateral congenital constriction ring in lower extremity: a case report.

    Science.gov (United States)

    Jaiman, Richa; Gangopadhyay, Ajay N; Gupta, Dinesh K; Srivastava, Punit; Upadhyaya, Vijay D; Sharma, Shiv P; Kumar, Vijayendra

    2009-07-23

    The congenital constriction ring of lower extremity is very uncommon and rare condition. The actual incidence in general population is not known. In English literature, very few cases are reported time to time as congenital constriction band syndrome associated with musculoskeletal disorder like congenital talipes equino varus. The lesion can involve skin only or goes to deeper structure up to bone, which can lead to gangrene of foot or auto amputation. We are presenting a case of bilateral congenital constriction ring in lower limb who presented at age of 4 year without any other associated congenital anomaly, simply managed by Z-plasty, which improves quality of life after physiotherapy. Congenital constriction ring of lower limb is extremely rare condition in children. Early diagnosis and management is mandatory, either in single stage or by stage procedure, to prevent auto-amputation of limb and to improve quality of life on feet.

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

    Directory of Open Access Journals (Sweden)

    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.

  15. 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.

  16. Eccrine porocarcinoma of the lower extremity: A case report and review of literature

    Directory of Open Access Journals (Sweden)

    Asarian Armand

    2011-08-01

    Full Text Available Abstract Eccrine porocarcinoma is a rare malignancy of the eccrine sweat gland. It is usually found frequently on the lower extremities, and it affects both sexes equally usually in the sixth to seventh decade. In our case, we present a 42-year-old male patient with a recurring exophytic tumor on the right lower extremity without local extension. The initial tumor was biopsied, excised and diagnosed as an eccrine poroma. The tumor then recurred 6 years later, was re-excised, reconstructed with a soleus muscle flap and diagnosed as an eccrine porocarcinoma.

  17. Managing a Rare Malignant Sweat Gland Tumor Invading the Brain: Case Report and Literature Review.

    Science.gov (United States)

    Jagannatha, Aniruddha Tekkatte; Khan, Mansoor A; Karanth, Shrithi; Srikantha, Umesh; Varma, Ravi; Mahadevan, Anita

    2016-02-01

    Malignant sweat gland adnexal tumors are rare with an incidence of 0.001%. Of these, clear cell hidradenocarcinoma is an extremely uncommon subtype that accounts for 6% of malignant eccrine sweat gland tumors. They occur commonly in the head, neck, and extremities. Although they have a propensity for local recurrence, intracranial extension with brain invasion is extremely rare. We report a 76-year-old man with a large, recurring, ulcerated, fungating scalp swelling of 14 years who presented with focal seizures and drowsiness. Neuroimaging revealed a massive tumor arising from the scalp to invade the left parietal lobe and extending to the right side with occlusion of the superior sagittal sinus. The overlying parietal bone was lytic with a "moth-eaten" appearance. He underwent wide excision of the scalp lesion, near-total cerebral tumor decompression followed by titanium mesh cranioplasty, rotation flap reconstruction of the scalp, and adjuvant radiotherapy to the skull vault. Histopathology revealed clear cell hidradenocarcinoma. Whole-body positron emission tomography scan did not reveal any other lesion. At 24 months' follow-up, he remains recurrence free. We report a rare indolent case of clear cell hidradenocarcinoma invading the brain, which was managed with near-total decompression and adjuvant radiotherapy. Intracranial extension in such aggressive tumors poses challenges in management, and regular neuroimaging surveillance is advised. Copyright © 2016 Elsevier Inc. All rights reserved.

  18. A Rare Case of Primary Tubular Adenocarcinoma of the Thymus, Enteric Immunophenotype: A Case Study and Review of the Literature

    Directory of Open Access Journals (Sweden)

    Hae Yoen Jung

    2015-07-01

    Full Text Available Thymic carcinomas are uncommon malignant tumors, and thymic adenocarcinomas are extremely rare. Here, we describe a case of primary thymic adenocarcinoma in a 59-year-old woman. Histological examination of the tumor revealed tubular morphology with expression of cytokeratin 20 and caudal-type homeobox 2 according to immunohistochemistry, suggesting enteric features. Extensive clinical and radiological studies excluded the possibility of an extrathymic primary tumor. A review of the literature revealed only two global cases of primary tubular adenocarcinomas of the thymus with enteric immunophenotype.

  19. 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.

  20. 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.

  1. 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.

  2. Congenital pseudoarthrosis of tibia: A rare case report

    Directory of Open Access Journals (Sweden)

    Sonal Balwantsingh Thakur

    2015-01-01

    Full Text Available Congenital pseudoarthrosis (CPT of the tibia is rare congenital anomaly occurring 1 in 190,000 live births. It is one of the rare causes of limb shortening. A 8-year-old male child born of consanguious marriage came to orthopedic OutPatient Department with history of significant shortening of left leg since birth so much, so that patient cannot use his left leg for walking. His right leg was normal. Family history of neurofibromatosis-I (NF-1 is present. History, photographs and X-rays were taken and patient was diagnosed as a case of CPT of the tibia. Significant association between CPT of tibia and NF-I has been found. The case is of significance to orthopedicians and pediatrician from the management point of view.

  3. 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.

  4. Pheochromocytoma: clinical review based on a rare case in adolescence.

    Science.gov (United States)

    Alface, Marisa Maibel; Moniz, Patricia; Jesus, Susana; Fonseca, Cândida

    2015-08-04

    Pheochromocytomas are rare tumours originating in chromaffin cells, representing 0.1-1% of all secondary hypertension (HT) cases. The majority are benign and unilateral, characterised by the production of catecholamines and other neuropeptides. Mainly located in the adrenal gland, they are more frequent between the 3rd and 5th decades of life; however, 10-25% can be associated with genetic familial syndromes (multiple endocrine neoplasia type 2 (MEN 2), type 1 neurofibromatosis and Von-Hippel-Landau disease in younger ages. The authors present a rare case of secondary HT due to a pheochromocytoma in a 15-year-old patient, whose metanephrine assay confirmed the diagnosis, and abdominal ultrasound and CT localised the tumour in the adrenal gland. HT was controlled with α and β blockers, with posterior retroperitoneal laparoscopic surgical intervention and subsequent resolution of HT. Age and concomitant hyperparathyroidism compelled genetic testing for the exclusion of MEN 2, which was negative. 2015 BMJ Publishing Group Ltd.

  5. 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.

  6. Double coin in esophagus at same location and same alignment - a rare occurrence: a case report.

    Science.gov (United States)

    Upadhyaya, Eti V; Srivastava, Punit; Upadhyaya, Vijay D; Gangopadhyay, An; Sharma, Sp; Gupta, Dk; Hassan, Zaheer

    2009-07-16

    Coin is the most common foreign body swallowed by pediatric age group. The multiple coin swallowing is extremely rare and very few cases had been reported in English literature. Most of them were present at different site and had different alignment in the esophagus. The location of the coin (trachea vs. esophagus) is commonly determined by the alignment of the coin on radiographic studies. A 4-year-girl was presented to us with history of coin ingestions one day back without any respiratory distress. On radiological study there was suspicion of two coins on same location and alignment. The diagnosis was confirmed after removal. The both coin was removed successfully by esophagoscopy. Unexpected second foreign bodies in pediatric esophageal coin ingestions are rare and it is mandatory to do post operative radiography after removal to exclude duplex coin or tracheal coin. We are presenting this case because of its rarity, difficulty in diagnosis especially when proper history is not available.

  7. Sclerosing thymoma: A rare case report and brief review of literature.

    Science.gov (United States)

    Li, Xin; Wang, Meng; Sun, Daqiang

    2018-04-01

    Sclerosing thymoma is an extremely rare mediastinal neoplasm; it was recognized for the first time in 1994 and to date only 15 cases have been reported. The present study report a case of a 65-year-old man who was incidentally found to have an anterior mediastinal nodule, without clinical symptoms including fever, chest pain, and myasthenia gravis. The chest computed tomography (CT) revealed the nodule was 4.9 × 4.2 × 3.0 cm in size. And the microscopic and immunohistochemical findings indicated that the final diagnosis was sclerosing thymoma. The anterior mediastinal nodule was completely removed. No evidence of recurrence or complication was found in the second year after surgery. The biologic behavior of the rare sclerosing thymoma is still largely mysterious; it is utmost importance to classify the sclerosing thymoma from other mediastinal tumors. Its prognosis is favorable and thymectomy is currently the mainstay of treatment.

  8. A RARE CASE OF EARLY URETHRAL OBSTRUCTION SEQUENCE

    OpenAIRE

    Gaurav Garg; S. Krishna; Dhiraj Shedabale; K.C. Jain

    2009-01-01

    Early urethral obstruction is most commonly the consequence of urethral valve formation during the development of prostatic urethra. Less commonly, it is due to urethral atresia, bladder neck obstructionor distal urethral obstruction. A rare case delivered at Pravara Rural Hospital ,Loni with bilateral hydronephrosis with imperforated anus, undescended testis and unilateral Congenital Talipes EquinoVarus. Child underwent Exploratory Laparotomy with descending loop colostomy and bilateral neph...

  9. A rare case report: Carcinoma pancreas with hepatocellular carcinoma

    Directory of Open Access Journals (Sweden)

    Vikas Yadav

    2014-01-01

    Full Text Available Synchronous double malignancies involving different organs are relatively rare and uncommon finding. We report an interesting case of double malignancy in which a patient exhibited synchronous two separate carcinomas, pancreatic and hepatocellular carcinoma (HCC. Patient was a 64-year-old male who presented primarily with symptoms pertaining to the biliary obstruction and ultrasound of abdomen revealing pancreatic head mass. HCC was detected incidentally during the investigations for carcinoma pancreas.

  10. Odontogenic Myxoma of the Maxilla- A Rare case Report

    OpenAIRE

    Subramaiam, Ramkumar; Narashiman, Sangeetha; Narasimhan, Malathi; Giri, Veda; Kumar, Santhosh

    2015-01-01

    Odontogenic myxoma (OM) is an uncommon, benign, locally invasive, non-metastasizing neoplasm arising from the odontogenic ectomesenchyme that usually occurs in the tooth bearing areas of the jaws. These lesions arouse special interest as they pose high diagnostic challenge. Here, we present a rare case of OM of the maxilla in an 18-year-old male. The clinical, radiographic and histopathological features of the lesion are discussed in this paper.

  11. A Rare Case of Adult Medulloblastoma with Spinal Metastasis

    Directory of Open Access Journals (Sweden)

    K. Quenum

    2012-01-01

    Full Text Available Medulloblastoma is a relatively common malignant brain tumor of childhood and relatively rare in adulthood, with a propensity for neuraxial spread via cerebrospinal fluid pathways. Osseous extraneural metastasis is uncommon and when it happens, radiologic findings are of sclerotic (60%, lytic (35%, and mixed patterns (5% (Algra et al. (1992. In this paper, we present a case of medulloblastoma metastiaszing to the lumbar spine and describe the magnetic resonance appearance, with emphasis on the image findings mimicking spondylodiscitis.

  12. A rare case of Aneurysmal Bone Cyst of the Talus

    Directory of Open Access Journals (Sweden)

    Sandesh S. Madi

    2014-08-01

    Full Text Available Talus bone is an unusual location for Aneurysmal Bone Cysts. Although considered benign, it has a locally aggressive behavior making it difficult both from diagnostic and therapeutic point of view. It becomes even more challenging when unusual sites are encountered. We report a rare and challenging case of Aneurysmal Bone Cyst in Talus in a young male patient who was treated with curettage and bone grafting. [Cukurova Med J 2014; 39(4.000: 932-937

  13. [Type III Monteggia lesion: a rare association, about a case].

    Science.gov (United States)

    Oukhouya, Mohamed Amine; El Jaoud, Hind Abou; Andaloussi, Saad; Abdellaoui, Hicham; Atarraf, Karima; Chater, Lamia; Afifi, My Abderrahman

    2017-01-01

    Type III Monteggia lesion is very rare, usually occurring within a context of violent trauma and often going unnoticed. We report the case of a 11-year old boy presenting to the Emergency Department with blunt trauma of the upper limb. The radiological evaluation showed olecranon fracture and radial epiphyseal separation associated with dislocation of the radial head. The patient underwent orthopedic treatment with good outcome after a mean follow-up of 3 months.

  14. Rare case of generalised aggressive periodontitis in the primary dentition

    OpenAIRE

    Spoerri, A; Signorelli, C; Erb, J; van Waes, H; Schmidlin, P R

    2014-01-01

    BACKGROUND Generalised aggressive periodontitis (AP) in the prepubescent age is an exceptionally rare disease in the primary dentition of otherwise healthy children. Characteristics of AP are gingival inflammation, deep periodontal pockets, bone loss, tooth mobility and even tooth loss. The most common way of treating this disease is the extraction of all the involved primary teeth. CASE REPORT A 4-year-old girl presented with signs of severe gingival inflammation. Clinical examination rev...

  15. A Rare Case of Spontaneous Empyema by Clostridium perfringens

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    Sijan Basnet

    2018-01-01

    Full Text Available Empyema caused by clostridial infections is rare especially in the absence of invasive thoracic procedures. We present the case of an 81-year-old man without a history of preceding trauma who presented with shortness of breath and nonproductive cough and grew Clostridium perfringens only in the pleural cavity. He was predisposed to the infection due to his swallowing dysfunction. He was treated with penicillin and chest tube placement for drainage and subsequently improved.

  16. 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.)

  17. 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 ...

  18. 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.

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

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    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.

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

    Science.gov (United States)

    Kukreja, Rahul; Suma, Gundareddy N.; Yadav, Bhawna; Sharma, Havi

    2015-01-01

    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. PMID:25793183

  1. 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.

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

    International Nuclear Information System (INIS)

    Bagewadi, Shivanand B.; Kukreja, Rahul; Suma, Gundareddy N.; Yadav, Bhawn; Sharma, Havi

    2015-01-01

    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.

  3. 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.

  4. 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.

  5. A Rare Case Report: Multiple Perforations Due To Diverticulitis Focus

    Directory of Open Access Journals (Sweden)

    Berke Manoglu

    2014-04-01

    Full Text Available Diverticulitis, in 10-25% of patients with diverticular disease is one of the most frequent complications. Only 5-10% of patients with diverticulitis attack %u2018is under the age of 50. However, this is more aggressive in patients with recurrent attacks of diverticulitis are more common and urgent surgical treatment has been shown that more is needed. . In patients with diverticulitis; patients receiving inpatient treatment, clinical, laboratory and radiological methods to be followed and if necessary to decide the appropriate surgical treatment is very important. In this case, we will present depending on the attack of diverticulitis with multipel perforation focus a rare case.

  6. A RARE CASE OF THIAZIDE-INDUCED BENIGN GYNAECOMASTIA

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    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

  7. A Rare Case of Pneumoureter: Emphysematous Pyelitis versus Emphysematous Pyelonephritis

    Science.gov (United States)

    Mahesh; Padala, Krishna Prasanthi; Swamy, Kumar; Pailoor, Aruna

    2015-01-01

    Emphysematous pyelitis is a rare benign entity which is defined as isolated gas production in the pelvicalyceal system, ureters or in the urinary bladder as a consequence of acute bacterial renal infection. In this case report we present a case with no prior medical history of diabetes or urinary obstruction who presented with uncontrolled hyperglycaemia for the first time associated with emphysematous pyelonephritis. Further, the imaging differences between emphysematous pyelonephritis (EPN) and emphysematous pyelitis (EP) has been discussed along with the approach and their management. PMID:26676155

  8. Gnathostomiasis: A rare case of cutaneous creeping eruptions

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    Vaibhavi Subhedar

    2014-01-01

    Full Text Available A 21-year-old vegetarian lady presenting with migratory erythema and pain in her right hand subsequently developed a blister on the palm. A worm-like structure came out of the wound and was identified as Gnathostoma spp. adult male worm. It is a very rare cause of creeping eruptions and so far only one case of cutaneous gnathostomiasis caused by a larva has been reported from India. This is the first case of cutaneous gnathostomiasis due to an adult Gnathostoma spp. in India.

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

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    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.

  10. 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.

  11. 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

  12. 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.

  13. A rare case of Fusobacterium necrophorum liver abscesses

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    Faris Hannoodi

    2017-01-01

    Full Text Available Liver abscesses are an uncommon disease that can present with vague symptoms. Fusobacterium necrophorum causing liver abscesses is a rare condition and only a few cases have been reported. An 88-year-old female presented to her primary care physician with one week of fevers, night sweats, chills, fatigue and vague right upper quadrant abdominal pain. She denied nausea, vomiting, constipation, diarrhea and unintentional weight loss. A computed tomography scan of the abdomen showed two liver abscesses in the right lobe as well as extensive diverticulosis. Percutaneous drainage was performed and draining catheters were placed in the abscesses. Culture of the abscess fluid grew Fusobacterium necrophorum. She was treated with ceftriaxone and metronidazole as per sensitivities. Rare cases of F. necrophorum hepatic abscesses have been published. The source of infection described in reported cases included hematogenous spread from dental caries/peritonsillar abscess and those involving the gastrointestinal tract resulting from inflammation of the bowel wall or from inflamed diverticuli via the portal circulation. In one study, thirteen cases of liver abscess due to F. necrophorum were studied, and two of these cases had diverticular disease without inflammation.

  14. 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.

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

    Energy Technology Data Exchange (ETDEWEB)

    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).

  16. 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.

  17. A rare case of ileus caused by ileum endometriosis.

    Science.gov (United States)

    Bratu, Dan; Chicea, Radu; Ciprian, Tanasescu; Beli, Laurentiu; Dan, Sabau; Mihetiu, Alin; Adrian, Boicean

    2016-01-01

    We report our experience involving a rare case of ileum endometriosis complicated with small bowel obstruction. 33 years old female patient, admitted to emergency service with abdominal pain, abdominal distension, and vomiting. Abdominal X-ray showed dilated small bowel loops. Computerized tomography scan showed dilated small intestine segments excepting last ileum loop, gastric distension, enlarged ovaries. Emergency laparotomy was performed, showing acute bowel obstruction due to a stenotic tumor placed on the terminal ileum, cecum tumors, multiple tumors in Douglas pouch, multiple mesenteric enlarged lymph nodes. Right colectomy is performed with an ileo-transverso stomy placed in right hypochondrium. Postoperative evolution without complication, patient discharged after 13-days hospitalization. After hormonal treatment, she returned for a second look and ileotransverso anastomosis. Gastrointestinal involvement of endometriosis has been found in 3%-37% of menstruating women. Ileum localization is very rare (1%-7%), causing intestinal obstruction 7%-23% of cases. Intraoperative differential diagnosis is difficult, predisposing at confusion with other types of tumors. In the absence of fast microscopic exam, the tumor was considered malignant and imposed a right hemicolectomy. Intestinal obstruction due to ileum endometriosis is a rare condition, however, it should always be considered in the differential diagnosis in women of reproductive age. Copyright © 2016 The Author(s). Published by Elsevier Ltd.. All rights reserved.

  18. 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.

  19. 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.

  20. A rare case of post traumatic isolated bilateral abducens nerve palsy

    Directory of Open Access Journals (Sweden)

    Jaiswal Manish

    2014-12-01

    Full Text Available Although unilateral abducens nerve palsy has been reported to be as high as 1% to 2.7% of traumatic brain injury, bilateral abducens nerve palsy following injury is extremely rare. In this report, we present the case of a patient who developed isolated bilateral abducens nerve palsy following minor head injury. He had a Glasgow Coma Score (GCS of 15 points. Computed tomography (CT images & Magnetic Resonance Imaging (MRI brain demonstrated no intracranial lesion. Herein, we discuss the possible mechanisms of bilateral abducens nerve palsy and its management.

  1. A rare case of renal vein thrombosis secondary to Klebsiella pneumoniae pyelonephritis

    Directory of Open Access Journals (Sweden)

    Kelly L Lurz

    2018-01-01

    Full Text Available Renal vein thrombosis (RVT is most often an implication of nephrotic syndrome. Pyelonephritis has been associated at a much lower rate, with the incidence of Klebsiella pneumoniae causation being extremely rare. In our case, a 35-year-old female patient presented with right-sided K. pneumoniae-positive acute pyelonephritis complicated by perinephric abscess and renal vein thrombosis. She was successfully treated with anticoagulation and extended antibiotic therapy. The possibility of RVT in patients with K. pneumoniae-induced pyelonephritis warrants consideration.

  2. Werewolf syndrome associated with gingival fibromatosis: A rare case report

    Directory of Open Access Journals (Sweden)

    Mysore K Sunil

    2016-01-01

    Full Text Available Hypertrichosis is a rare disorder that causes abnormal excessive body hair growth. Hypertrichosis has informally been called werewolf syndrome because the appearance is similar to that of a werewolf. It is often associated with additional anomalies including gingival hyperplasia, deafness, cardiomegaly and bone abnormalities. The association of gingival fibromatosis and a coarse facies could further worsen the esthetics. Thus, a multidisciplinary approach involving a psychologist, a dentist as well as a dermatologist would be mandatory. We present this rare syndrome describing a case of a 9-year-old boy with congenital hypertrichosis and gingival fibromatosis. The growth being normal, the features of this boy were similar to hypertrichosis. Hypertrichosis has informally been called werewolf syndrome because the appearance is similar to the werewolf.

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

    Directory of Open Access Journals (Sweden)

    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 .

  4. 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.

  5. 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.

  6. Symptomatic Bochdalek Hernia in Pregnancy: A Rare Case Report

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    Selçuk Yetkinel

    2017-01-01

    Full Text Available Introduction. Symptomatic Bochdalek hernia in pregnancy is quite rare. To the best of our knowledge, there are a total of 44 cases reported in the literature between 1959 and 2016 (Hernández-Aragon et al., 2015; Koca et al., 2016. Difficulty and delay in diagnosis may lead to life-threatening complications. Case Report. We report a case of Bochdalek hernia during the 30 gestational weeks’ pregnancy in whom pregnancy continued after surgical repair which resulted in term birth. Discussion. Bochdalek hernia is diagnosed with an incidence of 1 in 2200–12500 live births, while symptomatic diaphragm hernia is much less in adults. The actual incidence of diaphragmatic hernias during pregnancy is still unknown. Symptoms may include abdominal distension, recurrent abdominal pain, nausea, vomiting, inability to defecate, dyspnea, and chest pain. The patient with diaphragmatic hernia may be asymptomatic until the late weeks of gestation, as in our case, or herniation may occur during advanced gestational weeks with increased intraabdominal pressure. Conclusion. In conclusion, diagnosis of the diaphragm hernia during pregnancy is very rare. Diagnosis is rarer in symptomatic patients due to its rarity and the duration of diagnosis may, therefore, be delayed. Diaphragm hernia should be kept in mind in symptomatic patients due to its high maternal and fetal mortality rates.

  7. Atresia of the appendix vermiformis: A rare case of developmental abnormality

    Science.gov (United States)

    Yaylak, Faik; Algin, Mustafa Cem; Gungor, Yesim; Korkmaz, Mehmet

    2013-01-01

    Acute appendicitis, which requires immediate surgical intervention, is an important diagnosis in patients with acute abdomen. However, developmental abnormalities may interfere with the preoperative diagnosis and surgical treatment in some cases. Agenesis and atresia of the cecal vermiform appendix is an extremely rare clinical diagnosis. In addition, preoperative diagnosis may be difficult in some cases. Thus, diagnosis of the congenital absence of the vermiform appendix requires a thorough exploration of the retrocecal and ileocecal regions. A 59-year-old male was admitted from the emergency services with right lower abdominal pain. A celiotomy was performed with the suspicion of acute appendicitis. However, an atresia of the vermiform appendix was observed. The patient’s appendix was thus removed. Pathological examination confirmed suppurative appendicitis. This case underlines the importance of the clinical entity for surgeons who may deal with a similar case. PMID:23326173

  8. Atresia of the appendix vermiformis: a rare case of developmental abnormality.

    Science.gov (United States)

    Yaylak, Faik; Algin, Mustafa Cem; Gungor, Yesim; Korkmaz, Mehmet

    2013-01-07

    Acute appendicitis, which requires immediate surgical intervention, is an important diagnosis in patients with acute abdomen. However, developmental abnormalities may interfere with the preoperative diagnosis and surgical treatment in some cases. Agenesis and atresia of the cecal vermiform appendix is an extremely rare clinical diagnosis. In addition, preoperative diagnosis may be difficult in some cases. Thus, diagnosis of the congenital absence of the vermiform appendix requires a thorough exploration of the retrocecal and ileocecal regions. A 59-year-old male was admitted from the emergency services with right lower abdominal pain. A celiotomy was performed with the suspicion of acute appendicitis. However, an atresia of the vermiform appendix was observed. The patient's appendix was thus removed. Pathological examination confirmed suppurative appendicitis. This case underlines the importance of the clinical entity for surgeons who may deal with a similar case.

  9. 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.

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

    Science.gov (United States)

    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.

  11. 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.

  12. 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.

  13. 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

  14. Chondroblastic osteosarcoma of breast in a case of phyllodes tumour with recurrence, a rare case report

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    Sourav Sarkar, MS

    2016-01-01

    Conclusion: A diagnosis of chondroblastic osteosarcoma of the breast was made based on histology report and after excluding any osteogenic sarcoma arising from underlying ribs and sternum. It is a very rare disease with only few case reports in literature.

  15. Congenital anterior urethrocutaneous fistula: Report of two rare cases

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    Jayalaxmi Shripati Aihole

    2015-05-01

    Full Text Available Isolated congenital anterior urethrocutaneous fistiula (CAUF is a rare anomaly. CAUF can be defined as a fistula of the penile urethra associated with a normal foreskin, in which urethral meatus and prepuce are intact. Additionally, it may be associated with other anomalies such as congenital hernias and anorectal malformations. Treatment of CAUF is individualized according to site of fistula, associated anomalies and condition of the distal urethra. We are reporting our experience of two cases that had abnormal openings on the ventral aspect of distal penis with normal foreskin without hypospadias and chordee.

  16. 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.

  17. 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.

  18. 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.

  19. Acute amebic appendicitis: Report of a rare case

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    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.

  20. A rare case of spontaneous Aspergillus spondylodiscitis with epidural abscess in a 45-year-old immunocompetent female

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    K Ambedkar Raj

    2013-01-01

    Full Text Available Vertebral osteomyelitis and discitis are mainly due to bacterial infections though fungal infections are one of the important causes of morbidity and mortality in immunocompromised patients. Aspergillus involving intervertebral disc space is extremely rare. We report a case of aspergillosis of intervertebral L5-S1 disc space with spinal epidural abscess in an immunocompetent 45-year-old female which can add on to a few case reports described in literature as well as an insight for clinicians regarding this rare spontaneous infection in an immunocompetent patient.

  1. 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…

  2. '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.

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

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    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.

  4. Primary Pleomorphic Undifferentiated Sarcoma—a Rare Renal Localization: A Case Report

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    Soufiane Mellas

    2012-01-01

    Full Text Available Undifferentiated pleomorphic sarcoma is known as a soft tissue sarcoma. Very few cases of this tumor originating from the renal parenchyma or renal capsule have been reported. We report a case of a 70-year-old patient admitted for enormous ureterohydronephrosis and pyelonephritis due to a pelvic ureter lithiasis. After draining by ureteral double J catheter, a nephroureterectomy was performed for nonfunctional kidney confirmed by scintigraphy. The histopathological study shows a pleomorphic undifferentiated sarcoma. The patient was sent to oncologists. Chemotherapy was proposed but the family decided to stop the treatment. The patient passed away 10 months later. Clinicians and pathologists should be aware of the very low occurrence of this renal tumor, which is extremely rare. Currently there is no consensus about its management. Our case extends the literature concerning this tumor.

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

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    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.

  6. Postoperative keratitis due toPaecilomyces: a rare pediatric case.

    Science.gov (United States)

    Toker, Ebru; Ziyade, Nihan; Atici, Serkan; Eda, Kepenekli Kadayifçi; Türel, Özden; Toprak, Demet; Oray, Merih; Cerikcioglu, Nilgün; Soysal, Ahmet; Bakir, Mustafa

    2016-01-01

    Fungal infections like Paecilomyces keratitis have emerged in childhood recently. The diagnosis and treatment of Paecilomyces keratitis is difficult and the outcome is usually poor. Corneal culture should be performed on fungal media such as Sabouraud glucose neopeptone agar (SDA) as soon as possible for diagnosis. We report a rare case of Paecilomyces keratitis in an immunocompetent child, which was unresponsive to amphotericin B. The case was managed by a multidisciplinary approach involving the departments of ophthalmology, microbiology and pediatric infectious diseases. We want to draw attention once again that fungal keratitis caused by unusual agents are increasing. Physicians should consider fungal causes of keratitis, in patients with some predisposing factors like ocular surgery and prolonged use of topical corticosteroids.

  7. A Rare Case of Multifocal Prostatic Blue Nevus

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    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.

  8. 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.

  9. Paracetamol induced Steven-Johnson syndrome: A rare case report.

    Science.gov (United States)

    Rajput, Rajan; Sagari, Shitalkumar; Durgavanshi, Astha; Kanwar, Alpana

    2015-09-01

    In the contemporary era, use of drugs is the dominant paradigm of health care. The most quotidian drug used for fever and pain is paracetamol. Although adverse reactions to paracetamol in India are rare, at times they can cause life-threatening situations. Stevens-Johnson syndrome (SJS) is one such potentially lethal adverse drug reaction. The most reported cases of analgesic-induced SJS were due to oxicams or propionic acid derivatives. There are very few detailed reports of SJS due to the use of paracetamol. We report a case of SJS, which occurred due to the use of paracetamol. The clinical features of this condition and multidisciplinary management of the patient are described in brief.

  10. Tubular adenoid cystic carcinoma of palate: A rare case report

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    Deepanshu Garg

    2015-01-01

    Full Text Available Adenoid cystic carcinoma (ACC is a rare malignant tumor that affects the major and minor salivary glands, the lacrimal glands, the ceruminous glands and occasionally the excretory glands of the female genital tract. 30-40% of these carcinomas occur in the head and neck region. Approximately 50-70% of the cases have been reported in the minor salivary glands. The most common location is the palate, generally in the area of the greater palatine foramen. ACC is generally characterized by a slow growth rate, and it is often present for several years before the patient seeks treatment. Here we present a case of 42-year-old female who came to the department for the complaint of a swelling over the right side of palate since 2-3 months which was diagnosed as a benign tumor initially, but was confirmed as tubular ACC histologically.

  11. A rare case of Histoplasma fungemia in an AIDS patient

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    V P Baradkar

    2011-01-01

    Full Text Available Disseminated cases of histoplasmosis in acquired immune deficiency syndrome (AIDS are rarely reported from India. Most of these cases report isolation of this fungus from the bone marrow, lymph node aspirate, spleenic aspirate, and biopsies. We report isolation of Histoplasma capsulatum from the blood of an AIDS patient. A 30-year-old male from Utter Pradesh was admitted with fever, loss of appetite, and nausea since two months. Few intracellular and extracellular budding cells were observed on bone marrow examination on the fifth day of admission. Diagnosis was confirmed by blood cultures taken on the 11th day of admission. Amphotericin B was started, but the patient′s condition deteriorated and he died.

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

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    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.

  13. Paracetamol induced Steven-Johnson syndrome: A rare case report

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    Rajan Rajput

    2015-01-01

    Full Text Available In the contemporary era, use of drugs is the dominant paradigm of health care. The most quotidian drug used for fever and pain is paracetamol. Although adverse reactions to paracetamol in India are rare, at times they can cause life-threatening situations. Stevens-Johnson syndrome (SJS is one such potentially lethal adverse drug reaction. The most reported cases of analgesic-induced SJS were due to oxicams or propionic acid derivatives. There are very few detailed reports of SJS due to the use of paracetamol. We report a case of SJS, which occurred due to the use of paracetamol. The clinical features of this condition and multidisciplinary management of the patient are described in brief.

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

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    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.

  15. A Rare Case of Labial Myiasis caused by Dermatobia hominis.

    Science.gov (United States)

    Meurer, Maria I; Grando, Liliane J; Rivero, Elena Rc; Souza, Carlos Ecp; Marcondes, Carlos B

    2016-11-01

    The aim of this article is to present a rare case of myiasis caused by Dermatobia hominis. Myiasis is a disease caused by invasion of tissues of animals and humans by larval stages of dipterous (2-winged) flies. There are few reports of oral myiasis in the literature, mostly related to Cochliomyia hominivorax larvae. We present a case of a 53-year-old man with painful swelling of the left lower lip that was confirmed to be myiasis caused by D. hominis. Though more common in tropical and subtropical regions of North and South America, myiasis should be considered in the differential diagnosis of subcutaneous masses among residents or travelers in endemic areas.

  16. Cowden syndrome- Clinico-radiological illustration of a rare case

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    Prashant B Patil

    2013-01-01

    Full Text Available Cowden syndrome (CS or multiple hamartoma syndrome is an infrequent genodermatoses, which is inherited as an autosomal dominant trait resulting from the mutation in the Phosphatase and Tensin homolog gene on the arm 10q and is principally characterized by multiple hamartomas with an increased risk of development of malignancies. Facial and oral signs are remarkable in the form of multiple papules and trichilemmomas on the face. We report one such rare case of CS in a 19-year-old patient who was diagnosed on the basis of her oral mucosal lesions and was further investigated and diagnosed with other hamartomas. The present case report signifies the responsibility of the oral physician in the early diagnosis of this progressive pathological syndrome as it leaves its footmark in the oral cavity in the form of oral mucosal lesions.

  17. Recurrent Rhabdomyolysis and Extreme Exercise-A Case Study

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    Matthew Wagner

    2015-09-01

    Full Text Available Objective: To present the case of recurrent rhabdomyolysis in an active female Background: A healthy 21 year old female developed severe fatigue and peripheral edema in the upper extremities after an extreme exercise workout session. 4 ½ months later she participated in a similar exercise session with a personal trainer and experienced the same symptoms. Differential Diagnosis: In the initial case, upon physical examination and review of blood work a diagnosis of rhabdomyolysis was made. After the second exercise session athlete was diagnosed with recurrent rhabdomyolysis and was advised to seek treatment at the local emergency room. Treatment: In the initial case the athlete was told by medical personnel to increase fluid intake and rest. In the second case athlete was treated at the emergency room with IV fluids. Uniqueness: The athlete was female, and most cases of rhabdomyolysis occur in males. Athlete was treated for rhabdomyolysis and recurrent rhabdomyolysis, however these diagnosis were made 4 ½ months apart. Also, both cases appear to be the result of the same type of exercise session. Conclusion: There does not appear to be a definitive protocol for an accurate diagnosis of rhabdomyolysis. Guidelines for return to activity should be evaluated on a case by case basis, and a conservative increase in intensity of exercise is warranted to prevent a case of recurrent rhabdomyolysis.

  18. [Osteoblastoma in the os hamatum: a rare case report].

    Science.gov (United States)

    Ayan, Irfan; Serinsöz, Ebru

    2014-01-01

    Osteoblastoma in the os hamatum is rarely seen. Although curettage and grafting seems to be disadvantageous, it offers advantages in the functional protection in the treatment of carpal bone-located osteoblastoma. In a 39-year-old housewife who was admitted with painful left wrist through all day for the past one year, physical examination revealed painful hypothenar region with the wrist adduction to the ulna. Radiography showed radial inclination of the wrist, ring appearance in the scaphoid bone, and slight radiolucency in the hamatum and adjacent bones. Computed tomography demonstrated an expanded lesion which separated the surrounding tissue with a thin edge layer and perforated the cortex mildly. Intralesional curettage was performed. The pathological examination of the specimen obtained was consistent with osteoid osteoma or osteoblastoma. Curettage and grafting were performed in case of recurrence. In this article, we present a rare case of carpal bone and hamatum-located osteoblastoma. The patient was free of pain with normal wrist functions at 16 months postoperatively.

  19. A rare case of cutaneous leiomyosarcoma in budgerigar (Melopsittacus undulatus

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    Zamani-Ahmadmahmudi, M.

    2015-04-01

    Full Text Available Leiomyosarcoma in birds is relatively rare. This tumor as a muscle neoplasm was reported in captive and free ranging birds. Smooth muscle cells may develop to the leiomyosracoma, but splenic smooth muscle trabeculae is most common site of the tumor growth. Although budgerigars have an incidence of neoplastic diseases but smooth muscle tumors were rarely reported in this species. To the best of our knowledge, present case is the first report of cutaneous leiomyosarcoma in budgerigar. The bird was referred with a history of growing mass in subcutaneous tissue of abdomen. First bird was suspected to the egg impaction, but necropsy confirmed a firm, creamy structure that suspected to the tumor mass. No invasion was observed to the other organs. After excision of the mass, routine histopathologic evaluation and immunohisochemistry investogation were performed for desmin and smooth muscle actin (SMA. Histopathologic examination revealed spindle-shaped cell with cigar-shaped, round, or oval nuclei with cytologic criteria of malignancy including marked pleomorphism and high mitotic activity. These findings were consistent with immunohistochemistry profile of our case and thus confirmed as cutanous liomyosarcoma.

  20. 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.

  1. 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.

  2. Infectious endotheliitis: a rare case of presumed mycotic origin

    Science.gov (United States)

    Zapata, Luis Fernando; Paulo, José David; Restrepo, Carlos A; Velásquez, Luis Fernando; Montoya, Andrés E Toro; Zapata, Melissa A

    2013-01-01

    Purpose To report an interesting case of infectious endotheliitis of presumed mycotic origin. Methods A case report of a 56-year-old male farmer who sought medical attention after a month-long evolution of irritative symptoms in his right eye, accompanied by visual acuity (VA) impairment. The patient received topical and oral broad-spectrum antibiotic treatment with no improvement before being referred to a cornea specialist, where he was found to have VA of 20/150 and was noted on biomicroscopy to have endothelial feathery coalescent lesions. The patient was admitted to the hospital for an aqueous humor sample and intravenous voriconazole. Results The microbiological studies did not isolate any micro-organisms. However, clinical evidence of improvement was confirmed after 5 days of antimycotic intravenous therapy. Complete clinical resolution was achieved at 1 month after treatment completion with oral voriconazole, as evidenced by VA of 20/20 and disappearance of endothelial lesions. Conclusion Endothelial involvement by fungi is a rare condition. In this case, no microbes were isolated, but the characteristic morphology of the lesions, the history of onychomycosis, and the spectacular response to voriconazole turn this case into a valid presumptive diagnosis. PMID:23901253

  3. Colloidon baby – Rare case with preventable complications

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    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.

  4. 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.

  5. An Extremely Rare Manifestation of Multiple Myeloma: An Immunoglobulin D Secreting Testicular Plasmacytoma.

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    Sharma, Ashish; Binazir, Tina; Sintow, Alexandre; Lee, Chi Chan; Shaharyar, Sameer; Tache, Jason

    2017-06-27

    Multiple myelomas (MM) of the immunoglobulin D (IgD) subtype is rare amongst plasma cell malignancies. It can present a diagnostic challenge because of the low amount of immunoglobulin in the serum. The amount of monoclonal (M)-protein is often undetectable on electrophoresis. Historically, survival in these patients was typically shorter compared to the immunoglobulin A (IgA) and immunoglobulin G (IgG) subtypes due to advanced disease upon presentation. With the advent of better diagnostic techniques, the prognosis of this disease is changing. We describe a case of an extramedullary testicular plasmacytoma (EMP) of the IgD subtype as the primary feature of MM, which responded well to novel therapy. A 72-year-old White male presented to the emergency room with a right testicular mass for three months. He subsequently underwent right radical orchiectomy. Pathology of the specimen revealed plasmacytoid cells positive for cluster of differentiation (CD79a), lambda free light chain, IgD, and BCL-1 (Cyclin D1) on immunochemical stains. Urine and serum immunofixation were positive for monoclonal IgD with lambda light chain specificity and Bence Jones proteinuria. Bone marrow biopsy showed large sheets of plasma cells with greater than 90% cellularity. Flow cytometry displayed atypical plasma cells expressing cluster of differentiation (CD38, CD20, and CD56) with cytoplasm and lambda light chain, approximately 20%, consistent with a plasma cell dyscrasia. Stage 3 IgD lambda multiple myeloma was diagnosed. He received novel treatment with Bortezomib and dexamethasone for three months, followed by Lenalidomide. His performance status and lab data improved significantly. He had progression-free survival (PFS) of approximately three years and remained in complete remission low-dose dose of Lenalidomide daily. IgD myeloma was considered a diagnostic challenge due to undetectable M-protein levels on serum protein electrophoresis (SPEP). With the advent of serum free light chain

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

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    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.

  7. Imaging diagnosis of orbital Wegener granulomatosis: A rare case report.

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    Yang, Bin; Yin, Zhijian; Chen, Shuai; Yuan, Feng; Zhao, Wei; Yang, Yaying

    2017-06-01

    Wegener granulomatosis (WG) is a rare idiopathic autoimmune disease causing necrotizing granulomatous vasculitis. Whether as the first symptom or as part of systemic changes, ocular manifestations in WG patients are not specific. Any part of the eyes can be affected, with the anterior segment and orbit most commonly involved. So, early diagnosis and treatment are essential for controlling the progression of the disease and improving the quality of life for patients. Here we present a rare case of orbital WG of a 22-year-old woman was admitted to the hospital because of intense pain associated with decreased visual acuity in her right eye since 1 day. She had been previously diagnosed with WG at our hospital. Orbital computed tomography imaging showed diffuse swelling of intraorbital muscles, and space-occupying lesions were present in both eyes. Most postnasal anatomical structures were absent, appearing as a massive cavity shadow. Orbital magnetic resonance imaging showed a shadow of orbital soft tissues. WG is a serious, fatal disease. Early diagnosis and treatment are essential for controlling the progression of the disease and improving the quality of life for patients.

  8. A rare case of right lower quadrant abdominal pain.

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    Macedo, Mark; Kim, Brian; Khoury, Rami; Narkiewicz, Larry

    2017-04-01

    Isolated fallopian tube torsion without involvement of the ovary is a rare condition most frequently presenting during reproductive years. Imaging, vitals, physical exam, and laboratory findings all fail to help establish a definitive diagnosis. The majority of the diagnoses are made on the operating table. Physical exam most often reveals unilateral and localized abdominal pain, often with nausea and vomiting, but few other reliably common findings. Diagnosis becomes even more challenging due to the fact that isolated tubal torsion occurs often in pregnancy and preferentially on the right, further complicating the clinical picture. We describe a case of isolated tubal torsion, unique in that localized necrosis and inflammation from the torsion triggered a secondary appendicitis. The patient required surgical intervention, and an appendectomy and salpingectomy emergently. Given its elusive and rare nature, awareness and early intervention is required by the emergency physician to recognize tubal torsion, as operative intervention is crucial, and can lead to preservation of fertility and improved fetal survival. Copyright © 2016 Elsevier Inc. All rights reserved.

  9. Management dilemmas in a rare case of pituitary apoplexy in the setting of dengue hemorrhagic fever.

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    Balaparameswara Rao, S J; Savardekar, Amey R; Nandeesh, B N; Arivazhagan, A

    2017-01-01

    Pituitary apoplexy occurs due to infarction or hemorrhage, within a pituitary adenoma or a nontumorous pituitary gland and can have catastrophic consequences. Dengue hemorrhagic fever (DHF) is a severe manifestation of the spectrum of dengue virus infection and is characterized by high-grade fever, thrombocytopenia, hemorrhagic tendencies, and increased vascular permeability. Cases of incidentalomas complicated by DHF and presenting with apoplexy are extremely rare. We describe the case of a 45-year-old gentleman who suffered an attack of pituitary apoplexy while being treated for DHF. The issues pertaining to the management of hydrocephalus, timing of surgical intervention, and treatment of electrolyte imbalances encountered in the dual setting of DHF and pituitary apoplexy are discussed with reference to the outcome in our case. Although patients suffering from DHF harbor multiple factors, which may be precipitants of pituitary apoplexy, the association between these two conditions is rare and only few case reports document their coexistence. We review the pertinent literature and discuss the management dilemmas faced by us while dealing with these dual pathological states.

  10. Idiopathic systemic granulomatous pathology causing sudden death due to myocarditis: A rare case report

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    Harpal Singh

    2015-01-01

    Full Text Available Idiopathic granulomatous myocarditis is extremely rare, particularly since the introduction of drugs effective against tuberculosis (TB, viruses, fungi and the effective treatment of sarcoidosis. Here is a case of a 65-year-old female prisoner having history of sudden collapse and ultimately death. Autopsy findings of various viscera on histopathological examination show granulomatous pathology, that is, in spleen, liver and in the left ventricular wall of heart. Ziehl-Neelsen staining of the sections show the absence of acid fast bacilli, negative for fungal staining as most of the granulomas are noncaseating type with presence of giant cells having no asteroid body and Schuamann body, real-time polymerase chain reaction for TB is negative. Idiopathic giant cell myocarditis is a disease of relatively young adults, that is, between 3 rd and 4 th decade of life. So, this case is strongly considered to be a case of sudden death due to myocarditis as a result of idiopathic systemic granulomatous pathology, a rare case in in literature.

  11. Positive identification in a case of intentional extreme fragmentation.

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    Owsley, D W; Mann, R W; Chapman, R E; Moore, E; Cox, W A

    1993-07-01

    The investigation of the first of the murders of young males to which Jeffrey Dahmer confessed led to systematic survey of two acres of semirural property in Bath Township, Ohio. The survey revealed the fragmentary skeleton of a young adult male, as well as bones of several species of animals. Through archaeobiological analysis, the animal bones were identified and taphonomic modifications documented. The human bones were dry and weathered, and many were rodent-gnawed, indicating that they had been exposed for many years. The human bones displayed an extreme degree of splintering, warping, bending, and spiral breakage. The only relatively complete bone was a cervical vertebra. Dismemberment and breakage had been accomplished by cutting, blunt force, and, in the case of the ribs, manual bending. Even in so extreme a case of intentional fragmentation, however, forensic anthropological analysis resulted in positive identification, with the primary criteria based on matching osteological features evident in premortem and postmortem radiographs of a cervical vertebra.

  12. 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.

  13. Rapunzel syndrome - A rare form of trichobezoar: A case report

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    L Thayumanavan

    2013-01-01

    Full Text Available Hair ball in the GI tract is called trichobezoar and the Rapunzel syndrome is a very rare form of trichobezoar found in patients with psychiatric illness in whom the ingested hair extends from the stomach into the small intestine. Trichotillomania (hair pulling and trichophagia (chewing of hair are intrinsic to the development of Rapunzel syndrome. A small number of patients have been reported in whom the gastric trichobezoar has a long tail and extends throughout the small bowel to the cecum. This condition, known as the Rapunzel syndrome, occurs almost exclusively in young girls. We report a case Rapunzel syndrome who presented with history of upper GI obstruction and weight loss. Upper GI endoscopy revealed a large trichobezoar occupying fundus of stomach extending along the whole stomach into duodenum beyond its 3rd part. It was treated successfully with surgery followed by psychiatric consultation. We will review the literature on the subject as well.

  14. 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.

  15. GITELMAN SYNDROME AS A RARE CAUSE OF HYPOKALEMIA - CASE REPORT

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    Zorica Dimitrijević

    2014-09-01

    Full Text Available Gitelman syndrome is a rare autosomal recessive tubulopathy leading to hypokalemia, metabolic alkalosis, hypomagnesemia, hypocalciuria and low-to-normal blood pressure. Clinical signs are mostly secondary to chronic hypokalemia and include dizziness, fatigue, constipation and weakness. Patients can also present with muscle cramps, tetany and convulsions due to severe metabolic alkalosis or hypomagnesemia. Therefore, early recognition and treatment are important. Diagnosis of Gitelman syndrome is usually made incidentally during adolescence or early adulthood based on clinical and biochemical findings. In this paper we report a case of a young women with classic Gitelman syndrome. Treatment included magnesium and potassium salts and potassium saving diuretics. In general, the long-term prognosis of Gitelman syndrome is excellent. However, the severity of fatigue may seriously hamper some patients in their daily activities.

  16. A rare case of a groin hernia: the Hesselbach's hernia.

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    van den Heuvel, B; Munoz Brands, R M; Beuerle, E Y; Dwars, B J

    2015-06-01

    A 61-year-old woman presented with pain in the left groin. The pain radiated from the groin to the knee. At physical examination, a non-reducible swelling was found lateral to the femoral vessels. A CT-scan showed herniation of fatty tissue through the lacuna musculorum. A laparoscopic repair was carried out. A large adipose structure was found herniating through the lacuna musculorum and originating from the Bogros area. It ran along the iliopsoas muscle, suppressing the femoral cutaneous nerve. The tissue was reduced and excised, and a polypropylene mesh was placed to cover the defect. Inguinal hernias are categorized as medial, lateral or femoral hernias. We describe a case in which a hernia through the lacuna musculorum was found, which is very rare and referred to as a Hesselbach's hernia.

  17. A rare case of fibrostenotic endobronchial tuberculosis of trachea

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    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.

  18. A Rare Case of Septic Shock Secondary to Emphysematous Hepatitis

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    Khaled M. Nada

    2017-01-01

    Full Text Available Objective. To describe a case of emphysematous hepatitis which is a rare clinical entity, characterized by a fatal, rapidly progressive infection of the liver with a radiological appearance simulating emphysematous pyelonephritis and to help provide more data about the causative organisms and precipitating factors of this pathology. Data Sources and Synthesis. Relevant literature was reviewed and, to the best of our knowledge, there is limited data regarding the pathogenesis, causative organisms, and management of this condition. Conclusion. Emphysematous hepatitis is a rapidly progressive infection that can be fatal in the absence of appropriate therapeutic intervention. Initial clinical manifestations are usually subtle and thus high clinical suspicion is required for early diagnosis and management of this condition to help decrease the mortality rates.

  19. A rare case of ulcerative colitis exacerbated by VZV infection.

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    Nishimura, Satoshi; Yoshino, Takuya; Fujikawa, Yoshiki; Watanabe, Masaki; Yazumi, Shujiro

    2015-12-01

    A 16-years old man with severe ulcerative colitis (UC) was admitted to our hospital. After initiating treatment with corticosteroid for UC, chicken pox appeared. At the same time of appearance of chicken pox, the disease activity of UC was exacerbated. After initiating the treatment with acyclovir, both chicken pox and UC improved. Because colonoscopic findings revealed the remaining of moderately active UC, initiating the treatment with infliximab could induce clinical remission of UC without relapse of varicella-zoster virus (VZV) infection. This is a very rare case of UC with concomitant VZV infection. According to our report, the vaccination for VZV prior to immunosuppressive treatments would be necessary for VZV naïve patients with UC.

  20. 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.

  1. Adenoid cystic carcinoma of the mobile tongue: A rare case

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    Pavitra Baskaran

    2012-01-01

    Full Text Available Adenoid cystic carcinoma (ACC occurs more commonly in the minor salivary glands of the palate on than the tongue. ACC is a malignant neoplasm that accounts for 1-2% of all head and neck malignancies and 10-15% of all salivary gland malignancies. ACC affects the exocrine glands at any site, but the parotid gland is the most common site in the head and neck region. Many factors should be taken into account in the prognosis of ACC, including the histological and clinical stages of the disease. The most striking feature of ACC is that it is locally aggressive, with a high recurrence level, perineural invasion and distant metastases, especially to the lungs and bones. The most common presentation histologically is the presence of cribriform appearance (Swiss cheese pattern. The present case is a rare one present on the tongue.

  2. Localized renal cystic disease: Report of a rare case

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    Gupta Shilpi

    2010-01-01

    Full Text Available We report a case of a 32-year-old female who presented with right flank pain. Ultrasonography done for hematuria 10 years ago indicated the presence of renal cysts in the right kidney, but she had been asymptomatic since then. This history attracted our attention to the possibility of "localized renal cystic disease" (LRCD. Diagnosis was confirmed by computerized tomogram, negative renal ultrasound of one of the parents and absence of family history of renal disease. She is still being followed up for the last 3 years and is doing well. LRCD has been given various names since it was first recognized as a distinct clinical entity but the term LRCD appears to be the most accurate. Here, we present a brief discussion of clinical significance, diagnosis and differentials of this rare condition.

  3. Limbal dermoid in Nager acrofacial dysostosis: A rare case report

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    Rohit Malik

    2014-01-01

    Full Text Available Nager syndrome, also called preaxial acrofacial dysostosis, comprises two groups of defects involving the limbs and craniofacial region, respectively. This syndrome is rare and only 70 cases have been reported in the literature. The exact cause of this syndrome is unknown, but there is indication that it is genetically based. Ocular manifestations of this syndrome include widely separated downward slanting eyes, absence of eyelashes, ptosis of upper eyelids and colobomas on the inner aspect of lower eyelids. We report limbal dermoid in a patient with Nager syndrome. We did not find such an association of "Limbal dermoid in Nager acrofacial dysostosis syndrome" on PubMed using Nager acrofacial dysostosis, limbal dermoid and ocular manifestations as the keywords.

  4. 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.

  5. 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.

  6. 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.

  7. Adderall induced acute liver injury: a rare case and review of the literature.

    Science.gov (United States)

    Vanga, Rohini R; Bal, Bikram; Olden, Kevin W

    2013-01-01

    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.

  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. A rare case of persistent hypoglossal artery associated with contralateral proximal subclavian stenosis

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    Romeo Antonio

    2016-01-01

    Full Text Available The persistent hypoglossal artery is rare vascular anomalies. We report the case of a 50-year old man with right hypoglossal artery, ipsilateral hypoplasic internal carotid artery, associated with left proximal subclavian stenosis with subclavian steal syndrome. Power-Doppler-Ultra-Sonography spectral images obtained after the patient exercised the left arm showed mid-systolic deceleration with retrograde late-systolic velocities. A Computed Tomography Angiography demonstrated a proximal stenosis of the left SA, a mild right ICA hypoplasia and an anomalous artery arising from right ICA at C2–C3 level, entering the cranium via the hypoglossal canal and joining the basilar artery. Usually the presence of PHA may be completely asymptomatic, and detected as an incidental finding by CTA or MRA, but in our case its diagnosis is extremely important because it is often the only vessel supplying blood to the basilar trunk and posterior circulation.

  10. Mandible ameloblastoma with lung metastasis: a rare case report.

    Science.gov (United States)

    Yang, Rui-Na; Wang, Xin-Shuai; Ren, Jing; Xie, Yan-Fei; Zhou, Dan; Ge, Dong-Feng; Feng, Xiao-Shan; Gao, She-Gan

    2015-01-01

    The ameloblastoma is the most common odontogenic epithelial tumor, which belong to benign neoplasms that present a painless course, and usually occur in the oromaxillo-facial region. Although the histopathological manifestation of ameloblastoma is benign, it has unique biological behavior, for example local invasion and recurrence repeatedly. A few case of ameloblastoma was locally aggressive growth, and rarely metastasis to other tissue, for example the lungs, lymph nodes, and spine. A 64-year-old Chinese man, diagnosed with metastatic ameloblastoma, was treated with palliative chemotherapy consisting of cyclophosphamide, doxorubicin, and cisplatin for six cycles, and radiotherapy for 50 Gy after the last cycle chemotherapy. During the surveillance CT scan after the therapy, the tissues of the tumor were nearly complete response. The purpose of this study was to report a case of a patient with a right mandible ameloblastoma that recurred repeatedly and metastasized into bilateral lung. After the chemotherapy and radiotherapy, the tissues of the tumor were nearly complete response. This case is interesting because it investigated the diagnosis and treatment of the malignancy ameloblastoma, as this may help diagnose and treatment for clinician to the metastatic ameloblastoma.

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

    Directory of Open Access Journals (Sweden)

    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.

  12. Rare case of a strangulated intercostal flank hernia following open nephrectomy: A case report and review of literature

    Science.gov (United States)

    Akinduro, Oluwaseun O.; Jones, Frank; Turner, Jacquelyn; Cason, Frederick; Clark, Clarence

    2015-01-01

    Introduction Flank incisions may be associated with incisional flank hernias, which may progress to incarceration and strangulation. Compromised integrity of the abdominal and intercostal musculature due to previous surgery may be associated with herniation of abdominal contents into the intercostal space. There have been six previously reported cases of herniation into the intercostal space after a flank incision for a surgical procedure. This case highlights the clinical picture associated with an emergent strangulated hernia and highlights the critical steps in its management. Presentation of case We present a case of a 79-year-old adult man with multiple comorbidities presenting with a strangulated flank hernia secondary to an intercostal incision for a right-sided open nephrectomy. The strangulated hernia required emergent intervention including right-sided hemi-colectomy with ileostomy and mucous fistula. Discussion Abdominal incisional hernias are rare and therefore easily overlooked, but may result in significant morbidity or even death in the patient.. The diagnosis can be made with a thorough clinical examination and ultrasound or computed topographical investigation. Once a hernia has become incarcerated, emergent surgical management is necessary to avoid strangulation and small bowel obstruction. Conclusion Urgent diagnosis and treatment of this extremely rare hernia is paramount especially in the setting of strangulation. PMID:26629848

  13. Tracing electron-ion recombination in nanoplasmas produced by extreme-ultraviolet irradiation of rare-gas clusters.

    Science.gov (United States)

    Schütte, B; Campi, F; Arbeiter, M; Fennel, Th; Vrakking, M J J; Rouzée, A

    2014-06-27

    We investigate electron-ion recombination in nanoplasmas produced by the ionization of rare-gas clusters with intense femtosecond extreme-ultraviolet (XUV) pulses. The relaxation dynamics following XUV irradiation is studied using time-delayed 790-nm pulses, revealing the generation of a large number of excited atoms resulting from electron-ion recombination. In medium-sized Ar-Xe clusters, these atoms are preferentially created in the Xe core within 10 ps after the cluster ionization. The ionization of excited atoms serves as a sensitive probe for monitoring the cluster expansion dynamics up to the ns time scale.

  14. Rare-gas clusters in intense extreme-ultraviolet pulses from a high-order harmonic source.

    Science.gov (United States)

    Schütte, B; Arbeiter, M; Fennel, Th; Vrakking, M J J; Rouzée, A

    2014-02-21

    We report evidence for two previously unidentified effects in the ionization of rare-gas clusters by intense extreme-ultraviolet pulses. First, electron spectra indicate multistep photoemission with increasing isotropy for larger clusters due to electron-atom collisions. Second, very slow (meV) electrons are interpreted as the first experimental evidence for Rydberg-like atomic state formation in the nanoplasma expansion. Only small fractions of Xe2+ ions were found, in sharp contrast to previous results recorded under comparable conditions [Murphy et al., Phys. Rev. Lett. 101, 203401 (2008).

  15. Infectious endotheliitis: a rare case of presumed mycotic origin

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    Zapata LF

    2013-07-01

    Full Text Available Luis Fernando Zapata,1 José David Paulo,1 Carlos A Restrepo,1 Luis Fernando Velásquez,2 Andrés E Toro Montoya,2 Melissa A Zapata21Department of Ophthalmology Hospital Pablo Tobón Uribe; 2School of Medicine, Universidad Pontificia Bolivariana, Medellín, ColombiaPurpose: To report an interesting case of infectious endotheliitis of presumed mycotic origin.Methods: A case report of a 56-year-old male farmer who sought medical attention after a month-long evolution of irritative symptoms in his right eye, accompanied by visual acuity (VA impairment. The patient received topical and oral broad-spectrum antibiotic treatment with no improvement before being referred to a cornea specialist, where he was found to have VA of 20/150 and was noted on biomicroscopy to have endothelial feathery coalescent lesions. The patient was admitted to the hospital for an aqueous humor sample and intravenous voriconazole.Results: The microbiological studies did not isolate any micro-organisms. However, clinical evidence of improvement was confirmed after 5 days of antimycotic intravenous therapy. Complete clinical resolution was achieved at 1 month after treatment completion with oral voriconazole, as evidenced by VA of 20/20 and disappearance of endothelial lesions.Conclusion: Endothelial involvement by fungi is a rare condition. In this case, no microbes were isolated, but the characteristic morphology of the lesions, the history of onychomycosis, and the spectacular response to voriconazole turn this case into a valid presumptive diagnosis.Keywords: endotheliitis, mycotic, keratitis, voriconazole

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

    International Nuclear Information System (INIS)

    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

  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. 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.

  19. 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.

  20. 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.

  1. 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.

  2. 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.

  3. A Rare Case of Pediatric Lumbar Spinal Ependymoma Mimicking Meningitis.

    Science.gov (United States)

    Ekuma, Ezeali Mike; Ito, Kiyoshi; Chiba, Akihiro; Hara, Yosuke; Kanaya, Kohei; Horiuchi, Tetsuyoshi; Ohaegbulam, Samuel; Hongo, Kazuhiro

    2017-04-01

    Spontaneous acute subarachnoid hemorrhage (SAH) from lumbar ependymoma in children is rare. We report a case of a 14-year-old boy who developed sudden radicular low back pain while playing baseball. He was initially managed conservatively in a local hospital for suspected lumbar disc herniation, but he later developed meningeal symptoms and fever before being referred to our hospital. He underwent a diagnostic lumbar puncture in the emergency department; his cerebrospinal fluid suggested an SAH. Physical examination showed meningeal signs and cauda equina features. Cerebrospinal fluid analysis was negative for bacterial meningitis. Lumbar magnetic resonance imaging revealed a mass characterized as a hemorrhagic lesion. The patient had an emergent evacuation of the mass through the posterior approach. Postoperatively, his symptoms resolved completely. The histologic diagnosis was, surprisingly, an ependymoma (World Health Organization grade II). This case is particularly interesting because of its rarity in children, and its pattern of presentation. Although bacterial or viral meningitis is the most frequent cause of meningeal features in children, SAH from a hemorrhagic spinal tumor should be considered. Ultimately, a high index of suspicion is needed for prompt diagnosis. Copyright © 2017 Elsevier Inc. All rights reserved.

  4. Rare case of palatal schwannoma: Case report and review of literature

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    Uday Shankar Yaga

    2015-01-01

    Full Text Available Schwannomas, also known as neurilemmoma, are benign, slow-growing nerve sheath tumors arising from Schwann cells. Approximately 25-40% of schwannomas occur in the head and neck region. However, schwannomas that present in the oral cavity are relatively rare, constituting around 1% of all described cases in the head and neck region. [1],[2] Here, we report a rare case of an intraoral schwannoma, in a 28-year-old male, with painless swelling located in the posterolateral aspect of the soft palate on the right side. Definitive diagnosis was obtained after histopathology examination. Surgical excision of the tumor was done with no complications or recurrences.

  5. Rare case of pulmonary histoplasmosis complicated with bronchocentric granulomatosis in a non endemic area.

    Science.gov (United States)

    Botsa, Evanthia; Thanou, Ioanna; Kabanarou, Stamatina; Thanos, Loukas

    2017-01-01

    Pulmonary histoplasmosis is a fungal infection caused by histoplasma capsulatum, rarely diagnosed in non endemic areas and/or immunocompromised patients. Complication of pulmonary histoplasmosis with bronchocentric granulomatosis is extremely rare. A 48-year-old man with prolonged fever and nausea was admitted to our hospital. Clinical examination revealed pathological auscultatory sounds to the left lung. Computed tomography was performed and revealed a large solid mass of the left upper lobe, limited pleural and pericardial effusion and calcified lymphadenopathy of mediastinum. A computed tomography guided core biopsy of the lung lesion was performed and three samples were obtained. Culture and polymerase chain reaction (PCR) revealed Histoplasma capsulatum . Histological findings were compatible with bronchocentric granulomatosis. Extended laboratory investigation excluded immunosuppresion. Our patient although immunocompetent was diagnosed with chronic pulmonary histoplasmosis complicated with bronchocentric granulomatosis and treatment with antifungal medication and methylprednisoline started. Description of a rare case of chronic pulmonary histoplasmosis in a non endemic area like Greece, with atypical radiological findings, complicated with bronchocentric granulomatosis.

  6. A rare case of an aggressive osteoblastoma of the squamous temporal bone: a unique presentation with literature review.

    Science.gov (United States)

    Mohanty, Sujata; Rani, Amita; Urs, A B; Dabas, Jitender

    2014-10-01

    Aggressive osteoblastoma is a rare osteoid tissue forming tumour commonly affecting the spine with predilection for the posterior elements. Calvarial involvement is extremely rare with only two reported cases in the literature. Due to its overlapping clinical, radiographic and histological features with ossifying fibroma, benign osteoblastoma and osteosarcoma, it is very difficult accurately to diagnose this lesion at an early stage. A rare case of an aggressive osteoblastoma of the squamous temporal bone in a young male is presented here which was misdiagnosed twice before reaching the final diagnosis by correlating clinical, radiographic and histopathological features. Copyright © 2014 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.

  7. 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.

  8. Rare case of a strangulated intercostal flank hernia following open nephrectomy: A case report and review of literature.

    Science.gov (United States)

    Akinduro, Oluwaseun O; Jones, Frank; Turner, Jacquelyn; Cason, Frederick; Clark, Clarence

    2015-01-01

    Flank incisions may be associated with incisional flank hernias, which may progress to incarceration and strangulation. Compromised integrity of the abdominal and intercostal musculature due to previous surgery may be associated with herniation of abdominal contents into the intercostal space. There have been six previously reported cases of herniation into the intercostal space after a flank incision for a surgical procedure. This case highlights the clinical picture associated with an emergent strangulated hernia and highlights the critical steps in its management. We present a case of a 79-year-old adult man with multiple comorbidities presenting with a strangulated flank hernia secondary to an intercostal incision for a right-sided open nephrectomy. The strangulated hernia required emergent intervention including right-sided hemi-colectomy with ileostomy and mucous fistula. Abdominal incisional hernias are rare and therefore easily overlooked, but may result in significant morbidity or even death in the patient.. The diagnosis can be made with a thorough clinical examination and ultrasound or computed topographical investigation. Once a hernia has become incarcerated, emergent surgical management is necessary to avoid strangulation and small bowel obstruction. Urgent diagnosis and treatment of this extremely rare hernia is paramount especially in the setting of strangulation. Copyright © 2015 The Authors. Published by Elsevier Ltd.. All rights reserved.

  9. A rare case of osteoblastoma associated to aneurysmal bone cyst of the spine. Case report.

    Science.gov (United States)

    Pavanello, Marco; Melloni, Ilaria; Fiaschi, Pietro; Consales, Alessandro; Piatelli, Gianluca; Ravegnani, Marcello; Nozza, Paolo; Gandolfo, Carlo; Cama, Armando

    2016-01-01

    Osteoblastoma is a rare bone tumour. It is occasionally associated with an aneurysmal bone cyst (ABC). Several treatment strategies can be adopted. We report a challenging case of an osteoblastoma associated with ABC of the lumbar spine in a 2-year-old boy. The pathogenesis and the critical management of the disease are discussed.

  10. 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.

  11. A rare case of thrombotic microangiopathy triggered by acute pancreatitis.

    Science.gov (United States)

    Singh, Kevin; Nadeem, Ahmed Jamal; Doratotaj, Behzad

    2017-05-15

    Thrombotic microangiopathy (TMA) occurring after acute pancreatitis is rarely described. Without prompt intervention, TMA can be, and often is, lethal, so prompt recognition is important. Here, we present a case of a 61-year-old woman with a history of alcohol misuse who presented with epigastric pain, nausea and vomiting after binge drinking. Elevated serum lipase and imaging were suggestive of acute-on-chronic pancreatitis. Although the patient's symptoms of acute pancreatitis subsided, her anaemia, thrombocytopenia and acute kidney injury worsened. A peripheral blood smear revealed schistocytes, prompting suspicion for TMA. Therapeutic plasma exchange (TPE) was promptly initiated and she completed 10 TPE sessions that improved her anaemia and serum creatinine and resolved the thrombocytopenia. Since TPE was effective and the ADAMTS13 assay revealed 55% activity in the absence of anti-ADAMTS13 IgG prior to initiation of therapy, a confident diagnosis of TMA caused by acute pancreatitis was made. There was no evidence of relapse 2 years later. © BMJ Publishing Group Ltd (unless otherwise stated in the text of the article) 2017. All rights reserved. No commercial use is permitted unless otherwise expressly granted.

  12. A Rare Case of Noninsulinoma Pancreatogenous Hypoglycemia Syndrome

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    Jeffrey Nadelson

    2012-01-01

    Full Text Available As the obesity pandemic continues to worsen and medical interventions remain only moderately effective, bariatric surgery remains an important option for patients. In certain instances, patients who have undergone the procedure experience postprandial symptoms of neuroglycopenia caused by noninsulinoma pancreatogenous hypoglycemia syndrome (NIPHS. NIPHS is a recently described syndrome and is also very rare, and therapeutic approaches are still under debate. We report the case of a 51-year-old female who underwent Roux-en-Y gastric bypass and presented with episodic postprandial hypoglycemia 2 years after surgery. An insulinoma was absent from all abdominal imaging. Fasting C-peptide, insulin, and glucose were normal. Due to the possibility of NIPHS, clinical treatment was commenced with acarbose, leading to a significant reduction of hypoglycemic episodes. NIPHS occurs in approximately 0.5% to 7% of patients with hyperinsulinemic hypoglycemia. Sporadic hypoglycemia postgastric bypass is an important entity that should be understood by all surgeons and internists who are involved in postgastric bypass care.

  13. A Rare Case of an Irreducible Patella Dislocation

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    Dimitri E. Delagrammaticas

    2016-01-01

    Full Text Available Reports of irreducible patellar dislocations are exceedingly sparse throughout the literature. Obvious radiographic or physical exam findings including fracture or inversion of the patella are often present to explain the block to reduction. Not described previously in the literature is the instance of an irreducible patella dislocation in the setting of innocuous appearing injury imaging. We present a case of a healthy thirty-two-year-old female who sustained an irreducible lateral patella dislocation while participating in a dance aerobics class. Closed means of reduction were unsuccessful, necessitating open reduction. Intraoperative findings suggest incarceration of a nondisplaced fracture and a chondral defect as the block to reduction. Following open reduction, the patient has had no further episodes of pain or instability related to the patella at one-year follow-up. Irreducible patellar dislocations are exceedingly rare injuries, where associated osseous or chondral lesions may necessitate open reduction despite innocuous appearing initial imaging. A high index of suspicion to proceed with open reduction may limit repeated attempts at closed reduction and further injury.

  14. 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.

  15. A Rare Case of Meconium Periorchitis Diagnosed in Utero

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    Daigo Ochiai

    2015-01-01

    Full Text Available Meconium periorchitis is a rare disorder caused by fetal meconium peritonitis, with subsequent passage of meconium into the scrotum via a patent processus vaginalis. To date, clinical significance of meconium periorchitis for the prenatal diagnosis of meconium peritonitis and prediction for postnatal surgery remains to be determined. We present a clinical course of a fetus presenting with meconium periorchitis induced by meconium peritonitis. At 28 weeks’ gestation, fetal ultrasonography indicated fetal ascites associated with bilateral hydrocele and peritesticular calcification without other signs of meconium peritonitis. The pregnancy was uneventful until delivery and the infant was delivered at 37 weeks’ gestation. No abdominal distension was observed at birth, and radiography did not reveal any abdominal calcification except for scrotal calcification. Abdominal distension was observed 3 days after birth and laparotomy was performed. The diagnosis of meconium peritonitis was confirmed at surgery. Our case illustrated that careful examination of the scrotum during fetal life was helpful for prenatal diagnosis of meconium peritonitis as well as postnatal management.

  16. Case Histories of Four Extremely Intense Rockbursts in Deep Tunnels

    Science.gov (United States)

    Zhang, Chuanqing; Feng, Xia-Ting; Zhou, Hui; Qiu, Shili; Wu, Wenping

    2012-05-01

    In the process of excavating seven parallel tunnels at the Jinping II Hydropower Station, several extremely intense rockbursts occurred, killing and injuring construction workers and damaging several sets of equipment. Based on the characteristics and mechanisms of these rockbursts, four typical events were selected and their temporal and spatial characteristics were here described in detail. The geological conditions revealed after the rockbursts were surveyed carefully. The responses of support elements were also analyzed. The details documented in each case provide not only an important reference for understanding the development mechanisms of rockbursts but also a basis for the selection and development of rockburst prevention measures in deep hard rock tunnels.

  17. Clear cell sarcoma of soft tissue in right parapharyngeal region: report of a rare case.

    Science.gov (United States)

    Fan, Chuifeng; Yu, Juanhan; Yang, Lianhe; Lin, Xuyong; Wang, Enhua

    2015-01-01

    Clear cell sarcoma (CCS), initially named malignant melanoma of soft parts, is a rare malignant neoplasm typically involving deep soft tissue of the extremities, in close proximity to tendons and aponeuroses. Here we describe a case of clear cell sarcoma of the right parapharyngeal region in a young female aged 20 years. MRI detected a mass about 4.4 cm×3.4 cm×3.0 cm, located in the right parapharyngeal area and between the external pterygoid and the medial pterygoid. Microscopically, most of the tumor cells were epithelioid with palely eocinophilic cytoplasm arranged in sheets. Pleophorism of tumor cells were not marked. Immunohistochemical analysis shows that the tumor cells were positive for vimentin, S-100, HMB45 and MelanA, and negative for AE1/AE3, actin-sm, desmin, CD117, TFE-3, and P63. Ki67 index was about 5%.

  18. 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.

  19. 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.

  20. 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.

  1. 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.

  2. 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.

  3. A Rare Case of Subependymoma with an Atypical Presentation: A Case Report

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    Mohankumar Kurukumbi

    2011-10-01

    Full Text Available A rare case of subependymoma in a young patient presenting with sensory dysesthesia is reported. Computed tomography scan and magnetic resonance imaging revealed a posterior fossa mass occluding the fourth ventricle with infiltration to the right side immediately behind the pontine tegmentum and impinging on the right spinothalamic tract. Postoperative tumor histopathology revealed the classical appearance of subependymoma. Subependymoma is a rare, asymptomatic, slow-growing, low-grade glioma of the central nervous system. If symptomatic, the clinical features are commonly secondary to hydrocephalus, but subependymoma presenting with sensory dysesthesia has never been reported in the literature.

  4. 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.

  5. Lhermitte-Duclos disease associated to Cowden syndrome: de novo diagnosis and management of these extremely rare syndromes in a patient.

    Science.gov (United States)

    Gama, Ivo; Almeida, Leonor

    2017-01-30

    A 36-year-old woman, with history of cutaneous papilomatosis and thyroid carcinoma presented with headache, transitory visual blurring and nausea. Funduscopy showed papilloedema. MRI showed a tumour of the right cerebellar hemisphere with a striated, tigroid pattern, typical of Lhermitte-Duclos disease (LDD). Significant clinical and perimetric improvements were noted after surgery and the follow-up did not reveal recurrences of the tumour. LDD is an extremely rare differential diagnosis of posterior fossa tumours. LDD and the history of thyroid carcinoma permitted us to diagnose Cowden syndrome (CS). We present a clinical case that supports the possibility of performing a preoperative diagnosis of LDD based on MRI features. We review the diagnosis and management of LDD and CS. This report highlights the importance of excluding CS after LDD diagnosis, of monitoring the optic nerve postoperatively using optical coherence tomography and of prompt treatment that can potentially prevent visual function loss. 2017 BMJ Publishing Group Ltd.

  6. 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

  7. Immunosuppressant Medication-Induced Lower Extremity Pain After Combined Liver and Kidney Transplant: A Case Report.

    Science.gov (United States)

    Mohideen, Thanzeela Kausar; Wu, Hong

    2018-03-01

    Calcineurin inhibitors are imperative in the success of a transplanted organ. However, these immunosuppressants can lead to a rare complication known as calcineurin inhibitor-induced pain syndrome, which may not be recognized early and managed appropriately. We present a case of a 35-year-old woman who underwent a combined liver/kidney transplant and developed lower extremity pain while being maintained on tacrolimus. This case illustrates a patient with previously reported characteristic clinical features of calcineurin inhibitor-induced pain syndrome in addition to uncharacteristic neuropathic symptoms and imaging findings. The patient was treated successfully with gabapentin, calcitonin nasal spray, and acupuncture. Early recognition of this syndrome can help improve a patient's quality of life. V. Copyright © 2018 American Academy of Physical Medicine and Rehabilitation. Published by Elsevier Inc. All rights reserved.

  8. Plexiform neurofibroma in the submandibular gland along with small diffuse neurofibroma in the floor of the mouth but without neurofibromatosis-1: a rare case report

    OpenAIRE

    Kamra, Hemlata T; Dantkale, Sunita S; Birla, Khushboo; Sakinlawar, Pankaj W; Bharia, Paresh H

    2013-01-01

    Plexiform neurofibroma is more commonly seen in the orbit, neck, back, and inguinal region. It is extremely rare in the submandibular gland. These lesions rarely transform into malignancy but are locally infiltrative and can lead to haemorrhage. Therefore, plexiform neurofibroma should always be considered during differential diagnosis while excising a submandibular gland mass. We present here a case of plexiform neurofibroma in the submandibular gland and diffuse neurofibroma in the floor of...

  9. 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.

  10. Non-Syndromic Familial Keratocystic Odontogenic Tumour: A Rare Case Report in Japanese Identical Twins

    Science.gov (United States)

    Maruoka, Yutaka; Yamaji, Iena; Kawai, Shigeo

    2016-01-01

    Keratocystic Odontogenic Tumour (KCOT) is unicystic or multicystic intraosseous benign tumour of odontogenic origin that recurs due to locally destructive behaviour. KCOTs are usually the first manifestation of Nevoid Basal Cell Carcinoma Syndrome (NBCCS), an autosomal dominant disorder also known as Gorlin’s syndrome and they are most frequently observed familial symptom regardless of patients’ nationality. In addition, the recurrence rate and multiplicity of KCOTs is relatively high as compared to that of other sporadic carcinomas. KCOT has been considered as a non-hereditary lesion and its familial onset is an extremely rare event in non-NBCCS cases. Here, we describe previously unreported non-syndromic multiple KCOT cases in identical twins in a Japanese family. The subjects were female Japanese identical twins who were 26 and 27 years old, respectively, at the time of diagnosis for KCOT. They had no major or minor features of NBCCS other than KCOT. Although there were lesions that were likely to be dentigerous cysts based on radiographic findings, one of them was KCOT. This case report highlights the importance of precise diagnosis, choice of surgical method and careful observation for multiplicity or familial onset in sporadic KCOT cases without NBCCS. PMID:27656582

  11. Rare case of transverse testicular ectopia – Case report and review of literature

    Directory of Open Access Journals (Sweden)

    Vinod Raj

    Full Text Available This case report has been reported in line with the SCARE criteria; Consensus-based surgical case report guidelines of International Journal of Surgery 2016. Introduction: 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. Case presentation: 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. Discussion: 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. Conclusion: 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. Keywords: Transverse testicular ectopia, Case report, Persistent mullerian duct syndrome, Laparoscopy, Trans-septal orchidopexy and trans-septal contralateral orchidopexy

  12. Complex and compound odontomas: Analysis of 69 cases and a rare case of erupted compound odontoma.

    Science.gov (United States)

    Bereket, C; Çakır-Özkan, N; Şener, İ; Bulut, E; Tek, M

    2015-01-01

    The purpose of this study was to analyze clinic and radiologic features of the 69 odontoma cases and present a rare case of erupted compound odontoma in the oral cavity. The 69 cases were analyzed with regard to the following parameters: age, gender, location, associated unerupted teeth, missing teeth, radiological and histopathological features and treatment protocols. Of the 69 cases, 49 were compound odontoma and 20 were complex odontoma. There was a female predilection for both compound and complex odontomas. Compound odontoma occurred more often in the anterior region of the jaws; complex lesions occurred more often at the posterior mandible. The most common clinical manifestations were the retention of permanent teeth. Compound odontomas rarely erupt into the mouth. The presented case is the 13 th case of erupted compound odontoma reported in the literature. The treatment of choice is surgical removal of the odontoma. In the case of odontomas associated to impacted teeth, the teeth should be preserved in wait of spontaneous eruption, or alternatively fenestration followed by orthodontic traction is indicated. Regular follow-up period is crucial to evaluate the prognosis of these teeth.

  13. 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.

  14. A Rare Case Report: A Giant Angiomyolipoma Located in the Small Intestine

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    Sevilay Gürcan

    2015-04-01

    Full Text Available An angiomyolipoma is a mesenchymal neoplasm of the tumor and is composed of a varying heterogeneous mixture of blood vessels, smooth muscles, and adipose cells. Extra-renal angiomyolipomas are rarely seen and are most commonly found in the liver. Angiomyolipomas of the small intestine are extremely rare. We report the case of a 32-year-old man who had an ileal angiomyolipoma and who clinically presented with weakness and abdominal pain. A computed tomography scan of the abdomen showed a massive lesion, and segmental resection of the small intestine was performed. In the microscopic examination of the material that was sent for pathological evaluation, a tumor that included a cystic dilated vascular structure, adipose tissue, and muscular tissue extending from the serosa to the mucosa was seen. On immunohistochemical staining, various regions of the tumor were stained positive by actin, desmin, vimentin, CD31, CD34, and D2-40. With these histopathological findings, the patient was diagnosed with angiomyolipoma of the small intestinal mesentery.

  15. A rare case of anti-jk3 antibody detected on pre-transfusion investigation.

    Science.gov (United States)

    Yousuf, Rabeya; Abdul Aziz, Suria; Yusof, Nurasyikin; Leong, Chooi-Fun

    2014-09-01

    We report a 47-year-old Malay lady, para 4 + 1, with known medical history of hypertension whom presented at Emergency Department with severe anaemia, most likely secondary to menorrhagia caused by uterine fibroids. Her haemoglobin was 5.5 g/dl and she was transfused with three units of packed cell without any adverse reaction, her haemoglobin level increased to 9.8 g/dl. She was then planned for total abdominal hysterectomy and bilateral salpingo-oophorectomy later. Four months later when she came for the elective surgery, her pre transfusion investigations showed blood group as B Rh D positive, with a probable R1R1 phenotype. Her antibody screening was positive in all the three panel cells. Further testings showed a negative Direct Coomb's test and negative autocontrol, antibody identification showed pan-agglutination reaction on all 11 panel cells with enzyme enhancement. Patient's red cell phenotype was Jk(a-b-). Anti-Jk3 was suspected and further confirmed in the reference laboratory by phenotyping as well as negative urea lysis test. This case report highlights an extremely rare but clinically significant anti-JK3 antibody detected during pretransfusion testing. This phenotype is rare in the white population, more commonly seen in various polynesians. Increased awareness among the blood bank personnel regarding the variability of the blood group phenotype and the capricious nature of the Kidd antibodies may contribute to the better management of these patients.

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

    Science.gov (United States)

    Nadrowski, Karin; Pietsch, Katherina; Baruffol, Martin; Both, Sabine; Gutknecht, Jessica; Bruelheide, Helge; Heklau, Heike; Kahl, Anja; Kahl, Tiemo; Niklaus, Pascal; Kröber, Wenzel; Liu, Xiaojuan; Mi, Xiangcheng; Michalski, Stefan; von Oheimb, Goddert; Purschke, Oliver; Schmid, Bernhard; Fang, Teng; Welk, Erik; Wirth, Christian

    2014-01-01

    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.

  17. 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.

  18. 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.

  19. Coexistence of true talon cusp and double dens invaginatus in a single tooth: a rare case report and review of the literature.

    Science.gov (United States)

    Nu Nu Lwin, Hnin; Phyo Kyaw, Pyae; Wai Yan Myint Thu, Sai

    2017-12-01

    Co-occurrence of a talon cusp and double dens invaginatus is an extremely rare developmental dental anomaly. This case report represents a talon cusp with two dens invaginatus on a maxillary right lateral incisor. Early identification is needed for prevention of potential problems on the affected or opposing tooth.

  20. A rare case of Silver-Russell syndrome associated with growth hormone deficiency and urogenital abnormalities

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    Namburi Rajendra Prasad

    2012-01-01

    Full Text Available Introduction: Silver-Russell syndrome (SRS is a very rare genetic disorder. This is usually characterized by asymmetry in the size of the two halves or other parts of the body. Background: We are presenting a case of SRS with growth hormone (GH deficiency and urogenital abnormalities. Case Report: A 15-year-old boy a product of non-consanguineous marriage brought with a history of short stature and poor development of secondary sexual characters. There were no adverse perinatal events, but weighed 1500 g (<3 rd centile at birth. He had delayed developmental milestones. He has had a poor appetite and feeding difficulties since childhood. On physical examination, he had a broad forehead, triangular facies, and low-set prominent ears. Asymmetry of the face, upper and lower extremities was noted, with hemihypertrophy on the right side. His height was 119 cm (<3 rd centile and weight was 18 kg which were low (<3 rd centile as per his age. He was biochemically euthyroid and GH stimulation testing with clonidine (0.15 mg/m 2 showed low GH levels at 30×, 60×, and 90× were 1.7, 1.6, and 1.1ng/ml, respectively. On micturatingcystourethrogram, grade V complex was noted on the right side. Dimercaptosuccinic acid (DMSA showed normal functioning kidneys. He was started on recombinant GH with a height velocity of 10 cm/ year. Conclusion : Urogenital abnormalities are rare but well described anomalies associated with SRS, and all cases have to be screened for them. GH deficiency is not uncommon in SRS, and GH treatment proves to be beneficial.

  1. Sclerosing epithelioid fibrosarcoma as a rare cause of ascites in a young man: a case report

    Directory of Open Access Journals (Sweden)

    Smith Philip J

    2008-07-01

    Full Text Available Abstract Introduction Sclerosing epithelioid fibrosarcoma is a rare but distinct variant of fibrosarcoma that not only presents as a deep-seated mass on the limbs and neck but can also occur adjacent to the fascia or peritoneum, as well as the trunk and spine. We report the case of an intra-abdominal sclerosing epithelioid fibrosarcoma, which to best of the authors' knowledge has not been described previously. The patient discussed here developed lung metastases but is still alive 1-year post-diagnosis. Case presentation A 29-year-old man presented with a 2-week history of progressive abdominal distension and pain and was found to have marked ascites. A full liver screen was unremarkable with abdominal and chest computed tomography scans only confirming ascites. After a diagnostic laparotomy, biopsies were taken from the greater omentum and peritoneal nodules. Histopathology revealed a malignant tumour composed of sheets and cords of small round cells set in collagenized stroma. After further molecular investigation at the Mayo Clinic, USA, the diagnosis of a high-grade sclerosing epithelioid fibrosarcoma was confirmed. Conclusion Sclerosing epithelioid fibrosarcoma is an extremely rare tumour, which is often difficult to diagnose and which few pathologists have encountered. This case is particularly unusual because of the intra-abdominal origin of the tumour. Owing to the rarity of sclerosing epithelioid fibrosarcoma, there is no clear evidence regarding the prognosis of such a tumour, although sclerosing epithelioid fibrosarcoma is able to metastasize many years post-presentation. It is important that physicians and pathologists are aware of this unusual tumour.

  2. 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.

  3. Spinal Rosai–Dorfman disease: Case report of a rare disorde

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    A. Elsotouhy

    2015-12-01

    Conclusion: This is a rare case of spinal Rosai–Dorfman Disease with epidural and intradural components causing cord compression. To our knowledge, this represents the first case of combined epidural and intradural extramedullary lesions in the literature.

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

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    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.

  5. Case Report: Prune perineum syndrome: a rare case with an unfavourable outcome

    OpenAIRE

    Lopes, Roberto I.; D?nes, Francisco T.; Messi, Gustavo B.; Machado, Marcos G.

    2016-01-01

    Prune perineum syndrome (PPS) is a rare anomaly, with only two previous case reports, both dying in the perinatal period. We report the first case of PPS that reached childhood. The patient presented with a hypoplastic genitalia and bilateral cryptorchidism. There was no evidence of an anal orifice. A significant prune-like mass was observed, extending from the perineum to both gluteal regions and to a cephalic mid-line bony prominence, with a 1cm central orifice that discharged urine. MRI co...

  6. CASE REPORT- Vaginal Leech Infestation: A Rare Cause of ...

    African Journals Online (AJOL)

    BACKGROUND: Human leech infestation is a disease of the poor who live in rural areas and use water contaminated with leeches. Like any other body orifices, vagina can also be infested by leech when females use contaminated water for bathing and/or douching. Although this condition is very rare in postmenopausal ...

  7. Tubercular Sinus of Labia Majora: Rare Case Report

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    Kela Manoj

    2008-01-01

    Full Text Available Tuberculosis of the female external genitalia is unusual and primary infection is rare. We report a 50-year-old female patient admitted to Department to Surgery with swelling over left inguinal area with discharging sinus from labia majora to left inguinal crease which was found to be tubercular sinus on histopathology.

  8. Management of supernumerary testis: a rare case of polyorchidism ...

    African Journals Online (AJOL)

    this benefit against the risk for testicular cancer. Taken together, the management of polyorchidism remains controversial with some advocating definitive orchidectomy, whereas others endorse a close follow-up and self- examination to maximize future fertility. Polyorchidism is rarely encountered in children less than 5 ...

  9. 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.

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

    African Journals Online (AJOL)

    KIGZ

    Necrotizing fasciitis can readily be confused with Meleney's gangrene, but it is a much more acute and highly toxic infection which causes widespread necrosis and undermining of the surrounding tissues (10). The main distinguishing feature is the extensive necrosis of the superficial fascia (10). Conclusion. Though rare ...

  11. Kindler′s syndrome: A rare case report

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    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.

  12. 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 ...

  13. A rare association of major congenital malformations : a case report

    African Journals Online (AJOL)

    hypoplasia, cleft palate, pulmonary hypoplasia, imperforate anus and defects of the caudal. extremitiesI0. This woman used very high doses of Hedex in the first trimester to relieve headache and the symptoms of early pregnancy. Hedex is a mixture of paracetamol, caffeine and aspirin. The manufacturers recommend use of ...

  14. Intrauterine Extremity Gangrene and Cerebral Infarction at Term: A Case Report

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    M. Tanvig

    2011-01-01

    Full Text Available Intrauterine extremity gangrene in combination with cerebral infarction is a serious and rare event. We present a case with a healthy mother who gave birth to a child with this condition. At term, the mother presented at the antenatal clinic with decreased fetal movements. Cardiotocography (CTG showed signs of fetal distress and a caesarean section was performed. The left arm of the newborn was found gangrenous. Amputation of the arm was necessary and the child was subsequently treated with anticoagulant therapy due to thrombosis and cerebral infarction in the left hemisphere found by magnetic resonance imaging (MRI. At one year of age the boy was doing well and had prosthesis as a left arm. He had no signs of further complications. Despite thorough examination of the parents and the child, the reason for the thrombosis is still unknown.

  15. Spontaneous Upper Extremity Venous Thrombosis in a Collegiate Soccer Player: A Case Report.

    Science.gov (United States)

    Fundora, Michael P; Rudnick, Chad; Barbur, Carlos

    2016-01-01

    Spontaneous effort-induced thrombosis is a rare but reported phenomena that was originally described over 100 years ago. The pathogenesis of this thrombosis arises from an abnormality of the thoracic outlet usually combined with a history of physical activity that includes repetitive arm motions, usually of the dominant hand. We present the case of an adolescent patient who presented to a pediatric emergency department with progressive pain, discoloration, and swelling of the shoulder of his nondominant hand. The pain became acutely worse with graying appearance of his arm. The patient was diagnosed with spontaneous thrombosis of the upper extremity extending from the left subclavian vein extending to the axillary vein. Treatment of this patient included aggressive anticoagulation, thrombolysis, and costectomy.

  16. Triple dens invaginatus in a single tooth: Rarest of rare case report.

    Science.gov (United States)

    Chhina, Amandeep; Chhina, Kamalpreet; Kaushal, Nitin; Walia, Ira

    2017-01-01

    Dens invaginatus is a rare malformation affecting the teeth. The teeth that are most commonly affected are the permanent maxillary lateral incisors. The mandibular posterior teeth are rarely affected by dens invaginatus. In most of the cases, a single dens invaginatus is seen affecting the tooth. Cases of double dens invaginatus affecting a single tooth are rare, and those of triple dens invaginatus in a single tooth are even rarer. Here, we present a case of a 14-year-old where three dens invaginatus were seen in the mandibular second premolar. This is the fourth case of three dens invaginatus in a single tooth as only three have been reported previously.

  17. A RARE VARIANT OF TURNER SYNDROME- CASE REPORT

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    Ipsita Mishra

    2017-10-01

    Full Text Available Turner syndrome occurs in one out of every 2500-3000 live female births and the diagnosis is usually based on the clinical presentation. It is a genetic condition in which a female does not have the usual pair of two X chromosomes. Deletions of proportions of the X chromosome result in various Turner variants who have varied spectrum of clinical presentation. We report on a rare variant of deletion on long arm of X chromosome in a 35-year-old female with short stature, lack of secondary sexual characters, primary amenorrhea, average intelligence and diabetes mellitus. Chromosomal analysis using GTG-banding showed 46, X, del (X, (q13 in all cell lines. Hence, suspicion of rare variants of Turner syndrome in females must be done who present at a later age with atypical features.

  18. Right sided congenital diaphragmatic hernia: A rare case report

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    Amit Narkhed, Shrikhande DY, Prasant Nigwekar, Santosh Yadav, Haresh Kasodariya

    2014-01-01

    Full Text Available A diaphragmatic hernia is defined as a communication between abdominal and thoracic cavity with or without abdominal contents in the thorax. The true incidence of Congenital diaphragmatic hernia is 1 in 5000 live births while right side diaphragmatic hernia (15% is rare comparing to left side diaphragmatic hernia (85% because liver plugs the opening. Congenital diaphragmatic hernia typically refers to Bochdalek form, other forms are rarer. Despite advances in neonatal intensive care, congenital diaphragmatic hernia is associated with high mortality and morbidity. The posterolateral right congenital DH is a rare diaphragmatic defect. Females are twice affected than that of males. The symptoms are non characteristic and patients with this disease maybe without symptoms for a long period. The main tool for diagnosis of congenital DH is radiography. Surgical correction is required.

  19. A rare case of chronic lymphocytic leukemia/small lymphocytic lymphoma presenting in the thyroid gland.

    Science.gov (United States)

    Shin, Joyce; Chute, Deborah; Milas, Mira; Mitchell, Jamie; Siperstein, Allan; Berber, Eren

    2010-09-01

    Lymphoma involving the thyroid gland is rare. Diffuse large B-cell lymphoma and mucosa-associated lymphoid tissue lymphoma are the two most common histologic subtypes of primary thyroid lymphoma. Chronic lymphocytic leukemia/small lymphocytic lymphoma (CLL/SLL) presenting initially as a thyroid abnormality is extremely rare, with very few reported cases in the literature. We report a case of a patient with a long history of Hashimoto's thyroiditis and goiter who presented with a recent enlargement of her thyroid gland. The sonographic finding of a distinct thyroid nodule in the heterogeneous background of chronic lymphocytic thyroiditis led to the performance of a fine-needle aspiration biopsy and flow cytometry, with a high index of suspicion for thyroid lymphoma. Subsequent surgical removal of the thyroid gland, prompted by the patient's history of head and neck radiation, confirmed the diagnosis of CLL/SLL. The patient's systemic illness was recognized only after the management of her thyroid disease. Although thyroiditis has long been associated with lymphoma arising in the thyroid gland, CLL/SLL involving the thyroid has not been linked to chronic lymphocytic thyroiditis. Therefore, the patient also had coexisting thyroiditis. Due to the rarity of thyroid lymphomas, our experience in the detection and management of this disease is limited. Primary thyroid lymphoma should be suspected in a patient with a history of chronic lymphocytic thyroiditis presenting with a rapidly enlarging neck mass. The initial diagnostic method for thyroid lymphoma should consist of a fine-needle aspiration biopsy with the use of ancillary techniques such as flow cytometry and immunohistochemistry for improved diagnostic accuracy. Although controversial, the treatment of thyroid lymphoma is typically guided by the histologic subtype and extent of disease. CLL/SLL is one of the rarest subtypes of lymphoma that can involve the thyroid gland. Diagnosis of this entity is difficult

  20. Thanatophoric Dysplasia; a Rare Case Report on a Congenital Anomaly

    OpenAIRE

    Maria Francis Yuvaraj; Ponuswamy Kasirajan Sankaran; Gunapriya Raghunath; Zareena Begum Kumaresan; Kumaresan kumaresan M

    2017-01-01

    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...

  1. Werewolf syndrome associated with gingival fibromatosis: A rare case report

    OpenAIRE

    Mysore K Sunil; Ashwarya Trivedi; Saloni Arora; Sonam Gupta

    2016-01-01

    Hypertrichosis is a rare disorder that causes abnormal excessive body hair growth. Hypertrichosis has informally been called werewolf syndrome because the appearance is similar to that of a werewolf. It is often associated with additional anomalies including gingival hyperplasia, deafness, cardiomegaly and bone abnormalities. The association of gingival fibromatosis and a coarse facies could further worsen the esthetics. Thus, a multidisciplinary approach involving a psychologist, a dentist a...

  2. 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.

  3. Aceruloplasminemia: a rare disease - diagnosis and treatment of two cases

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    Maria do Rosário Ferraz Roberti

    2011-10-01

    Full Text Available Aceruloplasminemia is a rare autosomal recessive disease in which a mutation leads to the absence or dysfunction of ceruloplasmin. Deficiency of this enzyme leads to the accumulation of iron in various organs; aceruloplasminemia is usually characterized by diabetes, retinal degeneration and neurological disorders. Diagnosis is suspected by the presence of elevated levels of ferritin, anemia, decreased serum copper and absence of ceruloplasmin in serum. Treatment of aceruloplasminemia is mainly based on the control of iron overload.

  4. Uterus-like mass: A very rare and elusive entity a case report

    Science.gov (United States)

    He, Jian; Xu, Jie; Zhou, Hong-Yan

    2016-01-01

    Abstract Background: Uterus-like mass (ULM) is an extremely rare lesion. Gross morphology of ULM resembling a uterus. It can occur in various organs in the abdominal cavity, even in the spinal cord. The histogenesis of ULM remains uncertain. A number of hypotheses have been proposed including metaplasia, congenital anomaly, and heterotopia theory. Methods: We describe a case of 43-year-old male presented with a complaint of acute low abdominal pain. Pelvic ultrasound found a large pelvic mass embedded in the broad ligament. Results: The mass contains a variable thickness smooth muscle layer lined with endometrial glands and stroma which resembling a uterus. Eventually, the patient was diagnosed as ULM by histopathological examination. Conclusion: Except hypomenorrhea, the patient did not have any other associated abnormalities. We suggest this case supports the metaplasia theory that ULM is a benign mass formed by the proliferation of ectopic endometrial stromal cells or pluripotent mesenchymal cells of the 2nd Müllerian system. PMID:27684842

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

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    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.

  6. 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.

  7. 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 ...

  8. 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 ...

  9. Appendiceal Endometriosis and Carcinoid Presented as Acute Appendicitis in Pregnancy: A Rare Case Report and Review of the Literature

    Directory of Open Access Journals (Sweden)

    Panagiotis A. Dimitriadis

    2013-01-01

    Full Text Available A 22-year-old pregnant woman presented at the twenty-seventh week of gestation in the Emergency Department with acute abdominal pain and right iliac fossa tenderness. Urgent MRI was done and was suggestive of acute appendicitis. A laparoscopy was performed that confirmed an inflamed and purulent appendix that was removed. The technique used is described in detail. The histopathologic findings were those of acute appendicitis, carcinoid, and endometriosis of the appendix. We report the first case of this extremely rare triad presented in pregnancy.

  10. Rare presentation of pediatric acute promyelocytic leukemia as multiple lytic bone lesions: Case report and review of literature

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    Manjusha Nair

    2014-01-01

    Full Text Available Acute promyelocytic leukemia (APL is an uncommon malignancy in the pediatric population, accounting for only 5-10% of pediatric acute myeloid leukemias, and for this disease to present with bone lesions at diagnosis is extremely unusual. We wish to convey that very rarely, in a pediatric cancer patient presenting with multiple extensive lytic bone lesions, the diagnosis can be APL. The treatment protocol and prognostic implications are vastly different. Histopathology is the gold standard in arriving at a correct diagnosis and delivering proper treatment in such cases. This patient had excellent response to chemotherapy.

  11. A rare case of concomitant cervical disc herniation and intradural meningioma treated with one-stage posterior surgery.

    Science.gov (United States)

    Hu, Xiaojian; Chen, Zhong; Wang, Yue

    2017-12-14

    Case report. To present a rare case of cervical disc herniation concomitant with intradural meningioma which was successfully treated using a single one-stage posterior surgery of tumor resection and transdural discectomy. Coexistence of symptomatic disc herniation and intra-spinal tumor in the same cervical segment is extremely rare. Usually, two-stage anterior and posterior surgeries are needed to treat two conditions, respectively. One-stage posterior surgery to treat two pathologies simultaneously has not been reported in the literature. A 76-year-old man presented with leg weakness and numbness for 6 months and left arm pain for 2 months. Contrast MR imaging revealed C3/4 intervertebral disc herniation and a hyperintense intradural lesion at the right portion of C3 canal. A one-stage posterior surgery, including C3/4 laminectomy, intradural tumor resection, transdural C3/4 discectomy, and C3/4 lateral mass instrumentation and fusion, was performed to treat two distinct pathologies together. The patient's arm pain and numbness disappeared right after the surgery and symptoms of myelopathy fully recovered at 6-month follow-up. Histological studies confirmed a herniated disc and a meningioma. In rare case, intradural tumor coexists with cervical disc herniation. When suspicious findings were noticed, or clinical symptoms cannot be fully explained, contrast MR imaging is helpful in differential diagnosis. Microscopic transdural discectomy is safe, and could be used as an optional procedure for cervical disc herniation in some cases.

  12. The inside mystery of jejunal gastrointestinal stromal tumor: a rare case report and review of the literature.

    Science.gov (United States)

    Dhull, A K; Kaushal, V; Dhankhar, R; Atri, R; Singh, H; Marwah, N

    2011-01-01

    Gastrointestinal stromal tumors (GISTs) are malignant and rare form of soft tissue sarcoma of the digestive tract. The incidence of gastrointestinal stromal tumors is very low Kramer et al. 2005 Jejunal GISTs are extremely rare. Here we present a rare case of jejunal GIST with unusually large size at presentation. The patient presented with severe abdomen pain, exophytic growth, and dimorphic anemia. Surgical resection of the tumor was carried out, and operative findings revealed a 15 × 10 cm growth, arising from serosal surface of jejunum, at the antimesenteric surface. Diagnosis in this case was made by subjecting the resected specimen to immunohistochemical analysis. In view of large size of the resected tumor, and high-risk histopathological features, imatinib mesylate 400 mg once daily was given as adjuvant chemotherapy. Patient is asymptomatic without any evidence of tumor recurrence after six months of postoperative followup. Imatinib as such is recommended in metastatic, residual or recurrent cases of GISTs or which are surgically not removable; however, recent recommendations suggests the use of imatinib mesylate after radical surgery in high-risk cases, because it has shown a significant decrease in the recurrence rate, and the Food and Drug Administration (FDA) has also approved the use of imatinib as adjuvant therapy after complete resection of localized, primary GIST.

  13. The Inside Mystery of Jejunal Gastrointestinal Stromal Tumor: A Rare Case Report and Review of the Literature

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    A. K. Dhull

    2011-01-01

    Full Text Available Gastrointestinal stromal tumors (GISTs are malignant and rare form of soft tissue sarcoma of the digestive tract. The incidence of gastrointestinal stromal tumors is very low Kramer et al. 2005 Jejunal GISTs are extremely rare. Here we present a rare case of jejunal GIST with unusually large size at presentation. The patient presented with severe abdomen pain, exophytic growth, and dimorphic anemia. Surgical resection of the tumor was carried out, and operative findings revealed a 15 × 10 cm growth, arising from serosal surface of jejunum, at the antimesenteric surface. Diagnosis in this case was made by subjecting the resected specimen to immunohistochemical analysis. In view of large size of the resected tumor, and high-risk histopathological features, imatinib mesylate 400 mg once daily was given as adjuvant chemotherapy. Patient is asymptomatic without any evidence of tumor recurrence after six months of postoperative followup. Imatinib as such is recommended in metastatic, residual or recurrent cases of GISTs or which are surgically not removable; however, recent recommendations suggests the use of imatinib mesylate after radical surgery in high-risk cases, because it has shown a significant decrease in the recurrence rate, and the Food and Drug Administration (FDA has also approved the use of imatinib as adjuvant therapy after complete resection of localized, primary GIST.

  14. Thanatophoric Dysplasia; a Rare Case Report on a Congenital Anomaly

    Directory of Open Access Journals (Sweden)

    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.

  15. 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.

  16. Urinary Ascites in Newborn – A Rare Case Report

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    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.

  17. A rare case of congenital insensitivity to pain with anhydrosiss

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    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.

  18. BILATERAL UNICONDYLAR HOFFA FRACTURE : A RARE CASE REPORT

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    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

  19. A rare case of lymphangioleiomyomatosis with recurrent pneumothorax

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    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.

  20. PRIMARY GIANT HYDATID DISEASE OF THE SPLEEN: A RARE CASE REPORT WITH REVIEW OF LITERATURE

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    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

  1. A rare case of familial reactive perforating collagenosis

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    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.

  2. Morgagni hernia: A rare case report and review of literature

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    Manoj Kumar Pattnaik

    2016-01-01

    Full Text Available Morgagni hernias (MHs are rare and constitute about 2% of all diaphragmatic hernias. Although uncommon, it has potential for considerable morbidity if the diagnosis is missed. An elderly woman with known history of chronic asthma and constipation presented to us with vague right-sided chest pain. General physical examination was unremarkable and coincidentally diagnosed to have diabetes mellitus. Chest roentgenogram posteroanterior view revealed a right paracardiac opacity and right lateral view showed the opacity in the peridiaphragmatic area of anterior mediastinum. Computed tomographic scan of the chest and abdomen revealed a right-sided MH containing omental fat. Standard right posterolateral thoracotomy was done, and there was a rent at the medial end of the xiphoid process with hernia sac containing the omentum, which was compressing adjacent lungs and heart. The sac was opened; redundant omentum was resected, and rent closed with intercostal muscle with prolene. MH being rare must be addressed with appropriate investigation to prevent unnecessary morbidity and mortality.

  3. Mast Cell Leukemia: Review of a Rare Disease and Case Report of Prolonged Survival after Allogeneic Stem Cell Transplant

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    James Bauer, MD, PhD

    2017-11-01

    Full Text Available Mast cell leukemia is a rare and aggressive form of mastocytosis characterized by >20% mast cells found in the bone marrow aspirates of patients with signs of systemic mastocytosis-related organ damage. The prognosis for patients with mast cell leukemia is extremely poor, with resistance to both cytoreductive therapies and tyrosine kinase inhibitors being relatively common. While allogeneic hematopoietic stem cell transplantation has been associated with long-term survival in patients with advanced systemic mastocytosis, reports regarding its effectiveness in mast cell leukemia are limited to fewer than 20 cases described in the literature. Here, we report a patient with mast cell leukemia who remains in complete remission 24 months after allogeneic HSCT at the time of this writing, and briefly review the clinical, diagnostic, and therapeutic approaches to this rare disease.

  4. Intimal aortic sarcoma mimicking ruptured thoracoabdominal type IV aneurysm. a rare case report and review of the literature

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    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.

  5. A rare case of Osteogenesis Imperfecta Type III

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    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.

  6. A Rare Case Report of Spontaneous Resolution of Hepatic Portal ...

    African Journals Online (AJOL)

    should be performed.[7] There is a paucity of data describing the radiographic findings of cocaine‑induced intestinal ischemia. Our case report describes the unusual presentation of the portal vein air related to recreational cocaine use, which resolved within a few hours with just conservative treatment alone. Case Report.

  7. 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 ...

  8. Rare Locations of Epidermoid Cyst: Case Reports and Review ...

    African Journals Online (AJOL)

    The PubMed database search made on January 2016 yielded 674 articles of epidermoid cyst in the head and neck region. In that there is only one case reported occuring in the lower one third of face. In the current report, we outline the second case of epidermoid cyst localized to the right mandibular body region without ...

  9. Primary squamous cell carcinoma of stomach: A rare entity - case ...

    African Journals Online (AJOL)

    Very few case reports of pure squamous cell carcinoma (SCC) of stomach are available in the world literature. The exact pathology of this uncommon carcinoma in stomach remains unknown. This is an additional case report of SCC in an elderly female arising in the gastric antrum. She underwent distal gastrectomy, ...

  10. Bilateral Paratesticular Liposarcoma ‑ A Rare Case Report

    African Journals Online (AJOL)

    Intraoperative diagnosis of testicular tumor was made. High inguinal orchiectomy was done. Histopathological examination revealed it to be liposarcoma of the cord. To our knowledge, there is no reported case of bilateral paratesticular liposarcoma in English literature, hence we report this case. Key words: Epididymis ...

  11. Dumb-bell in the heart: rare case of biatrial myxoma with mitral regurgitation.

    Science.gov (United States)

    Ananthanarayanan, Chandrasekaran; Bishnoi, Arvind Kumar; Ramani, Jayadip; Gandhi, Hemang

    2016-10-01

    Cardiac myxomas are rare intracardiac tumors, and the majority are benign myxomas involving the left atrium. We report a case of the very rare occurrence of biatrial myxoma associated with mitral regurgitation, which was successfully treated. © The Author(s) 2016.

  12. 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

  13. 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.

  14. 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.

  15. 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.

  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. Report of a Rare Case of an Odontogenic Myxoma of the Maxilla ...

    African Journals Online (AJOL)

    Report of a Rare Case of an Odontogenic Myxoma of the Maxilla and Review of Literature. SM Manjunath, AA Gupta, P Swetha, NJ Moon, S Singh, A Singh. Abstract. Odontogenic myxoma (OM) is a mesenchymal tissue benign neoplasia, being relatively rare which is almost exclusively seen in tooth‑bearing areas.

  18. 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.

  19. 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.

  20. 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.

  1. 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.

  2. Periosteal Osteoblastoma of the Pelvis: A Rare Case

    Directory of Open Access Journals (Sweden)

    Swaroop Patel

    2015-01-01

    Full Text Available Among the rare bone tumors, the osteoblastoma is a fascinating tumor. The rarity, the predisposition to occur in any bone and the diagnostic dilemma makes this infrequent tumor interesting. It is sporadically reported in the literature and what is rarer is its occurrence in the pelvis. The unusual location and inconclusive radiographic findings with diffused diagnostic evidences delays the management of benign osteoblastoma. We encountered a patient with benign osteoblastoma of the pubic ramus of right side. An excisional biopsy was performed. Peroperatively, the tumor appeared as oval, reddish brown, bony hard mass lying just over the cortex of the right pubic ramus and not breaching the cortex. Histopathological study revealed an osteoid rich lesion. Its presence in pubis must not be ignored and periosteal osteoblastoma should be considered as a differential diagnosis.

  3. 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.

  4. 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

  5. A rare case of myxoid liposarcoma of the adult foot diagnosed using fine needle aspiration cytology (FNAC

    Directory of Open Access Journals (Sweden)

    Kure S

    2014-12-01

    Full Text Available Shoko Kure,1 Wei-Xia Peng,1 Mitsuhiro Kudo,1 Miyuki Matsubara,2 Takashi Tsunoda,3 Zenya Naito1,21Department of Integrated Diagnostic Pathology, Nippon Medical School, 2Department of Pathology, Nippon Medical School Hospital, 3Department of Orthopedics, Nippon Medical School Department of Orthopedics, Nippon Medical School, Tokyo, JapanAbstract: Liposarcoma is categorized as a soft tissue sarcoma that most commonly appears in the lower extremities during adulthood, but rarely in the feet. We present a rare case of a primary myxoid liposarcoma in the foot of a 63-year-old man that was diagnosed by fine needle aspiration cytology (FNAC. The patient presented to our hospital with a 10-year history of a slow-growing mass on the left lateral ankle. On physical examination, the 60 mm ×60 mm mass, was found to be soft and elastic, causing poor mobility without pain. Magnetic resonance imaging of the mass revealed high signal intensity on T1-weighted images and heterogeneously high signal intensity on T2-weighted images without fat suppression. As a result of the physical examination and imaging, the mass was suspected to be a lipoma or ganglion cyst. However, the FNAC procedure revealed atypical small, round, short spindle cells, or foamy cells with a myxoid background, suggesting myxoid liposarcoma. Histology using an open biopsy showed a proliferation of atypical spindle and asteroid cells with a few foamy lipoblasts in a capillary-rich and myxoid background. The atypical lipoblasts were slightly positive for S-100. These findings were consistent with myxoid liposarcoma. The patient was treated with a resection of the mass. Liposarcoma is considered to be the second most common soft-tissue sarcoma. It usually involves the lower extremities, particularly the thigh, and is very rare in the adult foot. Our case was a rare occurrence of myxoid liposarcoma of the foot diagnosed preoperatively using FNAC.Keywords: myxoid liposarcoma, foot, fine needle

  6. A rare case of choledochal cyst with pancreas divisum: case presentation and literature review.

    Science.gov (United States)

    Ransom-Rodríguez, Adrián; Blachman-Braun, Ruben; Sánchez-García Ramos, Emilio; Varela-Prieto, Jesús; Rosas-Lezama, Erick; Mercado, Miguel Ángel

    2017-02-01

    Choledochal cysts are rare congenital malformations of the bile duct characterized by dilatations of the intrahepatic and/or extrahepatic portion of the biliary tree, they are associated to an anomalous arrangement of the pancreaticobiliary duct. Pancreas divisum results from a fusion failure of the pancreatic buds. The coexistence of pancreas divisum and choledochal cyst in adults has been reported in less than 10 well documented cases. This article presents a case of a 42-year-old Peruvian man with intermittent episodes of abdominal pain, initially diagnosed with choledocholithiasis, who underwent open cholecystectomy. During surgery, a diagnosis of choledochal cyst and pancreas divisum was made, and therefore a hepaticoduodenostomy was performed. The patient was referred to our hospital due to persistence of abdominal pain. After admission, a papillectomy was achieved without further complications. A cyst resection and dismantling of hepaticoduodenostomy with Roux-en-Y was performed 8 years later. During the subsequent 18-month follow-up, the patient remains asymptomatic.

  7. A Rare Case of Nonsecretory Multiple Myeloma in Lagos, Nigeria: A Case Report and Literature Review

    Directory of Open Access Journals (Sweden)

    Ebele Uche

    2015-01-01

    Full Text Available Multiple myeloma (MM is a plasma cell disorder associated with clonal proliferation of plasma cells. Nonsecretory multiple myeloma (NSMM is a rare variant of MM and accounts for approximately 1% to 5% of all cases. It is defined as symptomatic myeloma without detectable monoclonal immunoglobulin on serum or urine electrophoresis. This variant usually poses a diagnostic challenge to the clinician. We present a 60-year-old Nigerian man who was investigated extensively for bone pain, weight loss, and anaemia. He was eventually diagnosed as having nonsecretory multiple myeloma based on histology and immunohistochemistry results of bone marrow trephine biopsy. He is currently being managed with bortezomib, doxorubicin, and thalidomide, as well as zoledronic acid. He is also on anticoagulation. He continues to show remarkable clinical improvement. We describe this case report and literature review for better awareness amongst medical practitioners and pathologists.

  8. Defining Political Extremism in the Balkans. The Case of Serbia

    Directory of Open Access Journals (Sweden)

    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.

  9. Thoracoscopic Resection of a Rare Case of Hemangioma of the Azygos Venous Arch

    Directory of Open Access Journals (Sweden)

    Ma Husai

    2017-01-01

    Full Text Available Hemangioma of the azygos venous arch is an exceedingly rare incident. This is a case of a thoracoscopic complete resection of a hemangioma of the azygos venous arch in a 37-year-old woman.

  10. 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.

  11. Eccrine Porocarcinoma: A Case Report of a Rare Skin Malignancy (Dermatopathologic View

    Directory of Open Access Journals (Sweden)

    Vladimír Bartoš

    2017-03-01

    Full Text Available Eccrine porocarcinoma is extremely rare malignant skin adnexal tumor histogenetically derived from the sweat gland ducts and acrosyringium. The authors described a 66-year-old woman, who was diagnosed to have a malignant cutaneous neoplasia arising in the lateral side of the right shin. The lesion displayed exophytic growth with central ulceration, it was rounded and well-defined having 25 mm in diameter. Microscopically, it exhibited a mixture of atypical basaloid and clear cell population arranged in solid-nodular fashion, that grew out from the epidermis and infiltrated the corium. Ductal formations were sporadically present as well. Tumor was immunohistochemically positive for HMWCK, CK7, EMA, weakly positive for pCEA and negative for mCEA and BerEP4. The spectrum of histomorphologic and immunophenotypic findings favored a diagnosis of primary eccrine porocarcinoma. Having made the retrospective analysis, the authors found only this single one biopsy-proven case of EP during the last seven years suggesting exclusive diagnosis in a routine dermatopathologic practice.

  12. 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.

  13. Pancreatic carcinosarcoma mimics malignant intraductal papillary mucinous neoplasm: A rare case report and literature review.

    Science.gov (United States)

    Li, Bing-Qi; Liu, Qiao-Fei; Chang, Xiao-Yan; Hu, Ya; Chen, Jie; Guo, Jun-Chao

    2017-06-01

    Carcinosarcoma, an extremely rare pancreatic primary tumor, is characterized by coexistence of both carcinomatous and sarcomatous components. Due to its rarity, the clinical manifestation and imaging features have not been recognized. An accurate diagnostic method has not been available and a widely accepted guidelines instructing treatment has not been established. We present an uncommon case of pancreatic carcinosarcoma (PCS) which has been preoperatively diagnosed as pancreatic malignant intraductal papillary mucinous neoplasm. A radical resection, including total pancreatectomy (TP) and splenectomy, was performed. The diagnosis of PCS was confirmed by postoperative pathology. A radical resection, including TP and splenectomy, was performed. The patient was followed up by abdominal contrast-enhanced computed tomography scan and blood tumor marker examination. The patient is still alive and self-sufficient 7 months after the surgery. No evidence of tumor recurrence is found during follow-up. Although, until recently, there are no widely accepted guidelines instructing treatment for PCS, a radical resection is still a possible way. All the pancreatic neoplastic patients with high surgical risk should be transferred to a specialized high-volume pancreatic center to get precise preoperative evaluation, fine operation technique, and careful postoperative management.

  14. CASE REPORT: Esophageal and Gastric T-Cell Lymphoma: A Rare Entity

    Directory of Open Access Journals (Sweden)

    Raja Shekhar R.Sappati Biyyani

    2012-01-01

    Full Text Available Background: Primary gastrointestinal T-cell lymphomas are extremely rare entity and are much less common than B-Cell lymphomas. Case History: A primary T-cell lymphoma was diagnosed in an octogenarian African American male with a history of diabetes mellitus type-II, remote history of prostate cancer, hypertension, obesity and hyperlipidemia. He had symptoms of dysphagia, early satiety, lossof appetite and loss of weight. He was Helicobacter pylori IgG antibody positive and on treatment. Result of first biopsy duringendoscopy showed only heavy lymphoid infiltrate. But, due to high suspicion of malignancy, a second upper gastrointestinal endoscopy and biopsy was performed .This biopsy from the large deep 3cm friable ulcer with nodular base was taken which showed atypical lymphoid cells positive for CD3 and CD7 and negative for CD5, CD4 , CD8 and CD56 . The combination of the histological, immunohistological stain results and the gene rearrangement results confirmed T cell lymphoma. The patient died after 5 months after5 cycles of chemotherapeutic agents of severe dehydration and complications from sepsis.

  15. [A rare complication of dysarthria in a patient with inclusion body myositis: a case report].

    Science.gov (United States)

    Isayama, Reina; Shiga, Kensuke; Tanaka, Eijirou; Itsukage, Masahiro; Tokuda, Takahiko; Nakagawa, Masanori

    2010-10-01

    We reported a 71-year-old man with inclusion body myositis with clinically overt dysarthria. He had been suffering from gradual progression of weakness in the hand muscles and lower extremities as well as dysarthria three years before admission. His neurological examination revealed muscle atrophy and weakness in the tongue, the forearm flexors, and the vastus medialis muscles. He had dysarthria to a moderate degree, while he denied any dysphasia. A biopsy from vastus lateralis muscle showed variation in fiber size, infiltration of mononucleated cells, and numerous fibers with rimmed vacuoles, leading to the diagnosis of definite inclusion body myositis. The EMG findings of the tongue demonstrated low amplitude motor unit potentials during voluntary contraction, abundant fibrillation potentials at rest, and preserved interference pattern at maximal contraction, implying myogenic changes. We surmised the dysarthria seen in this patient, an atypical clinical feature in IBM, presumably caused by muscle involvement in the tongue muscle. Dysphasia is common symptom in IBM patient and has been much reported previously. But dysarthria in IBM patient has not been aware, for this reason this report should be the rare case.

  16. A very rare Presentation of Bifocal Non Union Radius with Ipsilateral Ulnar Shaft Non Union: Case Report.

    Science.gov (United States)

    Padha, Vikas; Awasthi, Bhanu; Singh, Dhananjay; Kalia, Sandeep

    2016-01-01

    A bifocal non-union of shaft of radius associated with ipsilateral non-union shaft of ulna in an adult has not been reported in the literature till date to the best of our knowledge, though few similar cases of fresh fractures have been reported. The case being reported by us is the first of its kind. We report a case of bifocal non-union of shaft radius with non-union ipsilateral shaft of ulna in a 48-year-old right handed male along with discussion of alternative treatment options. We describe an extremely rare and complicated non-union in which our patient got excellent results along with satisfactory functional recovery as a result of appropriate surgical treatment.

  17. 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.

  18. 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.

  19. Pyogenic Arthritis of the Ankle Joint Following a High-Voltage Electrical Burn in the Lower Extremity: A Case Report

    International Nuclear Information System (INIS)

    Kim, Kuk Seon; Lee, Gyung Kyu; Kang, Ik Won; Hwang, Dae Hyun; Lee, Eil Seong; Min, Seon Jung; Han, You Mie; Lee, Eil Seong

    2011-01-01

    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.

  20. 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.