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Sample records for bilateral pulmonary agenesis

  1. Prenatal Diagnosis of Bilateral Pulmonary Agenesis: a Case Report

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    Lee, Kyung A.; Cho, Jeong Yeon; LEE, Seung Mi; Jun, Jong Kwan; Kang, Jieun; Seo, Jeong-Wook

    2009-01-01

    We report a case of bilateral pulmonary agenesis (BPA), which was suspected during a prenatal US examination and diagnosed by fetal magnetic resonance imaging (MRI). BPA is an extremely rare congenital anomaly and, although many fetal structural defects can be detected with a high degree of confidence after introducing high-resolution US, the prenatal diagnosis of BPA remains problematic. Other thoracic abnormalities, such as a congenital diaphragmatic hernia, congenital cystic adenomatoid ma...

  2. Prenatal Diagnosis of Bilateral Pulmonary Agenesis: a Case Report

    International Nuclear Information System (INIS)

    We report a case of bilateral pulmonary agenesis (BPA), which was suspected during a prenatal US examination and diagnosed by fetal magnetic resonance imaging (MRI). BPA is an extremely rare congenital anomaly and, although many fetal structural defects can be detected with a high degree of confidence after introducing high-resolution US, the prenatal diagnosis of BPA remains problematic. Other thoracic abnormalities, such as a congenital diaphragmatic hernia, congenital cystic adenomatoid malformation, and pulmonary sequestration, should be excluded from the list of possible diagnoses before coming to the conclusion of BPA, because BPA is absolutely incompatible with extrauterine life, and an accurate internal diagnosis can prevent a futile intervention from being performed

  3. Bilateral multicystic renal dysplasia with potter sequence: A case with penile agenesis

    International Nuclear Information System (INIS)

    Hereditary renal adysplasia (HRA) is rare autosomal dominant condition. Patients have several other anomalies including Potter facies, thoracic, cardiac, and extremity deformities. The case present dysmorphic facial features such as hypertelorism, prominent epicanthic folds, a flat and broad nose, choanal stenosis, low set ears and a receding chin. He had femoral bowing, hypoplastic right tibia and agenesis of the right foot. He had rich and thin skin. He had also a dysplastic empty scrotum, penile agenesis, and anal atresia. The autopsy revealed pulmonary hypoplasia, ventricular septal defect, bilateral multicystic renal dysplasia, agenesis of ureter and bladder, intraabdominal testicles, and a single umbilical artery. The penile agenesis was first reported, and including the consanguinity in the parents might further delineate the bilateral multicystic HRA. Vater/caudal regression anomalies, Mullerian duct/aplasia, unilateral renal agenesis, and cervicothoracic somite anomalies association and Coloboma, heart anomaly, choanal atresia, retardation, retardation, genital and ear anomalies syndrome has been considered in differential diagnosis. (author)

  4. Pulmonary Artery Agenesis: A Case Series

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    Meltem Ağca

    2015-04-01

    Full Text Available Pulmonary artery agenesis is a rare congenital abnormality in which atresia was encountered in the short segment of the right or left pulmonary arteries. It can be isolated or associated with cardiac abnormalities such as tetralogy of Fallot, septal defects or pulmonary stenosis.The majority of cases are diagnosed in childhood whereas some cases yield no symptoms until adulthood. We evaluated retrospectively 5 pulmonary artery agenesis cases diagnosed in our clinics between 1998-2010 with respect to the literature.

  5. Bilateral agenesis of the anterior cruciate ligament: MRI evaluation

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    Bedoya, Maria A.; Jaramillo, Diego [The Children' s Hospital of Philadelphia, Radiology Department, Philadelphia, PA (United States); McGraw, Michael H. [Hospitalof theUniversityof Pennsylvania, Divisionof Orthopaedics, Philadelphia, PA (United States); Wells, Lawrence [The Children' s Hospital of Philadelphia, Division of Orthopaedics, Philadelphia, PA (United States)

    2014-09-15

    Bilateral agenesis of the anterior cruciate ligament (ACL) is extremely rare. We describe a 13-year-old girl who presented with bilateral knee pain without history of trauma; she has two family members with knee instability. Magnetic resonance imaging showed bilateral absence of the ACL, and medial posterior horn meniscal tears. Bilateral arthroscopic partial meniscectomy and anterior cruciate ligament reconstruction was performed. (orig.)

  6. Unilateral Pulmonary Agenesis and Gastric Duplication Cyst: A Rare Association

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    Amir Halilbasic

    2013-01-01

    Full Text Available Lung agenesis and gastric duplication cysts are both rare congenital anomalies. Gastric duplication cysts can present with nausea, vomiting, hematemesis, or vague abdominal pain. Unilateral pulmonary agenesis can present with respiratory distress which usually occurs due to retention of bronchial secretions and inflammations. We report the unique case of right pulmonary agenesis associated with gastric duplication cyst.

  7. Unilateral Pulmonary Agenesis and Gastric Duplication Cyst: A Rare Association

    OpenAIRE

    Amir Halilbasic; Fahrija Skokic; Nesad Hotic; Edin Husaric; Gordana Radoja; Selma Muratovic; Nermina Dedic; Meliha Halilbasic

    2013-01-01

    Lung agenesis and gastric duplication cysts are both rare congenital anomalies. Gastric duplication cysts can present with nausea, vomiting, hematemesis, or vague abdominal pain. Unilateral pulmonary agenesis can present with respiratory distress which usually occurs due to retention of bronchial secretions and inflammations. We report the unique case of right pulmonary agenesis associated with gastric duplication cyst.

  8. Pulmonary agenesis and pulmonary sling anomaly in an infant with Down syndrome.

    Science.gov (United States)

    Takci, S; Yigit, S; Haliloglu, M; Boduroglu, K; Kiper, N

    2013-01-01

    Pulmonary agenesis is a rare congenital anomaly presenting with normal karyotype in most of the cases. Rarely pulmonary agenesis is associated with chromosomal abnormalities and other genetic disorders such as Oculo-auriculo-vertebral spectrum, VACTERL association and velo-cardio-facial syndrome. This report presents a patient with pulmonary agenesis, pulmonary sling anomaly and Down syndrome. PMID:24341150

  9. Isolated Left Pulmonary Artery Agenesis: A Case Report

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    Tansel Ansal Balcı

    2012-08-01

    Full Text Available Unilateral pulmonary artery agenesis without any cardiovascular malformation is a rare anomaly. We present the imaging findings of a patient who was diagnosed as isolated left pulmonary artery agenesis. A 27-year-old female patient was admitted to our hospital due to dyspnea during exercise for five years. Chest X-ray revealed minimally small left pulmonary hilum and left lung. She was admitted to our clinic with the suspicion of pulmonary artery pathology. Absent perfusion of the left lung with normal ventilation was visualized on scintigraphy. MDCT angiography of pulmonary arteries showed absent left main pulmonary artery with systemic collaterals around left hemithorax. Pulmonary artery agenesis can be asymptomatic and isolated until adulthood. Both scintigraphy and CT angiography images of pulmonary artery agenesis of a patient are rare in the literature. Pulmonary ventilation- perfusion scintigraphy can be used not only for pulmonary embolism but also pathologies involving pulmonary artery and its branches. (MIRT 2012;21:80-83

  10. Isolated Left Pulmonary Artery Agenesis: A Case Report

    OpenAIRE

    Tansel Ansal Balcı; Zehra Pınar Koç; Gamze Kırkıl; Ahmet Kürşad Poyraz

    2012-01-01

    Unilateral pulmonary artery agenesis without any cardiovascular malformation is a rare anomaly. We present the imaging findings of a patient who was diagnosed as isolated left pulmonary artery agenesis. A 27-year-old female patient was admitted to our hospital due to dyspnea during exercise for five years. Chest X-ray revealed minimally small left pulmonary hilum and left lung. She was admitted to our clinic with the suspicion of pulmonary artery pathology. Absent perfusion of the left lung w...

  11. Bilateral Congenital Anophthalmos and Agenesis of the Optic Pathways

    OpenAIRE

    Aktekin, Mustafa; Öz, Özay; Saygılı, Muaffak Refik; Kurtoğlu, Zeliha

    2005-01-01

    This report presents a rare example of a bilateral congenital anophthalmos and an agenesis of the optic pathways. The MR imaging studies revealed that the eyeballs, optic nerves, optic chiasm, optic tracts and optic radiation were absent. The chromosomal examination was normal. Mild mental retardation was also observed. Apart from the rarity of the anophthalmos and the total absence of the optic pathways, no etiologic reason for this pathology could be detected, which makes this case more sig...

  12. Bilateral meandering pulmonary veins

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    Thupili, Chakradhar R.; Udayasankar, Unni [Pediatric Imaging, Imaging Institute Cleveland Clinic, Cleveland, OH (United States); Renapurkar, Rahul [Imaging Institute Cleveland Clinic, Thoracic Imaging, L10, Cleveland, OH (United States)

    2015-06-15

    Meandering pulmonary veins is a rare clinical entity that can be mistaken for more complex congenital syndromes such as hypogenetic lung syndrome. We report imaging findings in a rare incidentally detected case of bilateral meandering pulmonary veins. We briefly discuss the role of imaging in diagnosing this condition, with particular emphasis on contrast-enhanced CT. (orig.)

  13. Polyhydramnios in Lrp4 knockout mice with bilateral kidney agenesis: Defects in the pathways of amniotic fluid clearance

    OpenAIRE

    Hiroshi Tanahashi; Qing-Bao Tian; Yoshinobu Hara; Hiroyuki Sakagami; Shogo Endo; Tatsuo Suzuki

    2016-01-01

    Amniotic fluid volume during mid-to-late gestation depends mainly on the urine excretion from the foetal kidneys and partly on the fluid secretion from the foetal lungs during foetal breathing-like movements. Urine is necessary for foetal breathing-like movements, which is critical for foetal lung development. Bilateral renal agenesis and/or obstruction of the urinary tract lead to oligohydramnios, which causes infant death within a short period after birth due to pulmonary hypoplasia. Lrp4, ...

  14. Surgical Treatment of a Rare Isolated Bilateral Agenesis of Anterior and Posterior Cruciate Ligaments

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    G. Cerulli

    2014-01-01

    Full Text Available The isolated bilateral agenesis of both cruciate ligaments is a rare congenital disorder. A 17-year-old male came to our attention due to an alteration in gait pattern, pain, and tendency to walk on the forefoot with his knee flexed. The patient did not recall previous injuries. Upon physical examination anterior and posterior chronic instability were observed. Radiographic examination of both knees showed hypoplasia of the tibial eminence, a hypoplastic lateral femoral condyle, and a narrow intercondylar notch. MRI brought to light a bilateral agenesis of both posterior cruciate ligaments. Arthroscopic evaluation confirmed bilateral isolated agenesis of both cruciate ligaments. We recommended a rehabilitation program to prepare the patient for the arthroscopic construction of both cruciate ligaments.

  15. Polyhydramnios in Lrp4 knockout mice with bilateral kidney agenesis: Defects in the pathways of amniotic fluid clearance

    Science.gov (United States)

    Tanahashi, Hiroshi; Tian, Qing-Bao; Hara, Yoshinobu; Sakagami, Hiroyuki; Endo, Shogo; Suzuki, Tatsuo

    2016-01-01

    Amniotic fluid volume during mid-to-late gestation depends mainly on the urine excretion from the foetal kidneys and partly on the fluid secretion from the foetal lungs during foetal breathing-like movements. Urine is necessary for foetal breathing-like movements, which is critical for foetal lung development. Bilateral renal agenesis and/or obstruction of the urinary tract lead to oligohydramnios, which causes infant death within a short period after birth due to pulmonary hypoplasia. Lrp4, which functions as an agrin receptor, is essential for the formation of neuromuscular junctions. Herein, we report novel phenotypes of Lrp4 knockout (Lrp4−/−) mice. Most Lrp4−/− foetuses showed unilateral or bilateral kidney agenesis, and Lrp4 knockout resulted in polyhydramnios. The loss of Lrp4 compromised foetal swallowing and breathing-like movements and downregulated the expression of aquaporin-9 in the foetal membrane and aquaporin-1 in the placenta, which possibly affected the amniotic fluid clearance. These results suggest that amniotic fluid removal was compromised in Lrp4−/− foetuses, resulting in polyhydramnios despite the impairment of urine production. Our findings indicate that amniotic fluid removal plays an essential role in regulating the amniotic fluid volume. PMID:26847765

  16. Polyhydramnios in Lrp4 knockout mice with bilateral kidney agenesis: Defects in the pathways of amniotic fluid clearance.

    Science.gov (United States)

    Tanahashi, Hiroshi; Tian, Qing-Bao; Hara, Yoshinobu; Sakagami, Hiroyuki; Endo, Shogo; Suzuki, Tatsuo

    2016-01-01

    Amniotic fluid volume during mid-to-late gestation depends mainly on the urine excretion from the foetal kidneys and partly on the fluid secretion from the foetal lungs during foetal breathing-like movements. Urine is necessary for foetal breathing-like movements, which is critical for foetal lung development. Bilateral renal agenesis and/or obstruction of the urinary tract lead to oligohydramnios, which causes infant death within a short period after birth due to pulmonary hypoplasia. Lrp4, which functions as an agrin receptor, is essential for the formation of neuromuscular junctions. Herein, we report novel phenotypes of Lrp4 knockout (Lrp4(-/-)) mice. Most Lrp4(-/-) foetuses showed unilateral or bilateral kidney agenesis, and Lrp4 knockout resulted in polyhydramnios. The loss of Lrp4 compromised foetal swallowing and breathing-like movements and downregulated the expression of aquaporin-9 in the foetal membrane and aquaporin-1 in the placenta, which possibly affected the amniotic fluid clearance. These results suggest that amniotic fluid removal was compromised in Lrp4(-/-) foetuses, resulting in polyhydramnios despite the impairment of urine production. Our findings indicate that amniotic fluid removal plays an essential role in regulating the amniotic fluid volume. PMID:26847765

  17. Pregnancy complicated by morbidly adherent placenta in a patient with bilateral ovarian agenesis: a case report

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    Wong HS

    2013-02-01

    Full Text Available Hong Soo WongAustralian Women's Ultrasound Centre, Brisbane, AustraliaAbstract: The author presents a case of in vitro fertilization pregnancy complicated by morbidly adherent placenta in a patient with congenital bilateral ovarian agenesis. A 31-year-old woman with congenital bilateral ovarian agenesis who had undergone two previous dilatation and curettage procedures conceived following in vitro fertilization with a donor egg. Spontaneous labor occurred at 38 weeks and 5 days' gestation. The labor was augmented in the active phase and resulted in instrumental vaginal delivery. The third stage was complicated by hemorrhage and retained placenta. Morbidly adherent placenta was diagnosed on attempt at manual removal of the placenta, and the adherent part of the placenta was left in situ. This was removed uneventfully at 5 weeks following childbirth when there was no blood flow observed between the placenta and the myometrium on Doppler ultrasound examination. In conclusion, successful parturition is possible in patients with congenital bilateral ovarian agenesis. When morbidly adherent placenta is managed conservatively, the placenta may be safely removed if there is no vascularity between the placenta and the myometrium.Keywords: IVF, placenta accreta, parturition, myometrium

  18. Adult patient with pulmonary agenesis: focusing on one-lung ventilation during general anesthesia

    OpenAIRE

    Yu, Yuetian; Zhu, Cheng; QIAN, XIAOZHE; Gao, Yuan; Zhang, Zhongheng

    2016-01-01

    Congenital pulmonary agenesis is a rare condition with high mortality. Mechanical ventilation in these patients is challenging and there has no such case been reported in the literature. We reported a 61-year-old female with lung agenesis who presented to our hospital with pneumonia and pelvic mass. In the past, she had undergone repairing of atrial septal defect and mitral valve forming surgery at 6-year-old. Thereafter she had remained asymptomatic until this time of hospital admission. The...

  19. Imaging features of isolated unilateral pulmonary artery agenesis presenting in adulthood: a review of four cases

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    Griffin, N. [Royal Brompton and Harefield NHS Trust, London (United Kingdom)]. E-mail: nyreegriffin@hotmail.com; Mansfield, L. [Royal Brompton and Harefield NHS Trust, London (United Kingdom); Redmond, K.C. [Royal Brompton and Harefield NHS Trust, London (United Kingdom); Dusmet, M. [Royal Brompton and Harefield NHS Trust, London (United Kingdom); Goldstraw, P. [Royal Brompton and Harefield NHS Trust, London (United Kingdom); Mittal, T.K. [Royal Brompton and Harefield NHS Trust, London (United Kingdom); Padley, S. [Royal Brompton and Harefield NHS Trust, London (United Kingdom)

    2007-03-15

    Aim: To highlight the variation in clinical manifestations, imaging and management of four cases of unilateral pulmonary artery agenesis presenting in adulthood. Method: Four patients with unilateral pulmonary artery agenesis were referred to our institution between 1995 and 2005. They underwent a series of investigations, including chest radiography, echocardiography, ventilation perfusion scintigraphy, angiography, computed tomography (CT) and magnetic resonance imaging (MRI). Results: Two of the four patients had absence of the right main pulmonary artery, whilst the remaining two patients had absence of the left main pulmonary artery. One patient showed a restrictive defect on pulmonary function tests. Two patients who had ventilation perfusion scintigraphy showed absent perfusion and reduced ventilation on the affected side. Angiography (where performed), CT and MRI confirmed the anatomy and the presence of multiple collaterals. Bronchiectasis was demonstrated on CT in two patients, with one also demonstrating a mosaic attenuation pattern. One patient had an incidental lung tumour on the side of the agenesis, which was diagnosed as a chondroid hamartoma on histology. Three of the four patients eventually underwent resection of the affected lung. Conclusion: Isolated unilateral pulmonary artery agenesis has a non-specific presentation. Awareness of this condition can lead to earlier diagnosis, with cross-sectional imaging making an important contribution.

  20. Pulmonary Artery Agenesis Associated With Emphysema and Multiple Invasive Non-Small Cell Lung Cancers.

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    Makdisi, George; Edell, Eric S; Maleszewski, Joseph J; Molina, Julian R; Deschamps, Claude

    2015-06-01

    Pulmonary artery (PA) agenesis in the absence of associated cardiac abnormalities is a rare congenital abnormality. It may remain undiagnosed until adulthood when patients present with respiratory symptoms such as hemoptysis, dyspnea, repeated respiratory infections, or pulmonary hypertension. Herein we present a case of a 50-year-old woman who was found to have multiple, morphologically distinct non-small cell lung cancers in association with agenesis of the PA. This instance represents the fourth reported case of such association in the English literature. PMID:26046873

  1. Bilateral Pulmonary Cyst in a Child

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    Ravinder K Gupta, Ritu Gupta

    2008-01-01

    A four year old male child presented with history of cough for last six months. Chest skiagram and CTscan of mediastinum revealed bilateral pulmonary hydatid cyst. Serology did authenticate diagnosis. Thechild was prescribed albendazole for 6 months and was advised to come for follow up. The case ofbilateral pulmonary hydatid cyst is being presentated because of rarity.

  2. Bilateral Pulmonary Cyst in a Child

    Directory of Open Access Journals (Sweden)

    Ravinder K Gupta, Ritu Gupta

    2008-04-01

    Full Text Available A four year old male child presented with history of cough for last six months. Chest skiagram and CTscan of mediastinum revealed bilateral pulmonary hydatid cyst. Serology did authenticate diagnosis. Thechild was prescribed albendazole for 6 months and was advised to come for follow up. The case ofbilateral pulmonary hydatid cyst is being presentated because of rarity.

  3. Implant-Prosthetic Rehabilitation in Bilateral Agenesis of Maxillary Lateral Incisors with a Mini Split Crest

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    Figliuzzi, M. M.; Giudice, A.; Pileggi, S.; Pacifico, D.; Marrelli, M.; Tatullo, M.; Fortunato, L.

    2016-01-01

    The reported clinical case describes the surgical procedure of ridge augmentation by using a “split crest” technique with a partial thickness flap and a subsequent implant-prosthetic rehabilitation aimed at treating a bilateral agenesis of the upper lateral incisors. In such cases with vestibule-palatal and mesial-distal scarce bone thicknesses associated with the need of a proper functional and aesthetic rehabilitation, the split crest technique is particularly suitable. In the case we reported, because of the poor bone thicknesses, we performed a minimally invasive split crest which allowed a correct insertion of the fixtures. This technique allowed us to achieve an optimal functional and aesthetic rehabilitation; moreover, we obtained a good emergency profile, ensuring the vitality of the close teeth and ensuring a good primary stability and the following osseointegration of dental implants. PMID:27190658

  4. Uterine Didelphys with blind hemivagina, hematocolpos, ipsilateral renal agenesis (UD-BHRA) and bilateral ovarian tumors : a case report

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    Lee, Young Rae; Choi, Byung Ik; Park, Hae Won; Kook, Shin Ho [Kangbuk Samsung Hospital, Seoul (Korea, Republic of)

    1998-03-01

    Uterine didelphys is a congenital malformation characterized by the presence of two separated hemiuteri and hemivaginas, due to lack of midfusion of the mullerian ducts. We report a case of UD-BHRA (uterine didelphys with blind hemivagina and ipsilateral renal agencies), a rare type of uterine dedelphys characterized by symptomatic unilateral hematocolpos due to blind hemivagina after menarche and ipsilateral renal agenesis. The MRI findings in 22-year-old woman with bilateral ovarian tumors demonstrated two separated uterine horns and cervical and vaginal canals, with left hematocolpos and left agenesis. (author). 7 refs., 1 fig.

  5. Uterine Didelphys with blind hemivagina, hematocolpos, ipsilateral renal agenesis (UD-BHRA) and bilateral ovarian tumors : a case report

    International Nuclear Information System (INIS)

    Uterine didelphys is a congenital malformation characterized by the presence of two separated hemiuteri and hemivaginas, due to lack of midfusion of the mullerian ducts. We report a case of UD-BHRA (uterine didelphys with blind hemivagina and ipsilateral renal agencies), a rare type of uterine dedelphys characterized by symptomatic unilateral hematocolpos due to blind hemivagina after menarche and ipsilateral renal agenesis. The MRI findings in 22-year-old woman with bilateral ovarian tumors demonstrated two separated uterine horns and cervical and vaginal canals, with left hematocolpos and left agenesis. (author). 7 refs., 1 fig

  6. Male Fertility after Spermatocele Formation from Tunica Vaginalis in Patients with Bilateral Vas Agenesis

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    Shamsa Ali

    2008-01-01

    Full Text Available To form spermatocele from vaginal layers as a sperm reservoir and intra-uterine insemination (IUI in infertile men with bilateral vas agenesis (BVA, we studied 19 patients with azoospermia due to BVA referred to our infertility clinic from March 1992 until May 2003. The ages of the patients ranged from 20-41 (mean 29.6 ± 5.8 years. After physical examination, hormone assay, testis biopsy, and confirming normal spermatogenesis, we have performed 23 alloplastic spermatoceles from the tunica vaginal layers in 11 patients. We retrieved sperms and performed IUI in 6 patients′ wives 3 months post-operation when scrotal sonography revealed spermatocele with a good volume of seminal liquid. Among 6 patients′ wives, 2 successful preg-nancies occurred, and 2 normal babies (one boy with normal bilateral vas and one girl were delivered successfully by cesarean section. We conclude that although the method of choice for fer-tility in BVA in artificial reproductive therapy era is percutaneous epididymal sperm aspiration (PESA and intracytoplasmic sperm injection (ICSI, but when the sophisticated facilities are not available or cost-effectiveness is matter of concern, alloplastic spermatocele from tunica vaginalis and IUI may be a viable option.

  7. Unilateral pulmonary agenesis associated with oesophageal atresia and tracheoesophageal fistula: A case report with prenatal diagnosis

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    Go Miyano; Keiichi Morita; Masakatsu Kaneshiro; Hiromu Miyake; Mariko Koyama; Hiroshi Nouso; Masaya Yamoto; Reiji Nakano; Yasuhiko Tanaka; Tomizo Nishiguchi; Takakazu Kawamura; Koji Fukumoto; Naoto Urushihara

    2015-01-01

    We describe herein a case of unilateral pulmonary agenesis (PA) with oesophageal atresia (EA)/tracheoesophageal fistula (TEF) that was diagnosed prenatally and repaired by esophagoesophagostomy with stable postoperative course. The patient was born at 34 weeks gestation, after ultrasonography at 22 weeks gestation showed possible right-sided diaphragmatic eventration or PA and EA was subsequently suspected due to hydramnios. The initial X-ray showed mediastinal shift to the right, and coil up...

  8. Bilateral spontaneous pneumothorax with pulmonary metastases of synovial sarcoma

    International Nuclear Information System (INIS)

    The association of bilateral spontaneous pneumothorax with pulmonary tumor is uncommon and with pulmonary metastases is rare. The clinical and radiological features of bilateral spontaneous pneumothorax from a synovial sarcoma in a 14 years old boy are described. (author)

  9. Bilateral stony lung: pulmonary alveolar microlithiasis.

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    Chandra, Subhash; Mohan, Anant; Guleria, Randeep; Das, Prasenjit; Sarkar, Chitra

    2009-01-01

    A 40-year-old male paddy field worker was referred for exertional shortness of breath and non-productive cough for 4 years. He had been treated for pulmonary tuberculosis twice. Chest radiograph showed extensive bilateral nodular opacities ("sandstorm-like") in the middle and lower lobe. Pulmonary function tests revealed a restrictive ventilatory defect. High resolution CT showed widespread nodular infiltration with "crazy paving" appearance and interrupted black pleura sign. This was confirmed as pulmonary alveolar microlithiasis (PAM) by trans-bronchial lung biopsy, which showed normal respiratory lining epithelium with dilated alveolar spaces containing many calcific bodies, some of which showed concentric calcification. The possibilities of silicosis (due to exposure to husk) and tuberculosis, both of which can mimic PAM clinically and radiologically, made this case a diagnostic challenge. PMID:21686505

  10. Bilateral multiple pulmonary tuberculous nodules mimicking metastatic disease

    International Nuclear Information System (INIS)

    We present CT findings of a young woman who has bilateral pulmonary nodules mimicking metastases. Clinical presentation with active multiple pulmonary macronodules without cavitation responsive to treatment is an atypical manifestation of pulmonary tuberculosis. We reviewed the causes of multiple pulmonary nodules, role of radiological findings in differential diagnosis and parenchymal manifestations of pulmonary tuberculosis in this report

  11. Bilateral pulmonary sequestration: a case report

    International Nuclear Information System (INIS)

    A 21-year-old woman presented with productive cough and hemoptysis. Chest radiograph shows a large thin-walled cystic lesion with an air-fluid level in medial portion of the left lower lung zone and opacity in paravertebral area of the right lower lung zone. Chest CT scan shows a thin-walled cavitary lesion in the posterior basal segment of left lower lobe with an air-fluid level. Area of consolidation containing air-filled cysts was also observed in medial aspect of right lower lobe. Selective angiogram obtained from aberrant artery arising from descending abdominal aorta showed two main branches of the artery supplying bilateral pulmonary lesions

  12. Bilateral renal agenesis/hypoplasia/dysplasia (BRAHD: postmortem analysis of 45 cases with breakpoint mapping of two de novo translocations.

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    Louise Harewood

    Full Text Available BACKGROUND: Bilateral renal agenesis/hypoplasia/dysplasia (BRAHD is a relatively common, lethal malformation in humans. Established clinical risk factors include maternal insulin dependent diabetes mellitus and male sex of the fetus. In the majority of cases, no specific etiology can be established, although teratogenic, syndromal and single gene causes can be assigned to some cases. METHODOLOGY/PRINCIPAL FINDINGS: 45 unrelated fetuses, stillbirths or infants with lethal BRAHD were ascertained through a single regional paediatric pathology service (male:female 34:11 or 3.1:1. The previously reported phenotypic overlaps with VACTERL, caudal dysgenesis, hemifacial microsomia and Müllerian defects were confirmed. A new finding is that 16/45 (35.6%; m:f 13:3 or 4.3:1 BRAHD cases had one or more extrarenal malformations indicative of a disoder of laterality determination including; incomplete lobulation of right lung (seven cases, malrotation of the gut (seven cases and persistence of the left superior vena cava (five cases. One such case with multiple laterality defects and sirelomelia was found to have a de novo apparently balanced reciprocal translocation 46,XY,t(2;6(p22.3;q12. Translocation breakpoint mapping was performed by interphase fluorescent in-situ hybridization (FISH using nuclei extracted from archival tissue sections in both this case and an isolated bilateral renal agenesis case associated with a de novo 46,XY,t(1;2(q41;p25.3. Both t(2;6 breakpoints mapped to gene-free regions with no strong evidence of cis-regulatory potential. Ten genes localized within 500 kb of the t(1;2 breakpoints. Wholemount in-situ expression analyses of the mouse orthologs of these genes in embryonic mouse kidneys showed strong expression of Esrrg, encoding a nuclear steroid hormone receptor. Immunohistochemical analysis showed that Esrrg was restricted to proximal ductal tissue within the embryonic kidney. CONCLUSIONS/SIGNIFICANCE: The previously unreported

  13. Unilateral pulmonary agenesis associated with oesophageal atresia and tracheoesophageal fistula: A case report with prenatal diagnosis

    Directory of Open Access Journals (Sweden)

    Go Miyano

    2015-01-01

    Full Text Available We describe herein a case of unilateral pulmonary agenesis (PA with oesophageal atresia (EA/tracheoesophageal fistula (TEF that was diagnosed prenatally and repaired by esophagoesophagostomy with stable postoperative course. The patient was born at 34 weeks gestation, after ultrasonography at 22 weeks gestation showed possible right-sided diaphragmatic eventration or PA and EA was subsequently suspected due to hydramnios. The initial X-ray showed mediastinal shift to the right, and coil up sign of the nasogastric tube, without intracardiac anomaly. Immediately after the diagnosis of EA/TEF and unilateral PA on day 0, the patient was intubated in the operating room, and a gastrostomy tube was placed. After pulmonary status stabilized, at 4 days old, EA/TEF was repaired through a thoracotomy in the right 4 th intercostal space. The right main bronchus was noted to continue into the distal oesophagus; this fistula was ligated and divided, and a single-layer esophagoesophagostomy was performed under mild tension with one vertebral gap. The neonate was maintained on mechanical ventilation and gradually weaned to extubation at 7 days old. The postoperative course was uneventful, with the exception of prolonged jaundice that emerged at 3 months old. Laparoscopic cholangiography at that time excluded biliary atresia, and jaundice resolved spontaneously. The patient has not shown any respiratory symptoms or feeding difficulties as of the 12-month follow-up.

  14. Unilateral pulmonary agenesis associated with oesophageal atresia and tracheoesophageal fistula: A case report with prenatal diagnosis.

    Science.gov (United States)

    Miyano, Go; Morita, Keiichi; Kaneshiro, Masakatsu; Miyake, Hiromu; Koyama, Mariko; Nouso, Hiroshi; Yamoto, Masaya; Nakano, Reiji; Tanaka, Yasuhiko; Nishiguchi, Tomizo; Kawamura, Takakazu; Fukumoto, Koji; Urushihara, Naoto

    2015-01-01

    We describe herein a case of unilateral pulmonary agenesis (PA) with oesophageal atresia (EA)/tracheoesophageal fistula (TEF) that was diagnosed prenatally and repaired by esophagoesophagostomy with stable postoperative course. The patient was born at 34 weeks gestation, after ultrasonography at 22 weeks gestation showed possible right-sided diaphragmatic eventration or PA and EA was subsequently suspected due to hydramnios. The initial X-ray showed mediastinal shift to the right, and coil up sign of the nasogastric tube, without intracardiac anomaly. Immediately after the diagnosis of EA/TEF and unilateral PA on day 0, the patient was intubated in the operating room, and a gastrostomy tube was placed. After pulmonary status stabilized, at 4 days old, EA/TEF was repaired through a thoracotomy in the right 4 th intercostal space. The right main bronchus was noted to continue into the distal oesophagus; this fistula was ligated and divided, and a single-layer esophagoesophagostomy was performed under mild tension with one vertebral gap. The neonate was maintained on mechanical ventilation and gradually weaned to extubation at 7 days old. The postoperative course was uneventful, with the exception of prolonged jaundice that emerged at 3 months old. Laparoscopic cholangiography at that time excluded biliary atresia, and jaundice resolved spontaneously. The patient has not shown any respiratory symptoms or feeding difficulties as of the 12-month follow-up. PMID:25659560

  15. Prenatal Diagnosis of Bilateral Ectrodactyly and Radial Agenesis Associated with Trisomy 10 Mosaicism

    OpenAIRE

    Jonathan Lévy; Jean-Marie Jouannic; Julien Saada; Ferdinand Dhombres; Jean-Pierre Siffroi; Marie-France Portnoï

    2013-01-01

    Ectrodactyly or split hand and foot malformations (SHFMs) are rare malformations of the limbs, characterized by median clefts of the hands and feet, syndactyly, and aplasia and/or hypoplasia of the phalanges. They represent a clinically and genetically heterogeneous disorder, with both sporadic and familial cases. Most of the genomic rearrangements identified to date in some forms of SHFM are autosomal dominant traits, involving various chromosome regions. Bilateral radial ray defects compris...

  16. Bifurcating stents in the pulmonary arteries: A novel technique to relieve bilateral branch pulmonary artery obstruction.

    Science.gov (United States)

    Narayan, Hari K; Glatz, Andrew C; Rome, Jonathan J

    2015-10-01

    Balloon angioplasty and stent placement in close proximity to the bifurcation of the branch pulmonary arteries can be challenging. Multiple approaches have been previously described, though none of these approaches both treats bilateral proximal branch pulmonary artery stenosis and provides an anchor for a transcatheter pulmonary valve replacement. We report a novel approach that involves serial stent placement and balloon dilation through the struts of the stent in each pulmonary artery, along with balloon expansion of the proximal portion of the stents to the diameter of the main pulmonary artery. In the two cases we describe, this strategy resulted in significant relief of branch pulmonary artery obstruction without compromising the anatomy of the main pulmonary artery segment. This technique can be an effective way to alleviate stenoses of the bilateral proximal branch pulmonary arteries and provides a landing zone for a future transcatheter pulmonary valve. PMID:26256829

  17. Prenatal Diagnosis of Bilateral Ectrodactyly and Radial Agenesis Associated with Trisomy 10 Mosaicism

    Directory of Open Access Journals (Sweden)

    Jonathan Lévy

    2013-01-01

    Full Text Available Ectrodactyly or split hand and foot malformations (SHFMs are rare malformations of the limbs, characterized by median clefts of the hands and feet, syndactyly, and aplasia and/or hypoplasia of the phalanges. They represent a clinically and genetically heterogeneous disorder, with both sporadic and familial cases. Most of the genomic rearrangements identified to date in some forms of SHFM are autosomal dominant traits, involving various chromosome regions. Bilateral radial ray defects comprise also a large heterogenous group of disorders, including trisomy 18, Fanconi anemia, and thrombocytopenia-absent-radius syndrome, not commonly associated with ectrodactyly. The present paper describes a case of ectrodactyly associated with bilateral radial ray defects, diagnosed in the first trimester of pregnancy, in a fetus affected by trisomy 10. Only four cases of sporadic and isolated ectrodactyly, diagnosed by ultrasonography between 14 and 22 weeks’ gestation, have been reported. To our knowledge, the present case is the first report of mosaic trisomy 10 associated with SHFM and radial aplasia. Trisomy 10 is a rare lethal chromosomal abnormality, most frequently found in abortion products. Only six liveborn mosaic trisomy 10 infants, with severe malformations, dead in early infancy, have been reported. A severe clinical syndrome can be defined, comprising ear abnormalities, cleft lip/palate, malformations of eyes, heart, and kidneys, and deformity of hands and feet and most often associated with death neonatally or in early infancy.

  18. Prenatal diagnosis of bilateral ectrodactyly and radial agenesis associated with trisomy 10 mosaicism.

    Science.gov (United States)

    Lévy, Jonathan; Jouannic, Jean-Marie; Saada, Julien; Dhombres, Ferdinand; Siffroi, Jean-Pierre; Portnoï, Marie-France

    2013-01-01

    Ectrodactyly or split hand and foot malformations (SHFMs) are rare malformations of the limbs, characterized by median clefts of the hands and feet, syndactyly, and aplasia and/or hypoplasia of the phalanges. They represent a clinically and genetically heterogeneous disorder, with both sporadic and familial cases. Most of the genomic rearrangements identified to date in some forms of SHFM are autosomal dominant traits, involving various chromosome regions. Bilateral radial ray defects comprise also a large heterogenous group of disorders, including trisomy 18, Fanconi anemia, and thrombocytopenia-absent-radius syndrome, not commonly associated with ectrodactyly. The present paper describes a case of ectrodactyly associated with bilateral radial ray defects, diagnosed in the first trimester of pregnancy, in a fetus affected by trisomy 10. Only four cases of sporadic and isolated ectrodactyly, diagnosed by ultrasonography between 14 and 22 weeks' gestation, have been reported. To our knowledge, the present case is the first report of mosaic trisomy 10 associated with SHFM and radial aplasia. Trisomy 10 is a rare lethal chromosomal abnormality, most frequently found in abortion products. Only six liveborn mosaic trisomy 10 infants, with severe malformations, dead in early infancy, have been reported. A severe clinical syndrome can be defined, comprising ear abnormalities, cleft lip/palate, malformations of eyes, heart, and kidneys, and deformity of hands and feet and most often associated with death neonatally or in early infancy. PMID:23401811

  19. Successful radiofrequency catheter ablation of atrioventricular nodal reentrant tachycardia in a patient with dextrocardia due to unilateral pulmonary agenesis: a case report

    Directory of Open Access Journals (Sweden)

    Aksu T

    2015-02-01

    Full Text Available Tolga Aksu, Tumer Erdem Guler, Ebru Golcuk, Ismail Erden, Kazim Serhan Ozcan Department of Cardiology, Kocaeli Derince Education and Research Hospital, Derince, Kocaeli, Turkey Abstract: Radiofrequency catheter ablation of the slow pathway is considered to be the treatment of choice for patients with atrioventricular nodal reentrant tachycardia. We report a 34-year-old female with mirror image dextrocardia due to unilateral pulmonary agenesis who underwent successful slow pathway ablation for typical atrioventricular nodal reentrant tachycardia. Using contrast injection, cardiac anatomy was identified in a short time and successfully ablated. Keywords: dextrocardia, AVNRT, ablation, pulmonary agenesis

  20. Bilateral pulmonary emboli after bilateral mastectomy in a 15-year-old boy with hypogonadism: A case report

    OpenAIRE

    Piggott, JR; Yazdani, Arjang

    2010-01-01

    Pulmonary emboli are rare, yet serious, complications of body contouring surgery. When they occur, they more often follow as complications of long, invasive procedures in adults. The present report details a case of bilateral pulmonary emboli in an obese 15-year-old boy with hypogonadism undergoing bilateral mastectomy for gynecomastia. The diagnosis of bilateral pulmonary emboli was made on the basis of clinical presentation and positive ventilation/perfusion scan. The patient responded well...

  1. Bilateral Endobronchial Seeding After Spontaneous Rupture of Pulmonary Hydatid Cyst

    OpenAIRE

    Berkant Özpolat; Çağrı Şenyücel

    2011-01-01

      A 10-year-old boy presented with  sudden unset cough and watery sputum. Chest CT demonstrated a  ruptured left sided pulmonary hydatid cyst with bilateral alveolar consolidation areas.  Albendazole theraphy was started. Cystotomy and capitonnage was performed and he was discharged uneventfully.

  2. Bilateral Endobronchial Seeding After Spontaneous Rupture of Pulmonary Hydatid Cyst

    Directory of Open Access Journals (Sweden)

    Berkant Ozpolat

    2011-09-01

    A 10-year-old boy presented with  sudden unset cough and watery sputum. Chest CT demonstrated a  ruptured left sided pulmonary hydatid cyst with bilateral alveolar consolidation areas.  Albendazole theraphy was started. Cystotomy and capitonnage was performed and he was discharged uneventfully.

  3. Postmortem MRI of bladder agenesis

    Energy Technology Data Exchange (ETDEWEB)

    Barber, Brendan R. [St George' s Hospital, Radiology Department, London (United Kingdom); Weber, Martin A. [Great Ormond Street Hospital for Children, Department of Histopathology, London (United Kingdom); Bockenhauer, Detlef [Great Ormond Street Hospital for Children, Department of Nephrology, London (United Kingdom); Hiorns, Melanie P.; McHugh, Kieran [Great Ormond Street Hospital for Children, Radiology Department, London (United Kingdom)

    2011-01-15

    We report a 35-week preterm neonate with bladder agenesis and bilateral dysplastic kidneys. A suprapubic catheter was inadvertently inserted into one of the larger inferior cysts of the left dysplastic kidney. A postmortem MRI scan was performed with the findings being confirmed on autopsy. We are unaware of another postmortem MRI study demonstrating bladder agenesis. (orig.)

  4. Simultaneous bilateral contrast injection in computed tomography pulmonary angiography

    Energy Technology Data Exchange (ETDEWEB)

    Bulla, Stefan; Pache, Gregor; Langer, Mathias; Blanke, Philipp (Department of Diagnostic Radiology, University Hospital Freiburg (Germany)), Email: stefan.bulla@uniklinik-freiburg.de; Bley, Thorsten (Department of Diagnostic Radiology, University Hospital Hamburg (Germany))

    2012-02-15

    Background. Computed tomography pulmonary angiography (CTPA) has evolved as the gold standard for diagnosing pulmonary embolism. However, subsegmental arteries are often not assessed to do insufficient attenuation. Purpose. To evaluate the influence of simultaneous bilateral versus unilateral injection of a fixed amount of contrast media on pulmonary artery opacification and image quality in CTPA. Material and Methods. In this institutional review board-approved prospective study, 180 patients (91 women, mean age 61.9 +- 16.5 years) referred for CTPA (100 kV) due to suspected pulmonary embolism were randomized in groups of 45 patients each, with either unilateral (A:4 mL/s; B:6 mL/s) or bilateral (C: 6 mL/s; D: 8 mL/s) (Y-shaped line) injection of 50 mL contrast media. Attenuation was assessed including the subsegmental arteries (4th order). Image quality was evaluated by two readers in consensus using a three-point grading scale (3 = excellent image quality, no artifacts, 1 = non-diagnostic). Results. Mean pulmonary artery attenuation was significantly higher with bilateral injection (1st to 3rd order: A: 303.6 +- 8.8HU; B: 371.1 +- 11.0HU vs. C: 443.2 +- 24.1HU; D: 562.3 +- 15.3HU, P < 0.001). Evaluation of subsegmental arteries was feasible for all patients in groups B-D, but only in 36/45 (80%) patients in group A. Subsegmental attenuation was significantly higher with bilateral injection (A: 284.7 +- 12.1HU; B: 367.4 +- 12.1HU vs. C: 494.2 +- 21.5HU; D: 562.3 +- 26.7HU, P < 0.001). Image quality was diagnostic for all patients but best for group C (A: 2.15 +- 0.4; B: 2.14 +- 0.5; C: 2.92 +- 0.3, and D: 2.51 +- 0.5). Conclusion. Using the same amount of contrast media, bilateral injection yields higher pulmonary artery attenuation and better image quality than unilateral injection. This technique may improve subsegmental pulmonary artery assessment

  5. Bilateral hydatid cyst of pulmonary arteries: MR and CT findings

    Energy Technology Data Exchange (ETDEWEB)

    Yaguee, D.; Lozano, M.P.; Lample, C.; Nunez, M.E. [Department of Diagnostic Radiology, Hospital Miguel Servet, Zaragoza (Spain); Sanchez, F. [Department of Thoracic Surgery, Hospital Miguel Servet, Zaragoza (Spain)

    1998-09-01

    The location of hydatid cysts inside pulmonary arteries has been reported only on rare occasions in the literature and is caused, in the majority of cases, by embolization due to the rupture of hydatid cysts located in the heart; more rarely, hematogenous dissemination from a hepatic focus is the cause. We report a case of a 44-year-old patient with hydatid cysts located in both the right and left pulmonary arteries, whose first clinical reference was hemoptysis. The patient had undergone surgery because of a hepatic hydatid cyst 5 months previously. The importance of this case lies in the infrequent bilateral location in both pulmonary arteries and in the absence of intracardiac hydatid cysts. (orig.) With 3 figs., 9 refs.

  6. Bilateral hydatid cyst of pulmonary arteries: MR and CT findings

    International Nuclear Information System (INIS)

    The location of hydatid cysts inside pulmonary arteries has been reported only on rare occasions in the literature and is caused, in the majority of cases, by embolization due to the rupture of hydatid cysts located in the heart; more rarely, hematogenous dissemination from a hepatic focus is the cause. We report a case of a 44-year-old patient with hydatid cysts located in both the right and left pulmonary arteries, whose first clinical reference was hemoptysis. The patient had undergone surgery because of a hepatic hydatid cyst 5 months previously. The importance of this case lies in the infrequent bilateral location in both pulmonary arteries and in the absence of intracardiac hydatid cysts. (orig.)

  7. Formation and resolution of bilateral traumatic pulmonary pseudocysts

    International Nuclear Information System (INIS)

    A case of bilateral traumatic pulmonary pseudocysts, an uncommon complication of blunt chest trauma, is presented. The radiologic changes were followed over 6 months until spontaneous resolution had occurred. A computed tomography (CT) was performed, which better demonstrated the presence of a single left-sided cavity with an air/fluid level within a contused area of the upper lobe and a multilocular cavity in the right lower lobe. The mechanism of formation of traumatic pseudocysts and the diagnostic implications are discussed. 10 refs., 5 figs

  8. Atresia of the bilateral pulmonary veins: a rare and dismal anomaly identified on cardiac CT

    Energy Technology Data Exchange (ETDEWEB)

    Goo, Hyun Woo; Park, Sang-Hyub; Koo, Hyun Jung; Cho, Young Hoon; Lee, Eunsol [University of Ulsan College of Medicine, Asan Medical Center, Department of Radiology and Research Institute of Radiology, Seoul (Korea, Republic of)

    2014-08-15

    Imaging findings of bilateral pulmonary vein atresia have not been described. To describe cardiac CT findings and clinical outcomes of bilateral pulmonary vein atresia. Three newborns with bilateral pulmonary vein atresia were encountered at our institution during a period of 8 years. We evaluated prenatal echocardiographic findings, clinical presentations, postnatal echocardiographic findings, chest radiographic findings, cardiac CT findings and clinical outcomes. All newborns presented immediately after birth with severe cyanosis, respiratory distress and acidosis that were unresponsive to medical management. Prenatal and postnatal echocardiographic studies and chest radiography were misleading, inconclusive or nonspecific in making the diagnosis in these children; however cardiac CT clearly demonstrated atresia of the bilateral pulmonary veins with multiple small mediastinal collateral veins and pulmonary edema. Surgical treatments were not feasible for this anomaly. Their clinical outcomes were universally dismal and all infants died within 3 days. Cardiac CT provides an accurate diagnosis of bilateral pulmonary vein atresia and leads to prompt treatment decision in these children. (orig.)

  9. Atresia of the bilateral pulmonary veins: a rare and dismal anomaly identified on cardiac CT

    International Nuclear Information System (INIS)

    Imaging findings of bilateral pulmonary vein atresia have not been described. To describe cardiac CT findings and clinical outcomes of bilateral pulmonary vein atresia. Three newborns with bilateral pulmonary vein atresia were encountered at our institution during a period of 8 years. We evaluated prenatal echocardiographic findings, clinical presentations, postnatal echocardiographic findings, chest radiographic findings, cardiac CT findings and clinical outcomes. All newborns presented immediately after birth with severe cyanosis, respiratory distress and acidosis that were unresponsive to medical management. Prenatal and postnatal echocardiographic studies and chest radiography were misleading, inconclusive or nonspecific in making the diagnosis in these children; however cardiac CT clearly demonstrated atresia of the bilateral pulmonary veins with multiple small mediastinal collateral veins and pulmonary edema. Surgical treatments were not feasible for this anomaly. Their clinical outcomes were universally dismal and all infants died within 3 days. Cardiac CT provides an accurate diagnosis of bilateral pulmonary vein atresia and leads to prompt treatment decision in these children. (orig.)

  10. An Unusual Radiologic Manifestation of Pulmonary Tuberculosis with Bilateral Multiple Lung Nodules and Diffuse Alveolar Hemorrhage: A Case Report

    International Nuclear Information System (INIS)

    Pulmonary tuberculosis presenting as bilateral multiple lung nodules or diffuse alveolar hemorrhage is very rare. Here, we report a case of pulmonary tuberculosis presenting as bilateral multiple lung nodules and diffuse alveolar hemorrhage mimicking granulomatous vasculitis, such as Wegener's granulomatosis.

  11. Case Report: Bilateral reexpansion pulmonary edema following treatment of a unilateral hemothorax

    OpenAIRE

    Steven P de Wolf; Deunk, Jaap; Cornet, Alexander D.; Elbers, Paul WG; Farag, Ehab; Coetzee, André

    2014-01-01

    Bilateral re-expansion pulmonary edema (RPE) is an extremely rare entity. We report the unique case of bilateral RPE following a traumatic, unilateral hemopneumothorax in a young healthy male. Bilateral RPE occurred only one hour after drainage of a unilateral hemopneumothorax. The patient was treated with diuretics and supplemental oxygen. Diagnosis was confirmed by excluding other causes, using laboratory findings, chest radiography, pulmonary and cardiac ultrasound and high resolution comp...

  12. Penile agenesis

    OpenAIRE

    Bangroo A; Khetri Ramji; Tiwari Sashi

    2005-01-01

    Penile agenesis is an extremely rare disorder with profound urological and psychological consequences. The goal of treatment is an early female gender assignment and feminizing reconstruction of the perineum.

  13. Case Report: Bilateral reexpansion pulmonary edema following treatment of a unilateral hemothorax.

    Science.gov (United States)

    de Wolf, Steven P; Deunk, Jaap; Cornet, Alexander D; Elbers, Paul Wg

    2014-01-01

    Bilateral re-expansion pulmonary edema (RPE) is an extremely rare entity. We report the unique case of bilateral RPE following a traumatic, unilateral hemopneumothorax in a young healthy male. Bilateral RPE occurred only one hour after drainage of a unilateral hemopneumothorax. The patient was treated with diuretics and supplemental oxygen. Diagnosis was confirmed by excluding other causes, using laboratory findings, chest radiography, pulmonary and cardiac ultrasound and high resolution computed tomography. His recovery was uneventful. The pathophysiology of bilateral RPE is not well known. Treatment is mainly supportive and consists of diuretics, mechanical ventilation, inotropes and steroids. In case of a pulmonary deterioration after the drainage of a traumatic pneumothorax, bilateral RPE should be considered after exclusion of more common causes of dyspnea. PMID:25713699

  14. Pulmonary Langerhans cell histiocytosis causing spontaneous bilateral pneumothorax in a child

    OpenAIRE

    Anupam Patra; Sujit K Bhattacharyay; Arnab Maji; Abhijit Mandal

    2015-01-01

    Bilateral pneumothorax is very rare in childhood. Moreover, if it is due to pulmonary involvement of Langerhans cell histiocytosis, it is even rarer in childhood. In our case, a nonsmoker 12-year-old boy presented with bilateral pneumothorax, whose high-resolution computed tomography scan was highly suggestive of pulmonary Langerhans cell histiocytosis. Excision biopsy of a clinically palpable cervical lymph node and histopathological examination and immunohistochemistry positivity for CD1a i...

  15. Radiological diagnosis of pulmonary sequestration: review of six cases, including one bilateral

    International Nuclear Information System (INIS)

    Radiological diagnosis of pulmonary sequestration: review of six cases, including one bilateral. Pulmonary sequestration is an uncommon disorder consisting of aberrant pulmonary tissue that has no normal connection with the bronchial tree or with the pulmonary arteries, but is supplied by a systemic artery which usually arises from the aorta. Six cases of pulmonary sequestration are presented and the radiological manifestation of this rare congenital disorder are discussed. These sequestrations were intralobar/unilateral in four patients, extralobar/unilateral in one and extralobar/bilateral in the other patient. Special attention is given to the extremely uncommon bilateral sequestration. To our knowledge only four cases of this form of disease has been described in the literature. (author)

  16. Pulmonary Langerhans cell histiocytosis causing spontaneous bilateral pneumothorax in a child

    Directory of Open Access Journals (Sweden)

    Anupam Patra

    2015-01-01

    Full Text Available Bilateral pneumothorax is very rare in childhood. Moreover, if it is due to pulmonary involvement of Langerhans cell histiocytosis, it is even rarer in childhood. In our case, a nonsmoker 12-year-old boy presented with bilateral pneumothorax, whose high-resolution computed tomography scan was highly suggestive of pulmonary Langerhans cell histiocytosis. Excision biopsy of a clinically palpable cervical lymph node and histopathological examination and immunohistochemistry positivity for CD1a indicated a diagnosis of Langerhans cell histiocytosis. Clinicians should consider pulmonary Langerhans cell histiocytosis in differential diagnoses in dealing such a case.

  17. Episode of massive pulmonary embolism after bilateral breast augmentation

    Science.gov (United States)

    Schonauer, Fabrizio; Nele, Gisella; Di Martino, Annalena; Santoro, Mariangela; Santanelli di Pompeo, Fabio

    2015-01-01

    Pulmonary embolism is a rare postsurgical complication, even more so following breast augmentation. Herein we present a case of a 23-year-old woman who survived an episode of massive pulmonary embolism after breast implant surgery. Current literature about this subject is very scarce.

  18. Pulmonary Langerhans Cell Histiocytosis X Presented with Bilateral Pneumothorax: A Case Report

    OpenAIRE

    Majed Al-Mourgi

    2014-01-01

    Background: The report describes the case of a 20-year-old Saudi male patient with Pulmonary Langerhans cell histiocytosis X (PLCH). He presented with bilateral chest pain of few hours duration starting first on the right side. The patient had also dyspnea and non productive cough. The patient was heavy smoker for 6 years. General examination revealed that the patient had tachypnea but he was hemodynamically stable. Chest examination showed bilateral distant breath sounds but no hyperresonanc...

  19. Compression of the Right Pulmonary Artery by a Massive Descending Aortic Aneurysm Causing Bilateral Perfusion Defects on Pulmonary Scintigraphy

    OpenAIRE

    Makis, William; Derbekyan, Vilma

    2011-01-01

    A 67-year-old woman, who presented with a 2 month history of dyspnea, had a ventilation and perfusion lung scan that showed absent perfusion of the entire right lung with normal ventilation, as well as a rounded matched defect in the left lower lung adjacent to midline, suspicious for an aortic aneurysm or dissection. CT pulmonary angiography revealed a massive descending aortic aneurysm compressing the right pulmonary artery as well as the left lung parenchyma, accounting for the bilateral p...

  20. Coinfection of pulmonary mucormycosis and aspergillosis presenting as bilateral vocal cord palsy

    Science.gov (United States)

    Mahadevaiah, Arun H; Rajagopalan, Natarajan; Patil, Mahantesh; C, Shivaprasad

    2013-01-01

    Invasive pulmonary coinfection with Mucor and Aspergillus is rare. Bilateral vocal cord paralysis resulting from coinfection with these two fungi to our knowledge has not been reported in the literature. We report a young woman with diabetes who presented with symptoms of community acquired pneumonia in association with hoarseness of voice. Investigations revealed air space consolidation of the right upper lobe, evidence of mediastinal involvement extending into the paratracheal space and entrapment of right upper lobe pulmonary artery. Bronchoscopy revealed bilateral vocal cord paralysis and sloughing of mucosa of the right upper lobe bronchus and the bronchus intermedius. Microbiological and pathological results confirmed Mucor and Aspergillus. Extensive vascular and mediastinal involvement precludes surgical debridement. Despite aggressive medical management the patient deteriorated and died of respiratory failure. Strong suspicion of invasive fungal infections in immune compromised patients presenting with unresolving pneumonia and hoarseness of voice, early aggressive treatment is crucial for the patient survival. PMID:23964036

  1. Bilateral perinephric fluid accumulation: an unusual manifestation of pulmonary hypertension--a case report.

    Science.gov (United States)

    Pentimone, F; Meola, M; Del Corso, L

    1993-06-01

    This is the case report of a forty-seven-year-old man with an ostium secundum atrial septal defect and a very high grade of pulmonary hypertension, associated with a large bilateral perinephric fluid accumulation. The fluid accumulation was remarkably reduced after eleven phlebotomies over a twelve-month period. A pathogenetic relation with the Eisenmenger's syndrome is discussed. No previous report of this association has been found in a survey of the literature. PMID:8503517

  2. PULMONARY LANGERHANS CELL HISTIOCYTOSIS PRESENTING AS SIMULTANEOUS BILATERAL SPONTANEOUS PNEUMOTHORAX IN A NON-SMOKER PATIENT

    OpenAIRE

    M. Vaziri; A. Pazooki L. Zahedi

    2008-01-01

    Pulmonary Langerhans Cell Histiocytosis (PLCH) is a rare idiopathic disorder that primarily affects young adult cigarette smokers. Affected patients often present with cough and dyspnea and about 20% of patients present with or later develop pneumothorax. It is striking that more than 90% of patients are smokers. We report a very unusual case of PLCH in a 20-year- old male patient with no smoking history in whom a life- threatening complication such as simultaneous bilateral pneumothorax was ...

  3. Mini-invasive resection and collapse therapy in patients with bilateral pulmonary tuberculosis

    Directory of Open Access Journals (Sweden)

    Korpusenko I.V.

    2015-06-01

    Full Text Available Objective. Improve the effectiveness of surgical treatment in patients with bilateral destructive pulmonary tuberculosis by mini-invasive resection and collapse therapy. Materials and Methods: Retrospective analysis of 222 patients’ cards with bilateral destructive pulmonary tuberculosis who were treated in the period from 1995 to 2014 in the thoracic department of Dnepropetrovsk regional clinical therapeutic and prophylactic association "Phthisiology". Patients were divided into 2 groups: basic (111 patients who underwent mini-invasive surgery and control (111 patients, who underwent standard surgical approach. The distribution of patients in investigated groups was representative by the majority of parameters. Results and discussion. The average duration of simultaneous bilateral VATS lung resections was 1,90 ± 0,12 hour, standard thoracotomies - 2,13 ± 0,19 per hour, estimated blood loss was 234±5,20ml and 433±3,70ml respectively. The average postoperative time in-patient was 52,40±2,63 days in basic and 80,10±3,58 days in the control group. Number of postoperative complications after lung resection with VATS was significantly lower (1.6 times, as compared with standard surgical approach. Volume of blood loss less than 400 ml was 93,40±3,20% in basic and 72,60±4,80% in the control group, the amount of intraoperative complications reduced by 2.2 times. Complete clinical response (decontamination and closing of cavities have been achieved in patients of the basic group by 1.6 times more often. Conclusions: For patients with bilateral pulmonary tuberculosis to perform mini-invasive surgical approach is the best option. Mini-invasive interventions with VATS due to its good abilities to visualize tissues and anatomical structures may significantly decrease the amount of intraoperative blood and plasma loss in the first postoperative day. It leads to the stabilization of tuberculosis process in the contralateral lung, responsible for

  4. Bilateral radial agenesis with absent thumbs, complex heart defect, short stature, and facial dysmorphism in a patient with pure distal microduplication of 5q35.2-5q35.3

    Directory of Open Access Journals (Sweden)

    Jamsheer Aleksander

    2013-01-01

    Full Text Available Abstract Background A partial duplication of the distal long arm of chromosome 5 (5q35-- > qter is known to be associated with a distinct phenotype referred to as Hunter-McAlpine syndrome. Clinical spectrum of this disorder mainly consists of mental retardation, microcephaly, short stature, skeletal anomalies, and craniofacial dysmorphism featuring flat facies, micrognathia, large, low-set dysplastic ears, hypertelorism, almond-shaped, down-slanted palpebral fissures, epicanthal folds, small nose, long philtrum, small mouth, and thin upper lip. Less frequent remarkable findings include craniosynostosis, heart defect, hypoplastic phalanges, preaxial polydactyly, hypospadias, cryptorchidism, and inguinal hernia. In most patients with a partial duplication of 5q the aberration occurred due to an inherited unbalanced translocation, therefore the phenotype was not reflective of pure trisomy 5q. Case presentation We report on a 9.5-year-old boy with some feature of Hunter-McAlpine syndrome including short stature, complex heart defect (dextrocardia, dextroversion, PFO, bilateral cryptorchidism, hypothyroidism, and craniofacial dysmorphism. Additionally, bilateral radial agenesis with complete absence of Ist digital rays, ulnar hypoplasia with bowing, choroidal and retinal coloboma, abnormal biliary vesicle were identified, which have never been noted in 5q trisomy patients. Karyotype analysis, sequencing and MLPA for TBX5 and SALL4 genes were unremarkable. Array comparative genomic hybridization detected a duplication on 5q35.2-5q35.3, resulting from a de novo chromosomal rearrangement. Our proband carried the smallest of all previously reported pure distal 5q trisomies encompassing terminal 5.4-5.6 Mb and presented with the most severe limb malformation attributed to the increased number of distal 5q copies. Conclusions We postulate that a terminal distal trisomy of 5q35.2-5q35.3, which maps 1.1 Mb telomeric to the MSX2 gene is causative for both

  5. Pulmonary endothelial dysfunction induced by unilateral as compared to bilateral thoracic irradiation in rats

    International Nuclear Information System (INIS)

    Rats were sacrificed 2 months after a single dose of 10-30 Gy of 60Co gamma rays delivered to either a right unilateral or a bilateral thoracic port. Four indices of lung endothelial function were measured: the activities of angiotensin-converting enzyme (ACE) and plasminogen activator (PLA) and the production of prostacyclin (PGI2) and thromboxane (TXA2). The number of macrophages recovered by bronchoalveolar lavage (BAL) and the degree of right ventricular hypertrophy (an index of pulmonary hypertension) also were determined. Right lung ACE and PLA activity decreased linearly, and PGI2 and TXA2 production increased linearly with increasing radiation dose. The response curves for right unilateral and bilateral thoracic irradiation were not significantly different. In contrast, bilateral irradiation was more toxic than unilateral, since rats exposed to the former exhibited decreased body weight, an increased incidence of pleural effusions, an increase in the number of macrophages recovered by BAL, and right ventricular hypertrophy. These data demonstrate that pulmonary endothelial dysfunction induced by hemithorax irradiation represents a direct response of the endothelium to radiation injury and is not secondary to other phenomena such as shunting of function to the shielded lung

  6. CT of tracheal agenesis

    International Nuclear Information System (INIS)

    Tracheal agenesis is a rare and usually lethal anomaly. In the past, opaque contrast medium was injected via the esophagus to demonstrate the anatomy. To demonstrate the utility of helical and multidetector CT in delineating the aberrant anatomy in newborns with tracheal agenesis. Four newborns with tracheal agenesis were identified from three institutions. Imaging studies and medical records were reviewed. Each child was imaged with chest radiography. One child was imaged on a single-detector helical CT scanner and the other three on multidetector scanners. Helical and multidetector CT with 2D and 3D reconstructions clearly delineated the aberrant tracheobronchial and esophageal anatomy in each infant. Minimum intensity projection reformatted CT images were particularly helpful. One infant each had type I and type II tracheal agenesis. Two infants had type III tracheal agenesis. All four infants died. CT is a useful tool for delineating the aberrant anatomy of newborns with tracheal agenesis and thus helps in making rational clinical decisions. (orig.)

  7. CT of tracheal agenesis

    Energy Technology Data Exchange (ETDEWEB)

    Strouse, Peter J.; Hernandez, Ramiro J. [C.S. Mott Children' s Hospital, Department of Radiology, Ann Arbor, MI (United States); Newman, Beverley [Children' s Hospital of Pittsburgh, PA (United States). Department of Pediatric Radiology; Afshani, Ehsan [Children' s Hospital of Buffalo, NY (United States). Departments of Radiology and Pediatrics; Bommaraju, Mahesh [Women' s and Children' s Hospital of Buffalo, Division of Neonatology, University Pediatrics Associates, Buffalo, NY (United States)

    2006-09-15

    Tracheal agenesis is a rare and usually lethal anomaly. In the past, opaque contrast medium was injected via the esophagus to demonstrate the anatomy. To demonstrate the utility of helical and multidetector CT in delineating the aberrant anatomy in newborns with tracheal agenesis. Four newborns with tracheal agenesis were identified from three institutions. Imaging studies and medical records were reviewed. Each child was imaged with chest radiography. One child was imaged on a single-detector helical CT scanner and the other three on multidetector scanners. Helical and multidetector CT with 2D and 3D reconstructions clearly delineated the aberrant tracheobronchial and esophageal anatomy in each infant. Minimum intensity projection reformatted CT images were particularly helpful. One infant each had type I and type II tracheal agenesis. Two infants had type III tracheal agenesis. All four infants died. CT is a useful tool for delineating the aberrant anatomy of newborns with tracheal agenesis and thus helps in making rational clinical decisions. (orig.)

  8. Fatal bilateral lower-limb deep vein thrombosis and pulmonary embolism following single digit replantation.

    Science.gov (United States)

    Leung, Anderson S M; Fok, Margaret W M; Fung, Boris K K

    2015-06-01

    Venous thromboembolism in hand surgery is rare. There is no report in the literature on postoperative mortality from venous thromboembolism following microsurgery in upper limbs. We report the case of a 56-year-old Chinese man who died from pulmonary embolism as a result of bilateral lower-limb deep vein thrombosis following prolonged surgery under general anaesthesia after replantation of a finger. This case raises awareness of the need for precautions against venous thromboembolism following prolonged microsurgery and identification of high-risk patients. PMID:26045073

  9. Pulmonary endothelial dysfunction induced by unilateral as compared to bilateral thoracic irradiation in rats

    International Nuclear Information System (INIS)

    Rats were sacrificed 2 mo after a single dose of 10-30 Gy of /sup 60/Co gamma rays delivered to either a right unilateral or a bilateral thoracic port. Four indices of lung endothelial function were measured: the activities of angiotensin converting enzyme (ACE) and plasminogen activator (PLA), and the production of prostacyclin (PGL/sub 2/) and thromboxane (TXA/sub 2/). The experimental results are given. These data indicate that pulmonary endothelial dysfunction induced by hemithoracic irradiation represents a direct response of a endothelium to injury, and is not secondary to other phenomena such as shunting of function to the contralateral lung

  10. Reconstruction of vaginal agenesis.

    Science.gov (United States)

    Ozkan, Ozlenen; Erman Akar, Münire; Ozkan, Omer; Doğan, N Utku

    2011-06-01

    Vaginal ageneses are by no means rare anomalies. Complete Mullerian agenesis is the most common reason for vaginal agenesis requiring reconstruction. Patients usually present with pain, hematocolpos, or hematometra in puberty, and later with amenorrhea and dyspareunia. Detailed information is given here regarding etiologies, timing of surgery, and current treatment options for vaginal agenesis. Outcomes and short- and long-term complications of recent treatment options are also discussed. PMID:21372677

  11. Agenesis of pancreas

    DEFF Research Database (Denmark)

    Voldsgaard, P; Kryger-Baggesen, N; Lisse, I

    1994-01-01

    Complete agenesis of pancreas is a rare and lethal condition. Four cases have previously been reported in combination with other malformations, such as severe intrauterine growth retardation, hyperglycaemia and meconium ileus. We report a case of pancreatic agenesis as a single anomaly. The child...

  12. Minimally invasive surgical treatment of patients with bilateral pulmonary tuberculosis complicated with pleural empyema

    Directory of Open Access Journals (Sweden)

    Korpusenko I.V

    2015-03-01

    Full Text Available Objective of our study was to increase the effectiveness of surgical treatment of bilateral destructive pulmonary tuberculosis complicated by pleural empyema by using VTS-technologies. The study was done in Dnepropetrovsk regional clinical therapeutic and prophylactic association "Phthisiology" in the period from 2008 to 2013. A retrospective analysis of 43 cases of bilateral destructive pulmonary tuberculosis complicated by pleural empyema on one side and dissemination focus or limited destructive process on contralateral side has been performed. Selected cases were divided into 2 groups: main (eighteen cases where the following procedures were done: performed transsternal occlusion of the main bronchus, sanation of empyema cavity using videothoracosopy, in 30-45 days followed by pleuropneumectomy with usage of minithoracothomy and control (nineteen cases who had undergone drainage of the empyema cavity, sanation, in 45-60 days followed by pleuropmeumectomy with usage of anterolateral access. The distribution of main and control groups for analyzed parameters was representative. Sanation of pleural cavity with videothoracosopy usage compared with Bulau’s drainage provides better antibacterial effect, effective sanitation of the pleural cavity as evidenced by following changes: significant decrease in the number of microbial cells; normalization of total white blood cells count and rod-shaped granulocytes in the peripheral blood 10 days after treatment; normalization of leukocyte intoxication index. The use of minimally invasive surgical treatment allowed to reduce intraoperative complications by 2 times, amount of intraoperative blood loss and hemotrasfusions by 1.5 times, postoperative mortality by 2.5 times. Pleural cavity sanation with videothoracoscopy usage with following pneumoectomy leads to reduce in the incidence and severity of postoperative complications. The most promising is stage-by-stage surgical approach with consecutive use

  13. Case Report: Bilateral reexpansion pulmonary edema following treatment of a unilateral hemothorax [v1; ref status: indexed, http://f1000r.es/4yb

    OpenAIRE

    Steven P de Wolf; Jaap Deunk; Cornet, Alexander D.; Paul WG Elbers

    2014-01-01

    Bilateral re-expansion pulmonary edema (RPE) is an extremely rare entity. We report the unique case of bilateral RPE following a traumatic, unilateral hemopneumothorax in a young healthy male. Bilateral RPE occurred only one hour after drainage of a unilateral hemopneumothorax. The patient was treated with diuretics and supplemental oxygen. Diagnosis was confirmed by excluding other causes, using laboratory findings, chest radiography, pulmonary and cardiac ultrasound and high resolution comp...

  14. PULMONARY LANGERHANS CELL HISTIOCYTOSIS PRESENTING AS SIMULTANEOUS BILATERAL SPONTANEOUS PNEUMOTHORAX IN A NON-SMOKER PATIENT

    Directory of Open Access Journals (Sweden)

    M. Vaziri

    2008-06-01

    Full Text Available Pulmonary Langerhans Cell Histiocytosis (PLCH is a rare idiopathic disorder that primarily affects young adult cigarette smokers. Affected patients often present with cough and dyspnea and about 20% of patients present with or later develop pneumothorax. It is striking that more than 90% of patients are smokers. We report a very unusual case of PLCH in a 20-year- old male patient with no smoking history in whom a life- threatening complication such as simultaneous bilateral pneumothorax was the presenting feature. The final diagnosis was made by open surgical biopsy and recurrent pneumothoraces necessitated surgical management with pleurodesis. We emphasize the early use of pleurodesis in managing patients with PLCH and spontaneous pneumothorax.

  15. Case Report: Bilateral reexpansion pulmonary edema following treatment of a unilateral hemothorax [v1; ref status: indexed, http://f1000r.es/4yb

    Directory of Open Access Journals (Sweden)

    Steven P de Wolf

    2014-12-01

    Full Text Available Bilateral re-expansion pulmonary edema (RPE is an extremely rare entity. We report the unique case of bilateral RPE following a traumatic, unilateral hemopneumothorax in a young healthy male. Bilateral RPE occurred only one hour after drainage of a unilateral hemopneumothorax. The patient was treated with diuretics and supplemental oxygen. Diagnosis was confirmed by excluding other causes, using laboratory findings, chest radiography, pulmonary and cardiac ultrasound and high resolution computed tomography. His recovery was uneventful. The pathophysiology of bilateral RPE is not well known. Treatment is mainly supportive and consists of diuretics, mechanical ventilation, inotropes and steroids. In case of a pulmonary deterioration after the drainage of a traumatic pneumothorax, bilateral RPE should be considered after exclusion of more common causes of dyspnea.

  16. Two cases of sacral agenesis

    International Nuclear Information System (INIS)

    Sacral agenesis is a central component in the spectrum of anomalies comprising the caudal regression syndrome. Sacral agenesis occurs more frequently in an infant born to mother of diabetes, rubella infection in first trimester than normal mothers. In a patient of sacral agenesis, it is important to recognize the neurologic deficit, neurogenic bladder dysfunction and other congenital anomalies. A case of partial sacral agenesis of 10 years old girl and another case of complete total agenesis associated with multiple anomalies in autopsied newborn are reported.

  17. Two cases of sacral agenesis

    Energy Technology Data Exchange (ETDEWEB)

    Choi, J. Y.; Bae, Y. K.; Hahm, C. K.; Kang, S. R. [Hanyang University College of Medicine, Seoul (Korea, Republic of)

    1979-06-15

    Sacral agenesis is a central component in the spectrum of anomalies comprising the caudal regression syndrome. Sacral agenesis occurs more frequently in an infant born to mother of diabetes, rubella infection in first trimester than normal mothers. In a patient of sacral agenesis, it is important to recognize the neurologic deficit, neurogenic bladder dysfunction and other congenital anomalies. A case of partial sacral agenesis of 10 years old girl and another case of complete total agenesis associated with multiple anomalies in autopsied newborn are reported.

  18. Successful Management of Intraoperative Acute Bilateral Pulmonary Embolism in a High Grade Astrocytoma Patient.

    Science.gov (United States)

    Khraise, Wail N; Allouh, Mohammed Z; Hiasat, Mohammad Y; Said, Raed S

    2016-01-01

    BACKGROUND Intraoperative pulmonary embolism (PE) is a rare life-threatening complication in patients undergoing surgical intervention. Generally, cancer patients have a higher risk for developing this complication. Unfortunately, there is no standard procedure for its management. CASE REPORT We report the case of a 39-year-old woman with high-grade glioma in the right frontal lobe who was admitted to the surgical theater for craniotomy and excision of the tumor. During the general anesthesia procedure and just before inserting the central venous line, her end-tidal CO2 and O2 saturation dropped sharply. The anesthesiologist quickly responded with an aggressive resuscitation procedure that included aspiration through the central venous line, 100% O2, and IV administration of ephedrine 6 mg, colloid 500 mL, normal saline 500 mL, and heparin 5000 IU. The patient was extubated and remained in the supine position until she regained consciousness and her vital signs returned to normal. Subsequent radiological examination revealed a massive bilateral PE. A retrievable inferior vena cava (IVC) filter was inserted, and enoxaparin anticoagulant therapy was prescribed to stabilize the patient's condition. After 3 weeks, she underwent an uneventful craniotomy procedure and was discharged a week later under the enoxaparin therapy. CONCLUSIONS The successful management of intraoperative PE requires a quick, accurate diagnosis accompanied with an aggressive, fast response. Anesthesiologists are usually the ones who are held accountable for the diagnosis and early management of this complication. They must be aware of the possibility of such a complication and be ready to react properly and decisively in the operation theater. PMID:27578311

  19. IVC agenesis: a rare cause of deep vein thrombosis.

    Science.gov (United States)

    Man, Louise; Hendricks, Nicholas; Maitland, Hillary

    2016-04-01

    We present the case of a healthy, young Caucasian female who presented to an outside hospital with phlegmasia cerulea dolens of both lower extremities. Computed tomography angiography revealed inferior vena cava (IVC) occlusion. She was initiated on heparin infusion and transferred to University of Virginia Medical Center. Our evaluation revealed aplasia of the IVC from the infrahepatic segment to the confluence of the common iliac veins and acute bilateral iliac vein thromboses. An extensive network of collateral veins was noted. These findings were consistent with IVC agenesis. She was not pregnant or using contraception. Primary thrombophilia workup was negative. She underwent bilateral iliac vein thrombolysis and was started on anticoagulation. While IVC agenesis is rare, it carries risk for development of thrombotic sequelae and bears consideration when evaluating young patients with unexplained deep vein thrombosis, especially if extensive and bilateral. PMID:26507132

  20. Unilateral renal agenesis associated with ovarian cysts in a 19 year old woman in Orlu, South-East Nigeria

    Directory of Open Access Journals (Sweden)

    Anyabolu E.N

    2015-10-01

    Full Text Available Background: Anomalies linked with unilateral renal agenesis are not completely known. Aim: We report here a rare occurrence of ovarian cysts associated with unilateral renal agenesis in Orlu, Nigeria. Methods: A 19 year-old woman was being evaluated for acute pyelonephritis. Findings: We noted that this patient had acute pyelonephritis on a background of right renal agenesis that was associated with bilateral ovarian cysts. Ovarian cyst in the index patient was an incidental finding whereas acute pyelonephritis, perhaps, was a complication of unilateral renal agenesis. Conclusion: This case report highlights the need for consideration of possibility of ovarian cysts during evaluation of unilateral renal agenesis as many abnormalities of the ovaries, uterus and vagina are associated with renal agenesis and may impact negatively on fertility.

  1. Moebius syndrome with Dandy-Walker variant and agenesis of corpus callosum

    Directory of Open Access Journals (Sweden)

    Jomol Sara John

    2013-01-01

    Full Text Available Moebius syndrome is a rare congenital neurological disorder. The most frequent mode of presentation is facial diplegia with bilateral lateral rectus palsy, but there are variations. Here, we report a rare case of Moebius syndrome in a 15-month-old child with unilateral facial palsy, bilateral abducens nerve palsy with Dandy Walker variant, and complete agenesis of corpus callosum.

  2. Post-operative Unexpected Bilateral Massive Pulmonary Embolism in a Thirty-Year-Old Woman: A Case Report

    Directory of Open Access Journals (Sweden)

    Öznur Şen

    2015-12-01

    Full Text Available Pulmonary embolism (PE is a rare, yet serious complication. That, generally follows invasive procedures in adults. The present report details the case of bilateral pulmonary embolism in a thirty-year-old women after ovarian cyst surgery. A thirty-year-old female patient, who had ovarian cystectomy ten days previously, consulted the emergency department with the complaints of sickness and syncope. Her blood pressure, pulse and respiratory rate were 90/40 mmHg, 150/min. and 35/min, respectively. During examination and phlebotomy, she had a cardiac arrest and responded to a 3-minute cardiopulmonary resuscitation (CPR with tracheal intubation. During imaging procedures, she had three further cardiac arrests and was admitted to the intensive care unit. The preliminary diagnoses were acute renal failure (ARF and PE. Fluid replacement, inotropic support, anticoagulant therapies, as well as sedation to prevent brain damage were started immediately. Thrombolytic therapy was not attempted because she had surgery ten days ago and hematoma was observed on abdominal ultrasound. Inhaled nitric oxide and inhaled iloprost with sildenafil were employed to reduce pulmonary hypertension. When discharged 136 days after hospitalization she was a tracheotomized and, spontaneously breathing home care patient with hypoxic brain injury. Early detection and treatment of PE are of great importance for a good prognosis and prevention of life-threatening complications.

  3. Bilobar thyroid agenesis with primary hyperparathyroidism: report of a case.

    Science.gov (United States)

    Şimşek, Turgay; Cantürk, Nuh Zafer; Cantürk, Zeynep; Gürbüz, Yeşim

    2015-06-01

    Congenital thyroid abnormalities are rarely seen. They mostly include hemiagenesis with or without involving the isthmus. In this report, we present a case of bilateral lobe agenesis with hypertrophied isthmus and high calcium and elevated PTH levels which were detected during routine examinations of a 49-year-old female patient. Some findings consistent with parathyroid pathology on the right side were determined in parathyroid scintigraphy. At thyroid scan and neck ultrasonography there was no sign of bilateral thyroid tissue except a mass localized isthmus. The right lower parathyroidectomy and thyroidectomy for isthmus were performed; the pathology report was shown as parathyroid adenoma and nodular colloidal goiter. This case of bilobar agenesis with incidental primary hyperparathyroidism due to single parathyroid adenoma is the first case reported in literature. PMID:25091455

  4. Three new patients with FATCO: fibular agenesis with ectrodactyly.

    Science.gov (United States)

    Bieganski, Tadeusz; Jamsheer, Aleksander; Sowinska, Anna; Baranska, Dobromila; Niedzielski, Kryspin; Kozlowski, Kazimierz; Czarny-Ratajczak, Malwina

    2012-07-01

    We document three new patients with fibular agenesis, tibial campomelia, and oligosyndactyly (FATCO). Two of these individuals had tetramelic manifestations while the third had bilateral abnormalities of the lower limbs. These patients and others reported as FATCO seem to belong to the phenotype "fibular aplasia with ectrodactyly." Genetic screening for CNVs and mutations in the TP63 and WNT10B genes did not show any genetic abnormalities. © PMID:22628253

  5. Agenesis of the dorsal pancreas

    Institute of Scientific and Technical Information of China (English)

    Lale Pasaoglu; Murat Vural; Hatice Gul Hatipoglu; Gokce Tereklioglu; Suha Koparal

    2008-01-01

    Developmental anomalies of the pancreas have been reported but dorsal pancreatic agenesis is an extremely rare entity. We report an asymptomatic 62-year-old woman with complete agenesis of the dorsal pancreas.Abdominal computed tomography (CT) revealed a normal pancreatic head, but pancreatic body and tail were not visualized. Magnetic resonance imaging (MRI)findings were similar to CT. At magnetic resonance cholangiopancreatography (MRCP), the major pancreatic duct was short and the dorsal pancreatic duct was not visualized. The final diagnosis was dorsal pancreatic agenesis.

  6. Incidence of agenesis of palmaris longus in the Andhra population of India

    Directory of Open Access Journals (Sweden)

    K Devi Sankar

    2011-01-01

    Full Text Available Background : The knowledge of Palmaris longus (PL is a growing interest for its wide role in reconstructive plastic surgeries as a donor tendon for transfer or transplant. The prevalence of the PL agenesis has been well-documented by many authors in different ethnic groups or populations. Many conventional tests for determining the presence of the PL has been described, but lamentably there are many discrepancies in confirming its presence or absence. Slight modifications of the prevailing methods can still give authenticate results. Aim : This prospective study was conducted to determine the incidence of unilateral and bilateral agenesis of PL and its association with sex and side of the limb in the Andhra population of India. Materials and Methods : A total of 942 subjects of both sexes belonging to 18-23 years were used to access the PL using various tendon examination techniques including our modified Schaeffer′s test. The data collected were analyzed by Pearsons χ2 test using SPSS software. Results : Overall agenesis of muscle in both sexes was 264 (28.0%, out of which 40.2% was seen in females and 14.7% in males with the ratio of 3:1. The unilateral agenesis was seen in 70.5% and bilateral agenesis in 29.5% subjects. The left side agenesis was seen in 51.6% and right side in 48.4% subjects. Conclusions : The prevalence of bilateral and unilateral agenesis was more common on left side with a greater likelihood in the female subjects. The proposed technique could bring better results in all subjects and can be implemented in manual examination of PL.

  7. Semen analysis in an infertile man with seminal vesicles cysts associated with ipsilateral renal agenesis.

    Science.gov (United States)

    Andrade-Rocha, Fernando Tadeu

    2006-01-01

    A case of an infertile man with bilateral seminal vesicles cysts with ipsilateral renal agenesis is presented with emphasis on the semen analysis. Ejaculates showed very low concentration of fructose, leukocytospermia, teratozoospermia, asthenozoospermia, hyperviscosity, marked sperm agglutination and a poor hypoosmotic swelling test. Clinical value of these findings is discussed. PMID:16502061

  8. Isolated complete corpus callosal agenesis

    Directory of Open Access Journals (Sweden)

    Jaiganesh S, Venkateshwaran A, Naresh Kumar C, Rajasekhar KV

    2014-11-01

    Full Text Available Isolated complete corpus callosal agenesis is a rare entity. Usually this condition will be an associated finding in other syndromes. 3 month old male child came with complaints of deformed foot on both sides, not having a social smile and neck holding. Patient referred to the Radiology department for MRI brain which showed complete absence of corpus callosum, widely separated and parallely placed lateral ventricles, colpocephaly, high riding of 3rd ventricle and absence of cingulate gyrus and radial arrangement of gyri along the interhemispheric fissure. Hence it was reported as isolated complete corpus callosal agenesis and this article describes the Embryogenesis, anatomy, developmental anomalies and its clinical manifestations & prognosis.

  9. Complete agenesis of dorsal pancreas

    Directory of Open Access Journals (Sweden)

    Malwinder Singh

    2014-04-01

    Full Text Available Introduction: Complete agenesis of body and tail of pancreas is a very rare type of developmental anomaly of pancreas. It is important regarding its presentations of diabetes mellitus, pancreatitis, and exocrine insufficiency. Case Report: An old man had presented with atypical symptoms of obstructive jaundice with exocrine insufficiency. CECT helped to reveal the complete absence of the body and tail of pancreas with radiologically normal head with no signs of pancreatitis or mass lesion. Conclusions: The cause of agenesis of the dorsal pancreas is currently not well understood. It can also present lately as the presenting case. The presentations are usually related to secretory malfunctions. CECT is an initial investigation for diagnosis

  10. Complete agenesis of dorsal pancreas

    OpenAIRE

    Malwinder Singh; Akhlak Hussain; Mohinder Singh; Sarabjit Singh

    2014-01-01

    Introduction: Complete agenesis of body and tail of pancreas is a very rare type of developmental anomaly of pancreas. It is important regarding its presentations of diabetes mellitus, pancreatitis, and exocrine insufficiency. Case Report: An old man had presented with atypical symptoms of obstructive jaundice with exocrine insufficiency. CECT helped to reveal the complete absence of the body and tail of pancreas with radiologically normal head with no signs of pancrea...

  11. Bilateral Congenital Diaphragmatic Hernia

    OpenAIRE

    Dhua, Anjan K; Aggarwal, Satish K; NB Mathur; GR Sethi

    2012-01-01

    Bilateral congenital diaphragmatic hernia (CDH) is a rare birth defect, with a poor prognosis. We describe a case of bilateral CDH discovered while repairing the right sided CDH. Diaphragmatic defect was repaired and a silo was applied on the abdominal wound to avoid abdominal compartment syndrome. The patient however died postoperatively due to severe pulmonary hypertension.

  12. Presentation of a keratocystic odontogenic tumor with agenesis: a case report

    OpenAIRE

    Lacarbonara, Mariano; Marzo, Giuseppe; Lacarbonara, Vitantonio; Monaco, Annalisa; Capogreco, Mario

    2014-01-01

    Introduction We analyzed the etiopathogenetic, clinical, radiographic, and histopathologic aspects of keratocystic odontogenic tumors, particularly in association with dental anomalies of number, with the aim of providing useful information for their correct diagnosis, treatment, and prognosis within a multidisciplinary approach. Case presentation A 14-year-old Caucasian girl presented for observation of bilateral agenesis of the upper incisors, which was diagnosed by orthopantomography. Appr...

  13. Tracheal agenesis with broncho-esophageal fistula in VACTERL / TACRD association

    OpenAIRE

    Mandrekar, Suresh R. S.; Sangeeta Amoncar; R. G. W. Pinto

    2013-01-01

    Tracheal agenesis (TA) is an extremely rare malformation. We report here autopsy findings in a case of TA with bronchoesophageal fistula of Floyd type III. The other malformations present included laryngeal atresia, Right lung hypolobulation, ventricular septal defect in membranous portion, bilateral cystic renal dysplasia, spleninculus, Meckel's diverticulum, and imperforate anus. The constellations of malformations present in our case have overlapping features with Vertebral anomalies, Anal...

  14. Seizure, deafness and renal agenesis: A rare case of barakat syndrome

    Directory of Open Access Journals (Sweden)

    Tanmayjyoti Sau

    2013-01-01

    Full Text Available Etiologic diagnosis of seizure requires proper consideration of apparently unrelated clinical features of the patient. Here, we report the case of a patient of status epilepticus with moderate-to-severe bilateral sensorineural deafness. Investigations showed extensive intracranial calcification, hypoparathyroidism and unilateral renal agenesis. The features were consistent with Barakat syndrome, a rare developmental disorder associated with mutations in the GATA3 gene. To the best of our knowledge, this is the first reported case of Barakat syndrome from India.

  15. Does asymptomatic septal agenesis exist? A review of 34 cases

    International Nuclear Information System (INIS)

    Primary septal agenesis (PSA) is a rare brain malformation that can be isolated or part of developmental brain abnormalities (holoprosencephaly, septo-optic dysplasia or cortical malformation). Such associated malformation can be subtle, leading to difficulties in the prenatal management of PSA. Moreover, the neurological prognosis of isolated PSA remains debatable. The aims of the study were to specify the patterns and frequency of brain malformations associated with septal agenesis (SA), to identify the clinical prognosis, and to discuss the aetiology of PSA with the new insights provided by molecular genetics. The study consisted of a 14-year retrospective review of brain MRI in 34 patients having PSA (mean age, 5 years). Chiasm and optic nerves were not evaluated. Post-hydrocephalus SA or incomplete data were excluded. The clinical data were correlated to the MRI patterns. The study disclosed 82.5% associated lesions with MRI (28/34): 11 neuronal migration disorders, 9 holoprosencephalies (HP), 7 pituitary stalk interruptions, 1 corpus callosum partial agenesis; 17.5% (6/34) of cases were apparently isolated PAS. Clinically, the patients had motor dysfunction in 68% (23/34), mental retardation in 65% (22/34), blindness in 24% (8/34), endocrinological defects in 21% (7/34) and epilepsy in 18% (6/34) of cases. Nine percent of patients (3/34) were neurologically normal (including one with scoliosis and two infants younger than 2 years at the last follow-up). Patients with bilateral cortical anomalies and HP (even if mild) had the worst neurological prognosis. A severe motor impairment was present without evidence of hemispheric anomaly in 12% of patients (4/34). Interestingly, the frontal lobes were involved in 90% of cortical anomalies and HP, supporting the malformative aetiology of PSA. PSA rarely appears isolated and severe psychomotor impairment may occur in apparently isolated forms. These unfavourable results should be highlighted and need to be confirmed

  16. Does asymptomatic septal agenesis exist? A review of 34 cases

    Energy Technology Data Exchange (ETDEWEB)

    Belhocine, Ouardia; Andre, Christine; Kalifa, Gabriel; Adamsbaum, Catherine [St Vincent de Paul Hospital, Radiology Department, Paris (France)

    2005-04-01

    Primary septal agenesis (PSA) is a rare brain malformation that can be isolated or part of developmental brain abnormalities (holoprosencephaly, septo-optic dysplasia or cortical malformation). Such associated malformation can be subtle, leading to difficulties in the prenatal management of PSA. Moreover, the neurological prognosis of isolated PSA remains debatable. The aims of the study were to specify the patterns and frequency of brain malformations associated with septal agenesis (SA), to identify the clinical prognosis, and to discuss the aetiology of PSA with the new insights provided by molecular genetics. The study consisted of a 14-year retrospective review of brain MRI in 34 patients having PSA (mean age, 5 years). Chiasm and optic nerves were not evaluated. Post-hydrocephalus SA or incomplete data were excluded. The clinical data were correlated to the MRI patterns. The study disclosed 82.5% associated lesions with MRI (28/34): 11 neuronal migration disorders, 9 holoprosencephalies (HP), 7 pituitary stalk interruptions, 1 corpus callosum partial agenesis; 17.5% (6/34) of cases were apparently isolated PAS. Clinically, the patients had motor dysfunction in 68% (23/34), mental retardation in 65% (22/34), blindness in 24% (8/34), endocrinological defects in 21% (7/34) and epilepsy in 18% (6/34) of cases. Nine percent of patients (3/34) were neurologically normal (including one with scoliosis and two infants younger than 2 years at the last follow-up). Patients with bilateral cortical anomalies and HP (even if mild) had the worst neurological prognosis. A severe motor impairment was present without evidence of hemispheric anomaly in 12% of patients (4/34). Interestingly, the frontal lobes were involved in 90% of cortical anomalies and HP, supporting the malformative aetiology of PSA. PSA rarely appears isolated and severe psychomotor impairment may occur in apparently isolated forms. These unfavourable results should be highlighted and need to be confirmed

  17. A rare case of sirenomelia with total bilateral cleft lip palate and kyphoscoliosis

    Directory of Open Access Journals (Sweden)

    Sangeeta Arya

    2013-04-01

    Full Text Available Sirenomelia, a rare congenital deformity, has fused lower limbs with variable combination of visceral anomalies. This condition is fatal within a day or two of birth so early antenatal detection and pregnancy termination is advocated. Antenatally it can be diagnosed by ultrasonography showing oligohydramnios, renal agenesis and fibulae positioned between the tibiae. A 24 years old Muslim primigravida came for first antenatal check up at 18 weeks of pregnancy. She didn’t turn up and came directly in emergency at 37 weeks in active labor and delivered a baby weighing 1.8 kg with a small sized placenta, complete bilateral cleft lip palate, Kyphoscoliosis and fused lower limbs upto ankles. Baby expired after 25 minutes of birth. Till now around 300 cases of Sirenomelia have been reported worldwide. All human cases of Sirenomelia have been found to be associated with variable degree of renal and urethral dysplasia or renal agenesis. External genitalia are affected, whereas gonads remain unaffected. In rare cases Sirenomelia is associated with upper body defects including cleft palate, cervical and upper thoracic vertebral abnormalities, pulmonary hypoplasia and cardiac defects. [Int J Reprod Contracept Obstet Gynecol 2013; 2(2.000: 242-244

  18. Periampullary carcinoma in a patient with agenesis of dorsal pancreas

    OpenAIRE

    Kapoor, A; Singh, RK

    2011-01-01

    Agenesis of dorsal pancreas is a rare developmental anomaly. We here report a case of agenesis of dorsal pancreas in a patient of periampullary carcinoma and highlight its implications on the management.

  19. Fetal lung growth in laryngeal atresia and tracheal agenesis.

    Science.gov (United States)

    Scurry, J P; Adamson, T M; Cussen, L J

    1989-02-01

    Three cases of airway obstruction in fetuses born at 21, 32 and 40 weeks gestation are reported. The first had laryngeal atresia, cystic dysplastic kidneys, oligohydramnios and immense fluid-filled lungs. The second had upper tracheal agenesis, a tracheo-oesophageal fistula, a cystic dysplastic horseshoe kidney, oligohydramnios and normal-sized lungs. The third had a pin-hole mucosal tract through an otherwise atretic larynx, normal kidneys, no oligohydramnios and normal-sized lungs. Lung weight:body weight ratios, radial alveolar or radial canalicular counts and point-counting of sections of lungs in cases 1 and 2 show that laryngeal or tracheal obstruction may prevent or reduce the pulmonary hypoplasia associated with renal dysplasia, and in cases 2 and 3, that grossly enlarged, hyperplastic lungs may not be seen unless obstruction is complete. PMID:2730470

  20. Bifurcation of the femur with tibial agenesis and additional anomalies

    NARCIS (Netherlands)

    van der Smagt, JJ; Bos, CFA; van Haeringen, A; Hogendoorn, PCW; Breuning, MH

    2005-01-01

    Bifurcation of the femur and tibial agenesis are rare anomalies and have been described in both the Gollop-Wolfgang Complex and the tibial agenesis-ectrodactyly syndrome. We report on two patients with bifurcation of the femur and tibial agenesis. Hand ectrodactyly was seen in one of these patients.

  1. MDCT Findings of Tracheal Agenesis: A Case Report

    Energy Technology Data Exchange (ETDEWEB)

    Jeong, Yu Mi; Kim, Jee Eun; Son, Dong Woo; Kim, Ha Na; Hwang, Hee Young [Gachon University, Gil Hospital, Incheon (Korea, Republic of)

    2009-01-15

    Tracheal agenesis is an extremely rare congenital anomaly with fatal consequences. We report a case of tracheal agenesis in a newborn infant who presented with no self-respiration and cyanosis that was confirmed by the use of 64-slice multidetector CT. Coronal and sagittal multiplanar reconstruction images clearly delineated the aberrant anatomy of tracheal agenesis and demonstrated the presence of a tracheoesophageal fistula.

  2. Bloqueio pleural bilateral: analgesia e funções pulmonares em pós-operatório de laparotomias medianas Bloqueo pleural bilateral: analgesia y funciones pulmonares en pós-operatorio de laparotomias medianas Bilateral pleural block: analgesia and pulmonary functions in postoperative of median laparotomies

    Directory of Open Access Journals (Sweden)

    Karl Otto Geier

    2004-08-01

    TODO: Bloqueo pleural bilateral fue realizado en decúbito dorsal horizontal en 21 pacientes con 20 ml de bupivacaína a 0,375% con adrenalina a 1:400.000 administrados por catéter en cada hemitórax durante el pós-operatorio inmediato. Soluciones aleatorias de bupivacaína y de solución fisiológica fueron administradas por estudiantes en la práctica o enfermeras que desconocían el contenido de las jeringas, y sus resultados analgésicos evaluados de acuerdo con la escala de dolor Prince Henry al comparar los valores pre y pós-bloqueo pleural bilateral. En función del dolor pós-operatorio, ensayos espirométricos de las funciones pulmonares también fueron determinados mediante un espirómetro portátil. RESULTADOS: Analgesia pós-operatoria, con duración media de 247,75 ± 75 minutos fue constatada en todos los pacientes con la bupivacaína, aunque haya persistido dolor residual de menor intensidad en la región suprapúbica en cinco pacientes (8% y en dos pacientes en la apófisis xifóide (3,2%. Ningún efecto analgésico fue obtenido con solución fisiológica. De frente al dolor pós-operatorio, las funciones pulmonares, evaluadas antes y después de los bloqueos, registraron mejoría con la bupivacaína en CVF (p BACKGROUND AND OBJECTIVES: Notwithstanding pleural block having become almost an analgesic panacea, contradictory results have been published. This study aimed at observing analgesic and spirometric behavior of pulmonary function in the immediate postoperative period of 21 patients submitted to urgency median laparotomies under bilateral pleural block. METHODS: Bilateral pleural block was induced in the supine position with 20 mL of 0.375% bupivacaine with 1:400,000 epinephrine administered via catheter in each hemithorax during the immediate postoperative period. Randomized bupivacaine and saline were administered by residents or nurses blind to syringes content, and their analgesic outcomes were evaluated according to Prince Henry's pain score by

  3. Unilateral absence of a pulmonary artery: Report of 3 cases

    Energy Technology Data Exchange (ETDEWEB)

    Bae, Oh Keun; Choi, Chul Seung; Choi, Yo Won; Jeon, Seok Cheol; Seo, Heung Suk; Hahm, Chang Kok [College of Medicine, Hanyang University, Seoul (Korea, Republic of)

    1994-07-15

    Unilateral absence of a pulmonary artery is an uncommon anomaly, which presents as an isolated lesion or in combination with other congenital heart disease such as TOF or PDA. We encountered three cases of isolated unilateral absence of a pulmonary artery; one was left pulmonary artery agenesis with right sided aortic arch and the others were right pulmonary artery agenesis with left sided aortic arch. Plain chest radiograph showed considerable loss of unilateral lung volume and lack of ipsilateral hilar shadow. Pulmonary angiogram which was done in two cases, revealed proximal interruption of a pulmonary artery. Chest CT was done in only one case, on which right pulmonary artery was absent and was replaced by adipose tissue. CT with its clean demonstration pulmonary artery without any evidence of acquired obstruction of a pulmonary artery by pulmonary embolism or tumor invasion, maybe a valuable method for evaluation of the unilateral absence of a pulmonary artery.

  4. Unilateral absence of a pulmonary artery: Report of 3 cases

    International Nuclear Information System (INIS)

    Unilateral absence of a pulmonary artery is an uncommon anomaly, which presents as an isolated lesion or in combination with other congenital heart disease such as TOF or PDA. We encountered three cases of isolated unilateral absence of a pulmonary artery; one was left pulmonary artery agenesis with right sided aortic arch and the others were right pulmonary artery agenesis with left sided aortic arch. Plain chest radiograph showed considerable loss of unilateral lung volume and lack of ipsilateral hilar shadow. Pulmonary angiogram which was done in two cases, revealed proximal interruption of a pulmonary artery. Chest CT was done in only one case, on which right pulmonary artery was absent and was replaced by adipose tissue. CT with its clean demonstration pulmonary artery without any evidence of acquired obstruction of a pulmonary artery by pulmonary embolism or tumor invasion, maybe a valuable method for evaluation of the unilateral absence of a pulmonary artery

  5. Tracheal agenesis with broncho-esophageal fistula in VACTERL / TACRD association

    Directory of Open Access Journals (Sweden)

    Suresh R. S. Mandrekar

    2013-01-01

    Full Text Available Tracheal agenesis (TA is an extremely rare malformation. We report here autopsy findings in a case of TA with bronchoesophageal fistula of Floyd type III. The other malformations present included laryngeal atresia, Right lung hypolobulation, ventricular septal defect in membranous portion, bilateral cystic renal dysplasia, spleninculus, Meckel′s diverticulum, and imperforate anus. The constellations of malformations present in our case have overlapping features with Vertebral anomalies, Anal atresia, Cardiovascular anomalies, Tracheo-esophageal fistula, Esophageal atresia, Renal anomalies, Limb anomalies and Tracheal atresia or laryngo tracheal atresia, Cardiac anomalies, Renal anomalies, Duodenal atresia association described previously in the literature.

  6. Agenesis of the Gallbladder in Monozygotic Twin Sisters.

    Science.gov (United States)

    Hoshi, Koki; Irisawa, Atsushi; Shibukawa, Goro; Yamabe, Akane; Fujisawa, Mariko; Igarashi, Ryo; Sato, Ai; Maki, Takumi

    2016-01-01

    Agenesis of the gallbladder, a rare anomaly, is generally regarded as an organogenic failure. Several reports suggest that this congenital defect is inherited but that supposition remains controversial. We described agenesis of the gallbladder in identical twins. A 21-year-old female presented with a history of acute pain in the epigastrium and right hypochondrium. Various imaging modalities showed "gallbladder agenesis." Moreover, her older identical twin sister had also no visualized gallbladder in imaging modalities. This case report strongly suggested that agenesis of the gallbladder would be caused by a genetic abnormality. PMID:26925274

  7. Agenesis of right upper lobe of lung

    OpenAIRE

    Kuo, Chiu-Ping; Lu, Yen-Ta; Lin, Rong-Luh

    2015-01-01

    Agenesis of the right upper lobe of the lung is a very uncommon congenital anomaly and may be referred to chest clinics in adulthood for an incidental finding of abnormal chest radiograph. The presentations of chest radiograph may imitate many common situations such as right upper lobe collapse presenting as an ipsilateral shifting of the mediastinum or elevation of the right hemidiaphragm due to eventration or subdiaphragmatic lesions. A chest computed tomography is considered the most concl...

  8. Agenesis of right upper lobe of lung.

    Science.gov (United States)

    Kuo, Chiu-Ping; Lu, Yen-Ta; Lin, Rong-Luh

    2015-06-01

    Agenesis of the right upper lobe of the lung is a very uncommon congenital anomaly and may be referred to chest clinics in adulthood for an incidental finding of abnormal chest radiograph. The presentations of chest radiograph may imitate many common situations such as right upper lobe collapse presenting as an ipsilateral shifting of the mediastinum or elevation of the right hemidiaphragm due to eventration or subdiaphragmatic lesions. A chest computed tomography is considered the most conclusive examination used to diagnose lung agenesis. Three-dimensional reconstructed images can be particularly helpful in delineating abnormalities of the bronchi and associated arterial and venous structures. We describe here a young woman with allergic rhinitis and bronchial asthma since her early childhood. She was referred to our clinic for an incidental finding of abnormal chest radiograph after a school health checkup. Right upper lobe atelectasis or intra-abdominal lesions were initially suspected. After a thorough image study, she was diagnosed as a case of agenesis of the right upper lobe. Our report emphasizes the importance that a high index of suspicion and adequate image investigation are necessary to diagnose congenital lung anomalies. PMID:26090110

  9. Tracheal agenesis: A report of two cases

    Directory of Open Access Journals (Sweden)

    A V Desai

    2016-01-01

    Full Text Available Tracheal agenesis is an extremely rare congenital anomaly involving the respiratory system. It is generally associated with anomalies of other systems. Antenatal diagnosis of this condition is difficult; therefore, it presents as a medical emergency in the labor room. Intubation in these babies is difficult. As many of these babies are born prematurely, respiratory distress syndrome (RDS adds to the management difficulties. Here, we describe two babies with this lethal anomaly and RDS where esophageal intubation and surfactant therapy proved beneficial. Furthermore, described are other associated anomalies.

  10. Pulmonary Hyalinising Granuloma: A rare pulmonary disorder

    International Nuclear Information System (INIS)

    Pulmonary hyalinising granulomas are rare, non-infectious fibros lesions of the lung which can mimic metastatic disease. It was first described in literature by Engleman et al in the year 1977. Its etiology is unknown but they may be caused by an exaggerated immune response. The patient typically presents with cough, chest pain, dyspnoea or haemoptysis in association with multiple bilateral parenchymal nodules. We report the case of a 20 years old male who presented with a 12-month history of worsening dry cough. His plain chest radiograph and subsequent CT scan revealed bilateral pulmonary nodules. A CT guided biopsy of the pulmonary lesions was consistent with Pulmonary Hyalinising Granuloma [PHG].

  11. Bilateral sciatic nerve block after orthopedic surgery in a pediatric patient

    OpenAIRE

    Levent Şahin; Mehrican Şahin; Rauf Gül

    2011-01-01

    Early postoperative pain is one of the most important problems in pediatric orthopedic surgery. Introduction of the use of ultrasound (US) has led to very important developments in pediatric regional anesthesia. We aimed to present with the literature data about that we applied the bilateral US-guided sciatic nerve block to the patient who was operated under bilateral knee disarticulation because of congenital tibia agenesis and talipes equinovarus. In conclusion we entertain that US-guided p...

  12. Magnetic resonance imaging in agenesis of corpus callosum

    International Nuclear Information System (INIS)

    The recent use of computed tomography (CT) scan is providing an easy diagnoses of agenesis of corpus callosum which had been difficult to diagnose only by clinical signs and symptoms. Since, more neuropsychological studies on agenesis of corpus callosum are being done, clinical details of agenesis of corpus callosum are being clarified. We examined magnetic resonance imaging (MRI) on 3 patients who were suspected to have agenesis of corpus callosum by CT scan. And we studied the usefulness of MRI in agenesis of corpus callosum. By sagittal MRI, we could easily make a diagnosis on one case (Case 3) of hypoplasia of corpus callosum which could not been diagnosed by CT scan. On all of the cases, MRI showed the high intensity area of anterior commissure which had not been found by CT scan. Anomaly of cingurate gyrus was shown on all of the patients. MRI can show some important points in detail which CT scan can not reveal. The sagittal view especially is the most helpful for diagnosis of agenesis of corpus callosum. And we proposed that neuropsychological study on agenesis of corpus callosum should have MRI data. (J.P.N.)

  13. Marfan's syndrome presenting as bilateral spontaneous pneumothorax.

    OpenAIRE

    Gawkrodger, D J

    1981-01-01

    A case of bilateral spontaneous pneumothorax in a 14-year-old girl with previously undiagnosed Marfan's syndrome is described. The pulmonary abnormalities of Marfan's syndrome are not commonly encountered and bilateral pneumothorax is itself a rare event which, in most instances, has been reported following invasive procedures.

  14. Familial aggregation of maxillary lateral incisor agenesis (MLIA)

    OpenAIRE

    Pandey, Pallavi; Ansari, Afroz Alam; Choudhary, Kartik; Saxena, Aditya

    2013-01-01

    Agenesis of maxillary lateral incisors and mandibular second premolars is the most frequent form of hypodontia. Its prevalence varies across population from 0.8% to 4.5%. Genetic aberrations and environmental factors may cause agenesis of one or more teeth. The management of child having such a problem is very important since diastema in teeth especially in upper anteriors not only affects child's physical appearance but also its psychological development as the child wants to look like other...

  15. Permanent Maxillary Canine Agenesis: A Rare Case Report

    OpenAIRE

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

    2015-01-01

    ABSTRACT 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 Indi...

  16. Facial emotion recognition in agenesis of the corpus callosum

    OpenAIRE

    Bridgman, Matthew W; Brown, Warren S.; Spezio, Michael L.; Leonard, Matthew K.; Adolphs, Ralph; Paul, Lynn K

    2014-01-01

    Background: Impaired social functioning is a common symptom of individuals with developmental disruptions in callosal connectivity. Among these developmental conditions, agenesis of the corpus callosum provides the most extreme and clearly identifiable example of callosal disconnection. To date, deficits in nonliteral language comprehension, humor, theory of mind, and social reasoning have been documented in agenesis of the corpus callosum. Here, we examined a basic social ability as yet not ...

  17. Bilateral dacryoadenitis

    Directory of Open Access Journals (Sweden)

    Charlotte Derr

    2012-01-01

    Full Text Available Acute dacryoadenitis is an uncommon condition that involves inflammation of the lacrimal gland. In rare instances, dacryoadenitis may be bilateral. A delay in proper treatment of an otherwise simple case of dacryoadenitis may lead to significant soft tissue morbidity such as cellulitis, lacrimal gland abscess, or orbital abscess. We report the case of a 24-year-old male who presented to the emergency department with acute bilateral dacryoadenitis. The patient′s symptoms did not respond to oral antibiotics and he subsequently required admission for intravenous antibiotics. During his hospitalization the patient had diagnostic testing to try to determine the etiology for his symptoms. The unique aspects of managing a case of bilateral dacryoadenitis as well as treatment recommendations are discussed in this case report.

  18. Bilateral pulmonary thromboembolism and Budd-Chiari syndrome in a patient with Crohn's disease on oral contraceptives Tromboembolismo de pulmón bilateral y síndrome de Budd-Chiari en paciente con enfermedad de Crohn y toma de anticonceptivos orales

    Directory of Open Access Journals (Sweden)

    M. Valdés Mas

    2009-09-01

    Full Text Available Budd-Chiari syndrome can be defined as an interruption or diminution of the normal blood flow out of the liver. Patients with Budd-Chiari syndrome present with varying degrees of symptomatology that can be divided into the following categories: fulminant, acute, subacute and chronic. The subacute form is the most common presentation. A majority of patients with Budd-Chiari syndrome have an underlying hypercoagulability state. We present the case of a young woman with Crohn's disease on oral contraceptives who developed bilateral pulmonary thromboembolism and Budd-Chiari syndrome.El síndrome de Budd-Chiari consiste en la interrupción o disminución de flujo de las venas suprahepáticas. Tiene una gran variabilidad clínica en cuanto a su forma de presentación siendo la más frecuente la forma subaguda. La gran mayoría de los pacientes responden a estados de hipercoagulabilidad. Presentamos el caso de una paciente joven con enfermedad de Crohn que estaba en tratamiento con anticonceptivos orales y desarrolló un cuadro clínico de tromboembolismo de pulmón bilateral y síndrome de Budd-Chiari.

  19. CONGENITAL VESICOUTERINE FISTULA ALONG WITH DISTAL VAGINAL AGENESIS, SOLITARY KIDNEY AND TONGUE TIE: A RARITY

    OpenAIRE

    Khalid; Amit; Rana Pratap; Ahsan; Takallum

    2015-01-01

    : Congenital vesicouterine fistula in association with vaginal agenesis and solitary renal agenesis has been rarely reported. We present a case of 19 year old female suffering from cyclical menouria for last years. Physical examination revealed absence of vagina. IVP revealed left renal agenesis and CT scan revealed left renal agenesis with vesicouterine fistula. Cystoscopy showed vesicouterine fistula located above trigone near midline. Vesicouterine fistula was repaired along wi...

  20. Bilateral hemimelia of radio in canine – case report
    Hemimelia bilateral de rádio em canino – relato de caso

    OpenAIRE

    Thalita Priscila da Silva Peres; Wilma Neres da Silva Campos; Thaís Ruiz; Yara Silva Meireles; Pedro Brandini Néspoli; Roberto Lopes de Souza

    2013-01-01

    Hemimelia or agenesis is a congenital morphological change that may affect the appendicular skeleton. This rare anomaly is the total or partial absence of one or more bones matched. It may present unilaterally or bilaterally, with the most frequent unilateral. In dogs, the most common form of the radial hemimelia is that causes deformity of the affected limb and severe functional limitations. Was Veterinary Hospital of the Federal University of Mato Grosso (HOVET/UFMT), a canine, female mongr...

  1. Familial aggregation of maxillary lateral incisor agenesis (MLIA).

    Science.gov (United States)

    Pandey, Pallavi; Ansari, Afroz Alam; Choudhary, Kartik; Saxena, Aditya

    2013-01-01

    Agenesis of maxillary lateral incisors and mandibular second premolars is the most frequent form of hypodontia. Its prevalence varies across population from 0.8% to 4.5%. Genetic aberrations and environmental factors may cause agenesis of one or more teeth. The management of child having such a problem is very important since diastema in teeth especially in upper anteriors not only affects child's physical appearance but also its psychological development as the child wants to look like other children. In this article is presented a case of non-syndromic agenesis of maxillary lateral incisors (MLIA) and mandibular central incisors in a 10-year-old boy (patient) in permanent dentition with its management along with the radiographic investigations and photographic presentations of the other members of his family affected with this condition. PMID:23536620

  2. Msx1 Mutations: How Do They Cause Tooth Agenesis?

    OpenAIRE

    Wang, Y.; Kong, H; Mues, G.; D’Souza, R.

    2011-01-01

    Mutations in the transcription factors PAX9 and MSX1 cause selective tooth agenesis in humans. In tooth bud mesenchyme of mice, both proteins are required for the expression of Bmp4, which is the key signaling factor for progression to the next step of tooth development. We have previously shown that Pax9 can transactivate a 2.4-kb Bmp4 promoter construct, and that most tooth-agenesis-causing PAX9 mutations impair DNA binding and Bmp4 promoter activation. We also found that Msx1 by itself rep...

  3. Role of OHVIRA syndrome in renal agenesis: a case report

    Directory of Open Access Journals (Sweden)

    Hulya Ozturk

    2014-03-01

    Full Text Available A 13-year-old female was admitted with abdominal pain and dysmenorrhea. Ultrasonography and magnetic resonance imaging revealed OHVIRA syndrome (uterus didelphys, obstructed hemivagina, and ipsilateral renal agenesis. A left hysterectomy was performed. The patient recovered fully following treatment with vaginal septum resection and drainage of a hematometrocolpos.

  4. Uterus didelphys with obstructed hemivagina and renal agenesis : mri findings

    OpenAIRE

    De Mey, J; Yazdi, A Talebian; De Smet, K; Ernst, C.; Desprechins, B

    2011-01-01

    Müllerian duct abnormalities (MDA) are developmental disorders leading to dysmorphism of the female genital tract. Currently the Buttram and Gibbons classification of these entities is widely used. We present a case of a young girl with uterus didelphys and ipsilateral renal agenesis.

  5. Role of OHVIRA syndrome in renal agenesis: a case report

    OpenAIRE

    Hulya Ozturk; Emine Dagistan; Tulay Ozlu

    2014-01-01

    A 13-year-old female was admitted with abdominal pain and dysmenorrhea. Ultrasonography and magnetic resonance imaging revealed OHVIRA syndrome (uterus didelphys, obstructed hemivagina, and ipsilateral renal agenesis). A left hysterectomy was performed. The patient recovered fully following treatment with vaginal septum resection and drainage of a hematometrocolpos.

  6. Renal tubular dysgenesis with hypocalvaria and ileocecal valve agenesis: an autopsy report

    Directory of Open Access Journals (Sweden)

    Ariel Barreto Nogueira

    2012-12-01

    Full Text Available Renal tubular dysgenesis (RTD is a rare, lethal, autosomal recessive disorder characterized by non-differentiation of the renal proximal convoluted tubules, resulting in oligohydramnios. It is usually diagnosed in the second trimester of pregnancy, following the oligohydramnios sequence, pulmonary hypoplasia and hypocalvaria. The prognosis is poor, and death usually occurs in utero or within the first few days of life. The pathogenesis of RTD is associated with the perinatal use of drugs, such as angiotensin- converting enzyme inhibitors, angiotensin II receptor antagonists, and anti- inflammatory drugs, as well as with fetal transfusion syndrome, genetic mutations in the pathway of the renin-angiotensin system pathway, cocaine snorting, or other pathological mechanisms that reduce renal blood flow. Here, we report the autopsy of a neonate born to consanguineous parents at 38 weeks of gestation, with RTD, decreased amniotic fluid, oligohydramnios sequence, hypocalvaria, pulmonary hypoplasia, and ileocecal valve agenesis. To our knowledge, the latter has never been reported associated with RTD.

  7. Bilateral agreements

    International Nuclear Information System (INIS)

    Ten bilateral agreements are presented. These are: 1) Co-operation agreement relating to the peaceful uses of nuclear energy between Argentina and EURATOM (1996); 2) Agreement on co-operation in the peaceful uses of nuclear energy between Argentina and Greece (1997); 3) Implementing arrangement for technical exchange and co-operation in the area of peaceful uses of nuclear energy between Argentina and the United States (1997); 4) Agreement concerning co-operation in nuclear science and technology between Australia and Indonesia (1997); 5) Implementation of the 1985 Agreement for co-operation concerning the peaceful uses of nuclear energy between the People's Republic of China and the United States (1998); 6) Protocol of co-operation between France and Lithuania (1997); 7) Agreement on co-operation in energy research, science and technology, and development between Germany and the United States (1998); 8) Agreement on early notification of a nuclear accident and exchange of information on nuclear facilities between Greece and Romania (1997); 9) Agreement on early notification of nuclear accidents and co-operation in the field of nuclear safety between Hungary and the Ukraine (1997); 10) Agreement in the field of radioactive waste management between Switzerland and the United States (1997). (K.A.)

  8. Unexplained childhood anaemia: idiopathic pulmonary hemosiderosis.

    Science.gov (United States)

    Siu, K K; Li, Rever; Lam, S Y

    2015-04-01

    This report demonstrates pulmonary haemorrhage as a differential cause of anaemia. Idiopathic pulmonary hemosiderosis is a rare disease in children; it is classically described as a triad of haemoptysis, pulmonary infiltrates on chest radiograph, and iron-deficiency anaemia. However, anaemia may be the only presenting feature of idiopathic pulmonary hemosiderosis in children due to occult pulmonary haemorrhage. In addition, the serum ferritin is falsely high in idiopathic pulmonary hemosiderosis which increases the diagnostic difficulty. We recommend that pulmonary haemorrhage be suspected in any child presenting with iron-deficiency anaemia and persistent bilateral pulmonary infiltrates. PMID:25904566

  9. Waardenburg syndrome with familial unilateral renal agenesis: a new syndrome variant?

    Science.gov (United States)

    Webb, Katie M; Smith, Alisha J; Dansby, Linda M; Diskin, Charles J

    2015-06-01

    A 64-year-old man with Waardenburg syndrome presented with anuria and was subsequently discovered by renal ultrasound to have unilateral renal agenesis. The patient is one of three generations with incidental finding of renal agenesis also marked by the presence of Waardenburg syndrome. To our knowledge, there has been no mention elsewhere in the scientific literature of a variant of Waardenburg syndrome with associated renal agenesis. PMID:25402868

  10. Multivariate Analysis of Factors Affecting Presence and/or Agenesis of Third Molar Tooth

    OpenAIRE

    Alam, Mohammad Khursheed; Hamza, Muhammad Asyraf; Khafiz, Muhammad Aizuddin; Rahman, Shaifulizan Abdul; Shaari, Ramizu; Hassan, Akram

    2014-01-01

    To investigate the presence and/or agenesis of third molar (M3) tooth germs in orthodontics patients in Malaysian Malay and Chinese population and evaluate the relationship between presence and/or agenesis of M3 with different skeletal malocclusion patterns and sagittal maxillomandibular jaw dimensions. Pretreatment records of 300 orthodontic patients (140 males and 160 females, 219 Malaysian Malay and 81 Chinese, average age was 16.27±4.59) were used. Third-molar agenesis was calculated with...

  11. BICORNUATE UTERUS WITH CERVICAL ATRESIA AND VAGINAL AGENESIS ASSOCIATED WITH OVARIAN ENDOMETRIOSIS - A CASE REPORT

    Directory of Open Access Journals (Sweden)

    Alpana

    2013-06-01

    Full Text Available ABSTRACT: Vaginal agenesis combined with a functional uterus is a rare type of Mullerian duct an omaly. Only 7 – 8% of patients with vaginal agenesis have a functional uterus. Women born with vaginal agenesis, cervical atresia combined with a functioning endometrium typically present with hematometra , disabling pelvic pain and progressively worsening en dometriosis. Almost all need an abdominal hysterectomy for relief. We report a case of severe endometriosis in a 29 years old woman having a bicornuate uterus with cervical atresia and vaginal agenesis.

  12. CONGENITAL VESICOUTERINE FISTULA ALONG WITH DISTAL VAGINAL AGENESIS, SOLITARY KIDNEY AND TONGUE TIE: A RARITY

    Directory of Open Access Journals (Sweden)

    Khalid

    2015-11-01

    Full Text Available : Congenital vesicouterine fistula in association with vaginal agenesis and solitary renal agenesis has been rarely reported. We present a case of 19 year old female suffering from cyclical menouria for last years. Physical examination revealed absence of vagina. IVP revealed left renal agenesis and CT scan revealed left renal agenesis with vesicouterine fistula. Cystoscopy showed vesicouterine fistula located above trigone near midline. Vesicouterine fistula was repaired along with uterine preservation. Sigmoid colon neovagina was created. Patient is doing well and menstruating per vagina till last followup

  13. Corpus callosum agenesis: Role of fetal magnetic resonance imaging

    Directory of Open Access Journals (Sweden)

    Achour Radhouane

    2016-05-01

    Full Text Available Corpus callosum agenesis (CCA was evaluated by ultrasound examination and magnetic resonance imaging (MRI with many studies. Ultrasonography was able to suspect CCA by indirect signs but a definitive diagnosis of CCA was achieved in rare cases. MRI was able to diagnose complete CCA in majority of cases. Additional neurological abnormalities including heterotopia, gyration anomaly, asymmetry of the cerebral hemispheres, and Dandy-Walker variant were documented, as well as an ocular anomaly which was described, by MRI examination. Prenatal counseling for fetal agenesis of the corpus callosum is difficult as the prognosis is uncertain. The association with other cerebral abnormalities increases the likelihood of a poor outcome and ultrasonographic assessment of the fetal brain is limited. We found MRI to be a safe and useful additional procedure to complement ultrasonographic diagnosis or suspicion of CCA.

  14. A rare case of Turner's syndrome presenting with Mullerian agenesis.

    Science.gov (United States)

    Vaddadi, Suresh; Murthy, Ramana S V; Rahul, C H; Kumar, Vinod L

    2013-10-01

    Turner's syndrome also called as Ullrich Turner's syndrome, is a disease of unclear pathogenesis characterized by complete or partial absence of one sex chromosome, with or without cell line mosaicism in a phenotypic female with short stature. Various anomalies result in a constellation of features, of which the most disturbing is primary amenorrhea due to gonadal dysgenesis. Hormone therapy in these patients can often result in successful menstruation, and scope for subsequent pregnancy because of anatomically normal uterus and vagina. Coexisting Mullerian agenesis in these patients can jeopardize the chances of future pregnancy as they have associated structural abnormalities of the uterus and vagina. We report a rare case of middle-aged female with Turner's syndrome and Mullerian agenesis having absent secondary sexual characters and missing uterus with incompletely formed vagina. PMID:24672170

  15. Tooth agenesis: from molecular genetics to molecular dentistry

    Czech Academy of Sciences Publication Activity Database

    Matalová, Eva; Fleischmannová, Jana; Sharpe, P. T.; Tucker, A. S.

    2008-01-01

    Roč. 87, č. 7 (2008), s. 617-623. ISSN 0022-0345 R&D Projects: GA AV ČR KJB500450802; GA MŠk OC B23.001; GA ČR GC524/08/J032 Institutional research plan: CEZ:AV0Z50450515 Keywords : tooth agenesis Subject RIV: FF - HEENT, Dentistry Impact factor: 3.142, year: 2008

  16. Thyroid isthmus agenesis associated with solitary nodule: A case report

    OpenAIRE

    Schanaider, Alberto; de Oliveira, Paschoal Josias

    2008-01-01

    Background Agenesis of the isthmus associated with nontoxic solitary nodular goiter is a rare congenital anomaly. Imaging data exceptionally has been previously reported in the English language literature. Case presentation Preoperative assessment of a 22-year-old white man patient showed an asymptomatic nodule of the thyroid at the left lobe, measuring 2.3 × 1.5 cm in diameter without regional lymphadenopathy. At surgical exploration it was seen an absence of the thyroid isthmus. Conclusion ...

  17. Renal Agenesis with Full Length Ipsilateral Refluxing Ureter

    Science.gov (United States)

    Chandra, Vipin; Banerjee, Manju

    2016-01-01

    Unilateral renal agenesis with vesicoureteral reflux in the ipsilateral full length ureter is a rare phenomenon. Herein we report a case of 10-year old boy who presented with recurrent urinary tract infections. No renal tissue was identified on left side in various imaging studies. Micturating cystourethrogram (MCUG) showed left sided refluxing and blind ending ureter. Left ureterectomy was done because of recurrent UTI in the refluxing system.

  18. Tracheal agenesis: A rare but fatal congenital anomaly

    OpenAIRE

    Ergun, Suzan; Tewfik, Ted; Daniel, Sam

    2011-01-01

    In this report we describe a newborn with a rare case of Type II tracheal agenesis and bronchoesophageal fistula. Polyhydramnios and suspected esophageal atresia were identified during routine pre-natal ultrasound screening. Upon delivery, rigid bronchoscopy, esophagoscopy, and intraoperative fluoroscopy were performed, where both bronchi and the carina showed unusual horizontal orientation making it difficult to identify the fistula. However, a post mortem CT confirmed the diagnosis of an is...

  19. Bilateral anterior shoulder dislocation

    OpenAIRE

    Meena, Sanjay; Saini, Pramod; Singh, Vivek; Kumar, Ramakant; Trikha, Vivek

    2013-01-01

    Shoulder dislocations are the most common major joint dislocations encountered in the emergency departments. Bilateral shoulder dislocations are rare and of these, bilateral posterior shoulder dislocations are more prevalent than bilateral anterior shoulder dislocations. Bilateral anterior shoulder dislocation is very rare. We present a case of 24-year-old male who sustained bilateral anterior shoulder dislocation following minor trauma, with associated greater tuberosity fracture on one side...

  20. Primary pulmonary alveolar proteinosis

    OpenAIRE

    Šarac Sanja; Milić Rade; Zolotarevski Lidija; Aćimović Slobodan; Tomić Ilija; Plavec Goran

    2012-01-01

    Introduction. Pulmonary alveolar proteinosis is an uncommon disease characterized by the accumulation of surfactant proteins and phospholipids within the alveolar spaces. Acquired disease can be idiopathic (primary) and secondary. The prevalence of acquired pulmonary alveolar proteinosis is about 0.37 per 100,000 persons. Common symptoms are dyspnea and cough. Chest X-ray shows bilateral perihilar infiltrates. Open-lung biopsy is the gold standard for the diagnosis. Treatment includes w...

  1. Agenesis of Permanent Mandibular Central Incisors: A Concordant Condition in Siblings

    Science.gov (United States)

    Namineni, Srinivas; Tupalli, Abhinaya Reddy; Challa, Santhosh Kumar

    2016-01-01

    ABSTRACT Concordance is an identical expression of phenotype in two related individuals. Concordance expression of hypodontia is an uncommon condition where associated individuals are affected with exactly similar kind and number of missing teeth. There is very limited documentation of this condition either in twins or in siblings, and literature shows paucity of data with regard to this anomaly. To the best of our knowledge, there is only one such case reported in the literature, which has actually showed similar missing lower central incisors in siblings. This report presents a case of two girl siblings aged 11 and 13 years with congenital bilateral missing of permanent mandibular central incisors, which is an absolute concordant condition. Apart from discussing etiology, clinical implications and management, this article highlights the significance of concordant and discordant condition of hypodontia and expression of this condition in twins and siblings. How to cite this article: Kagitha PK, Namineni S, Tupalli AR, Challa SK. Agenesis of Permanent Mandibular Central Incisors: A Concordant Condition in Siblings. Int J Clin Pediatr Dent 2016;9(1):74-77. PMID:27274160

  2. Uterus didelphys with unilateral distal vaginal agenesis and ipsilateral renal agenesis: Common presentation of an unusual variation

    OpenAIRE

    Adair, Luther; Georgiades, Maria; Osborne, Rhonda; Ng, Tsz

    2011-01-01

    Mullerian duct abnormalities are congenital malformations that are easily missed and can lead to incorrect diagnosis and unnecessary operative procedures. In this case, a young female presented with cyclic pelvic pain that continued after previous surgical resection of an ovarian cyst. Further investigation with clinical examinations and multimodality imaging demonstrated ipsilateral renal agenesis and a Class III Mullerian duct anomaly (MDA) requiring a second operative procedure. It is beli...

  3. Morphologic and Demographic Predictors of Third Molar Agenesis: A Systematic Review and Meta-analysis.

    Science.gov (United States)

    Carter, K; Worthington, S

    2015-07-01

    We aimed to consolidate all available data on worldwide third molar agenesis frequencies, with a particular emphasis on exploring the factors leading authors to find contradictory results for the demographic and morphologic predictors of this anomaly. A total of 12,376 studies were originally identified, then narrowed down to 1,312 for title/abstract screening. On the basis of our inclusion and exclusion criteria, we selected 92 studies, containing 100 effect sizes and 63,314 subjects, for systematic review and metaregression. The worldwide rate of agenesis was found to be 22.63% (95% confidence interval = 20.64% to 24.76%), although the estimates ranged from 5.32% to 56.0%. Our subgroup analyses revealed that women are 14% more likely than men to have agenesis of ≥1 third molars and that maxillary agenesis was 36% more likely than mandibular agenesis in both sexes. Furthermore, we found that having agenesis of 1 or 2 molars was most common, while agenesis of 3 or 4 molars was least common. Finally, we found large differences among agenesis frequency depending on geographic region. This information is expected to be of use not only to clinicians and patients but also to policy makers, given the implications for third molar extraction protocols. PMID:25883107

  4. Imaging Findings in Agenesis of the Dorsal Pancreas. Report of Three Cases

    Directory of Open Access Journals (Sweden)

    Manoranjan Mohapatra

    2012-01-01

    Full Text Available Context Agenesis of the dorsal pancreas is rare. The dorsal pancreatic agenesis is described in two forms, the partial and the complete form. Patients with this anomaly may be asymptomatic or may present with diabetes mellitus, epigastric pain, acute or chronic pancreatitis. Case report We report the computed tomography (CT, magnetic resonance imaging (MRI and magnetic resonance cholangiopancreatography (MRCP findings in three cases with dorsal pancreatic agenesis, one with partial and the other two with complete form. Speckled calcification at pancreatic head was observed in one patient. Lateral contour lobulation of pancreatic head which is seen in one third of normal population is believed to be due to variation in fusion between ventral and dorsal pancreas. In contrast, we observed lateral contour lobulation of pancreatic head in a case of complete agenesis of the dorsal pancreas where structures derived from dorsal pancreas are undeveloped. The ventral and dorsal pancreatic duct lengths were measured on MRCP images and we observed that in partial agenesis, the duct of Wirsung was shorter in length, compared to the duct of Santorini. The duct of Wirsung was relatively longer in cases of complete agenesis of the dorsal pancreas. Conclusion The CT, MRI and MRCP findings in dorsal pancreatic agenesis and the relationship between the length of ventral duct with the type ofdorsal pancreatic agenesis will provide a new insight into this particular anomaly.

  5. Pulmonary Aplasia in an Adult : A Case Report

    Directory of Open Access Journals (Sweden)

    Nurettin Yiyit

    2016-01-01

    Full Text Available Pulmonary aplasia is a rare congenital anomaly in which there are absence of pulmonary vessels, bronchus and parenchyma. It is distinguished from pulmonary agenesis by the presence of rudimentary stump bronchus. Patients are usually diagnosed in childhood. Patients without additional anomaly and the patients with mild disease can be diagnosed in adulthood. The left lung was not observed at the chest X-ray of 19-year-old male patient with respiratory distress in exercise. Left lung aplasia was diagnosed by computed tomography and ventilation perfusion scintigraphy. The patients with lung aplasia have an increased risk of infections. Therefore the follow-up of the patients is important.

  6. Bilateral Gonadal Cysts and Late Diagnosis of Androgen Insensitivity Syndrome Treated by Laparoscopic Gonadectomy

    Directory of Open Access Journals (Sweden)

    D. Tourlakis

    2011-01-01

    Full Text Available Background. Complete androgen insensitivity syndrome is a rare syndrome in which the uterus is absent and testes rather than ovaries are present. Patients usually visit a gynecologist due to primary amenorrhea. Case. A forty-eight-year-old woman with lower abdominal pain and anamnesis of uterus agenesis was operated on due to bilateral cystic masses. A 5 × 3 × 1.2 cm left adnexal cyst revealed the presence of a serous cyst with a hypoplastic ductus deferens. A smaller cyst of the right adnexa revealed immature testis tissue with Leydig-cell hyperplasia. After karyotype and hormonal examinations, laparoscopic gonadectomy was performed. Conclusion. Attention should be paid in all cyst-removing operations in cases of uterus agenesis, due to the high incidence of malignancy. Not of less importance is the issue of informing the patient in the most appropriate way.

  7. Agenesis of Ductus Venosus Presenting with Dilated Inferior Vena Cava with Favorable Outcome

    Directory of Open Access Journals (Sweden)

    Omer Dai

    2014-04-01

    Full Text Available The ductus venosus regulates the distribution of oxygen and placental nutrients by restricting the centralization of blood flow in fetal circulation. The ductus venosus is a small vein transmitting oxygen-rich blood from the umbilical vein to the fetal heart.. Increasing numbers of case reports are being published about ductus venosus agenesis with cardiac structural abnormalities, other malformations, chromosomal abnormalities, and stillbirth. Heart failure, hydrops and unexplained polyhydramnios could also be associated with ductus venosus agenesis. In the follow-up of fetuses with ductus venosus agenesis, care must be taken to examine for hydrops and heart failure. The agenesis of ductus venosus is often only found in fetuses by coincidentally. Herein we discuss the prenatal diagnosis of isolated agenesis of the ductus venosus presenting with a dilated inferior vena cava and without any disturbance of fetal hemodynamics and any other malformation. In conclusion a dilated inferior vena cava can be a sign of ductus venosus agenesia.

  8. Multivariate analysis of factors affecting presence and/or agenesis of third molar tooth.

    Directory of Open Access Journals (Sweden)

    Mohammad Khursheed Alam

    Full Text Available To investigate the presence and/or agenesis of third molar (M3 tooth germs in orthodontics patients in Malaysian Malay and Chinese population and evaluate the relationship between presence and/or agenesis of M3 with different skeletal malocclusion patterns and sagittal maxillomandibular jaw dimensions. Pretreatment records of 300 orthodontic patients (140 males and 160 females, 219 Malaysian Malay and 81 Chinese, average age was 16.27±4.59 were used. Third-molar agenesis was calculated with respect to race, genders, number of missing teeth, jaws, skeletal malocclusion patterns and sagittal maxillomandibular jaw dimensions. The Pearson chi-square test and ANOVA was performed to determine potential differences. Associations between various factors and M3 presence/agenesis groups were assessed using logistic regression analysis. The percentages of subjects with 1 or more M3 agenesis were 30%, 33% and 31% in the Malaysian Malay, Chinese and total population, respectively. Overall prevalence of M3 agenesis in male and female was equal (P>0.05. The frequency of the agenesis of M3s is greater in maxilla as well in the right side (P>0.05. The prevalence of M3 agenesis in those with a Class III and Class II malocclusion was relatively higher in Malaysian Malay and Malaysian Chinese population respectively. Using stepwise regression analyses, significant associations were found between Mx (P<0.05 and ANB (P<0.05 and M3 agenesis. This multivariate analysis suggested that Mx and ANB were significantly correlated with the M3 presence/agenesis.

  9. Agenesis of the dorsal mesentery presenting in an adolescent

    Directory of Open Access Journals (Sweden)

    Anith Chacko

    2013-03-01

    Full Text Available Agenesis of the dorsal mesentery is a rare occurrence that usually presents in children. It is associated with proximal small bowel malrotation as well as high jejunal atresia with discontinuity of the small bowel. We present a case report of an adolescent presenting with clinical features of proximal small bowel obstruction (confirmed on imaging as well as acute pancreatitis. At laparotomy, he was found to have no dorsal mesentery, without small bowel atresia, and the duodenum was fixed to the posterior abdominal wall. The patient recovered well and remained symptom-free.

  10. Rubinstein-Taybi syndrome with agenesis of corpus callosum

    Directory of Open Access Journals (Sweden)

    Shubhankar Mishra

    2015-01-01

    Full Text Available Rubinstein-Taybi syndrome (RSTS is a rare genetic disorder with characteristic morphological anomaly. Our patient was a 4.5-year-old girl came with features like broad thumbs, downward slanting palpebral fissures and mental retardation. Systemic abnormalities such as repeated infection, seizure with developmental delay were also associated with it. She was having head banging behavior abnormal slurring speech, incoordination while transferring things from one hand to other. Galaxy of clinical pictures and magnetic resonance imaging report helped to clinch the diagnosis as a case of "RSTS with corpus callosal agenesis" which to the best of our knowledge has never been reported in past from India.

  11. Rubinstein-Taybi syndrome with agenesis of corpus callosum.

    Science.gov (United States)

    Mishra, Shubhankar; Agarwalla, Sunil Kumar; Potpalle, Dnyaneshwar Ramesh; Dash, Nishant Nilotpal

    2015-01-01

    Rubinstein-Taybi syndrome (RSTS) is a rare genetic disorder with characteristic morphological anomaly. Our patient was a 4.5-year-old girl came with features like broad thumbs, downward slanting palpebral fissures and mental retardation. Systemic abnormalities such as repeated infection, seizure with developmental delay were also associated with it. She was having head banging behavior abnormal slurring speech, incoordination while transferring things from one hand to other. Galaxy of clinical pictures and magnetic resonance imaging report helped to clinch the diagnosis as a case of "RSTS with corpus callosal agenesis" which to the best of our knowledge has never been reported in past from India. PMID:26167229

  12. Bilateral acute acquired toxoplasmic retinochoroiditis after steroid therapy for hantavirus pulmonary syndrome: case report Retinite toxoplásmica bilateral adquirida após terapia com corticoesteróide para síndrome pulmonar por hantavírus: relato de caso

    OpenAIRE

    Rubens Camargo Siqueira; Rodrigo Jorge; Luiz Tadeu Moraes Figueiredo

    2007-01-01

    Description of a case of acute acquired ocular toxoplasmosis following hantavirus pulmonary syndrome. A 41-year-old man presenting hantavirus pulmonary syndrome, confirmed in the laboratory by detection of IgM antibodies to the virus, was submitted to high doses of intravenous corticosteroids for two months. After clinical improvement of hantavirus pulmonary syndrome the patient presented visual loss in both eyes that was secondary to a toxoplasmosis retinitis. The retinitis resolved with ant...

  13. Language and development in FG syndrome with callosal agenesis.

    Science.gov (United States)

    McCardle, P; Wilson, B

    1993-06-01

    The FG syndrome is characterized by unusual facies, sudden infant death, developmental delay, and abnormalities of the cardiac, gastrointestinal, and central nervous systems. No longitudinal data on development in surviving patients are currently available. Serial evaluations of a patient with FG syndrome, whose sole central nervous system anomaly was agenesis of the corpus callosum, showed a consistent pattern over time. Specific language impairments in syntactic and pragmatic-semantic areas are emerging. These findings represent the first detailed data on which expectations for children with the FG syndrome can be based. The findings also fit theoretical constructs on the function of the corpus callosum and may therefore be generalized to provide expectations for other patients with isolated agenesis of the corpus callosum. Given the information gained from this case, it is clear that language intervention/consultation should be a pivotal service for such children, and that the speech-language pathologist should play a role in the development of an integrated educational services plan. PMID:7688382

  14. MR in complete dorsal pancreatic agenesis: Case report and review of literature

    Directory of Open Access Journals (Sweden)

    Shruti Thakur

    2014-01-01

    Full Text Available The morphogenesis of the pancreas is a complex process having a very low frequency of anatomic variation. The congenital anomalies are rare. Complete pancreatic and ventral pancreatic agenesis are incompatible with life. Dorsal pancreatic agenesis is exceedingly rare with less than 100 cases reported in the world literature. Patients with this anomaly may be asymptomatic or may present with abdominal pain, hyperglycemia, diabetes mellitus, and acute or chronic pancreatitis. Such anomalies are rarely reported; therefore, clinical awareness of agenesis of the dorsal pancreas as a cause of these symptoms can expand the differential diagnosis and improve patient management.

  15. The WNT10A gene in Ectodermal Dysplasias and Selective Tooth Agenesis

    OpenAIRE

    Mues, Gabriele; Bonds, John; Xiang, Lilin; Vieira, Alexandre R; Seymen, Figen; Klein, Ophir; D’Souza, Rena N.

    2014-01-01

    Mutations in the WNT10A gene were first detected in the rare syndrome odonto-onycho-dermal dysplasia (OODD, OMIM257980) but have now also been found to cause about 35-50% of selective tooth agenesis (STHAG4, OMIM150400), a common disorder that mostly affects the permanent dentition. In our random sample of tooth agenesis patients, 40 percent had at least one mutation in the WNT10A gene. The WNT10A Phe228Ile variant alone reached an allele frequency of 0.21 in the tooth agenesis cohort, about ...

  16. Bilateral lateral periodontal cyst

    OpenAIRE

    Govil, Somya; Gupta, Vishesh; Misra, Neeta; Misra, Pradyumna

    2013-01-01

    The bilateral lateral periodontal cyst is a rare nasological entity, which despite clinical and radiological presentation is being diagnosed by histological characteristics. It is asymptomatic in nature and is observed in routine radiography. The aim and objective of this article is to present a rare case of bilateral lateral periodontal cyst in a 14-year-old child. The clinical and radiographical findings, along with its management have been discussed. Enucleation of bilateral cyst without e...

  17. Primary pulmonary alveolar proteinosis

    Directory of Open Access Journals (Sweden)

    Šarac Sanja

    2012-01-01

    Full Text Available Introduction. Pulmonary alveolar proteinosis is an uncommon disease characterized by the accumulation of surfactant proteins and phospholipids within the alveolar spaces. Acquired disease can be idiopathic (primary and secondary. The prevalence of acquired pulmonary alveolar proteinosis is about 0.37 per 100,000 persons. Common symptoms are dyspnea and cough. Chest X-ray shows bilateral perihilar infiltrates. Open-lung biopsy is the gold standard for the diagnosis. Treatment includes whole-lung lavage, application of granulocyte-macrophage colonystimulating factor and lung transplantation. Case report. We reported a 51 year-old man with primary form of the disease. It was the second case of this extremely rare disease in the past 30 years in our clinic. The symptoms were longlasting dry cough, fever and physical deterioration. Chest Xray revealed bilateral pulmonary infiltrates; computed tomography showed patchy ground-glass opacification with interlobular thickening. The diagnosis was established by open lung biopsy. Additional tests were performed to exclude secondary form of the disease. Conclusion. We presented a rare clinical entity with typical clinical features and clinical and radiological course of the disease, in order to improve differential diagnostic approach to patients with bilateral lung infiltrations. In patients with pulmonary alveolar proteinosis timely diagnosis and adequate treatment can improve a prognosis.

  18. Bilateral cystic dysplasia of the rete testis with renal adysplasia.

    Science.gov (United States)

    Bouron-Dal Soglio, Dorothée; Harvey, Isabelle; Jovanovic, Mubina; Oligny, Luc L; Fournet, Jean-Christophe

    2006-01-01

    Cystic dyplasia of the rete testis (CDRT) is an uncommon, generally unilateral lesion characterized by anastomosing cystic spaces lined by a flattened simple cuboidal epithelium in the rete testis. In the literature this lesion often is associated with an ipsilateral urogenital lesion such as renal agenesia or multicystic dysplasia of the kidney, in order of frequency. The pathogenesis is explained by some authors by their common embryologic origin. We are reporting the finding of bilateral CDRT associated with ultrasound-diagnosed renal adysplasia in a 20-week gestational age fetus with oligohydramnios. Although CDRT has been referred to as being associated with multicystic renal dysplasia or renal agenesis, the present case appears to be unique in combining all the malformations together. PMID:16822083

  19. Agenesis of dorsal pancreas with eventration of diaphragm and intrapancreatic pseudocyst: a rare entity

    Directory of Open Access Journals (Sweden)

    Poras Chaudhary

    2013-02-01

    Full Text Available A case of acute pancreatitis in a 43-year-old male patient, which was eventually diagnosed as agenesis of dorsal pancreas with eventration of left hemidiaphragm and intrapancreatic pseudocyst is being reported.

  20. Role of Endoscopic Ultrasound in the Diagnosis of Agenesis of the Dorsal Pancreas

    Directory of Open Access Journals (Sweden)

    Laura Sempere

    2006-07-01

    Full Text Available Context: Complete agenesis of the dorsal pancreas is an unusual congenital anomaly. Nowadays, the diagnosis is based on four imaging studies: transabdominal ultrasonography, computed tomography, magnetic resonance cholangiopancreatography and, the gold-standard, endoscopic retrograde cholangiopancreatography. Sometimes the ability of these studies are limited to distinguishing agenesis of the dorsal pancreas from other congenital abnormalities. Endoscopic ultrasound is a minimally invasive technique which permits us to obtain high resolution images of the pancreatic parenchyma and ductal system. Case report :We report the case of a 23-yearold woman with recurrent episodes of acute pancreatitis and non-conclusive classic imaging studies. Complete agenesis of the dorsal pancreas was demonstrated by endoscopic ultrasound. Conclusion: Endoscopic ultrasound may be useful in the diagnosis of agenesis of the dorsal pancreas.

  1. Delayed dental maturity in dentitions with agenesis of mandibular second premolars

    DEFF Research Database (Denmark)

    Daugaard, S; Christensen, I J; Kjaer, I

    2010-01-01

    To evaluate dental maturity in the mandibular canine/premolar and molar innervation fields in children with agenesis of the 2nd mandibular premolar and to associate these findings with normal control material....

  2. Agenesis of dorsal pancreas with eventration of diaphragm and intrapancreatic pseudocyst: a rare entity

    OpenAIRE

    Poras Chaudhary; Moninder P. Arora

    2013-01-01

    A case of acute pancreatitis in a 43-year-old male patient, which was eventually diagnosed as agenesis of dorsal pancreas with eventration of left hemidiaphragm and intrapancreatic pseudocyst is being reported.

  3. Bilateral Gonadal Cysts and Late Diagnosis of Androgen Insensitivity Syndrome Treated by Laparoscopic Gonadectomy

    OpenAIRE

    Tourlakis, D.; Fersis, N; Schnabel, J.

    2011-01-01

    Background. Complete androgen insensitivity syndrome is a rare syndrome in which the uterus is absent and testes rather than ovaries are present. Patients usually visit a gynecologist due to primary amenorrhea. Case. A forty-eight-year-old woman with lower abdominal pain and anamnesis of uterus agenesis was operated on due to bilateral cystic masses. A 5 × 3 × 1.2 cm left adnexal cyst revealed the presence of a serous cyst with a hypoplastic ductus deferens. A smaller cyst of the right adnexa...

  4. Lung hypoplasia and patellar agenesis in Ehlers-Danlos syndrome.

    Science.gov (United States)

    Pradhan, P; Deb, J; Deb, R; Chakrabarti, S

    2009-12-01

    A 22-year-old male patient was admitted with severe cough associated with purulent expectoration, left-sided chest pain and breathlessness. There was a history of recurrent respiratory ailments since childhood. The patient appeared younger than his chronological age. His face and ears were both dysmorphic. Clinically, the patient was diagnosed with Ehlers-Danlos syndrome (EDS). Computed tomography of the thoracic region revealed hypoplasia of the left lung and hyperplasia of the right lung. Both the patellae were absent. However, ultrasonography of his abdomen, echocardiography and other routine blood and urine examination showed no gross abnormalities. Although other respiratory tract abnormalities with EDS are not uncommon, unilateral lung hypoplasia and patellar agenesis in EDS make this case unique. PMID:20087544

  5. Obstructed hemivagina and ipsilateral renal agenesis with intestinal malrotation.

    Science.gov (United States)

    Morino, Masaaki; Hoshino, Masaya; Musha, Ikuma

    2013-08-01

    The combination of uterus didelphys, obstructed hemivagina, and ipsilateral renal agenesis represents a rare congenital anomaly called Herlyn-Werner-Wunderlich syndrome (HWWS) or obstructed hemivagina and ipsilateral renal anomaly (OHVIRA) syndrome. Several anomalies have recently been reported to be associated with this syndrome. The present patient with HHWS had multiple anomalies: intestinal non-rotation, anomalies of the large vessels of the abdomen including duplication of the inferior vena cava and a high-riding aortic bifurcation, and hypodontia. Hypodontia has never been reported in a patient with HWWS. The patient underwent a preventative Ladd's procedure and vaginal reconstruction. To prevent serious complications from concomitant anomalies such as intestinal malrotation, a patient with HWWS should be evaluated in detail for associated malformations. PMID:23910815

  6. Agenesis of premolar associated with submerged primary molar and a supernumerary premolar: An unusual case report

    Directory of Open Access Journals (Sweden)

    S. V. S. G. Nirmala

    2012-01-01

    Full Text Available The combination of submerged primary molar, agenesis of permanent successor with a supernumerary in the same place is very rare. The purpose of this article is to report a case of submerged mandibular left second primary molar with supernumerary tooth in the same region along with agenesis of second premolar in an 11-year-old girl, its possible etiological factors, and a brief discussion on treatment options.

  7. Herlyn-Werner-Wunderlich syndrome (uterus didelphys, blind hemivagina and ipsilateral renal agenesis) - a case report

    OpenAIRE

    Arıkan, İlker İnan; Harma, Müge; Harma, Mehmet İbrahim; Bayar, Ülkü; Barut, Aykut

    2010-01-01

    Uterovaginal duplication with obstructed hemivagina and ipsilateral renal agenesis is referred to as the Herlyn-Werner-Wunderlich (HWW) syndrome. A 17 year old woman presented with right pelvic pain and dysmenorrhea, present since menarche at 13 and worsening over the past year. Ultrasound examination revealed a right pelvic mass (5×5 cm), double endometrial echoes, and hematocolpos. A right pelvic mass, agenesis of the right kidney, double uterus, and blind hemivagina with hematocolpos were ...

  8. Agenesis of maxillary permanent first molars: literature review and two case reports

    OpenAIRE

    Anthonappa, RP; Yiu, CKY; King, NM

    2009-01-01

    Congenital absence of teeth is the most commonly known developmental dental anomaly in humans. Agenesis of the permanent first molars has the least frequency of all the tooth types and when present, usually occurs in association with oligodontia or anodontia. Generally, it is easy to diagnose agenesis of the permanent first molars retrospectively, based on the clinical morphology and x-ray photographic features of the permanent second molars; while it is often debatable when made prospectivel...

  9. Left lung agenesis discovered by a spontaneous pneumothorax in a 20-year-old girl.

    Science.gov (United States)

    Hentati, Abdessalem; Neifar, Chawki; Abid, Walid; M'saad, Sameh

    2016-01-01

    Lung agenesis is a rare condition which prognosis widely depends on associated malformations. Clinical presentation is so variable and diagnosis is often made in childhood. Here, we present a case of a 20-year-old girl who was admitted because of a spontaneous pneumothorax. Explorations concluded at a left lung agenesis, a hyperinflated right lung crossing the midline with a corresponding pneumothorax. There was no malformation else. This congenital condition and treatment for this rare presentation are discussed in detail. PMID:27051112

  10. Heterotaxy syndrome with associated agenesis of dorsal pancreas and polysplenia: A case report

    Directory of Open Access Journals (Sweden)

    Syed Althaf Ali1

    2015-01-01

    Full Text Available Heterotaxy syndrome is a rare embryological disorder comprising of polysplenia, partial agenesis of dorsal pancreas, malrotation of gut, cardiac and vascular anomalies resulting from failure of development of the usual left–right asymmetry of organs. We report a rare case of heterotaxy syndrome with polysplenia, partial agenesis of dorsal pancreas and malrotation of gut in a 28 year female presenting with subacute intestinal obstruction along with imaging illustrations, brief discussion and thorough review of literature.

  11. Pulmonary edema

    Science.gov (United States)

    ... congestion; Lung water; Pulmonary congestion; Heart failure - pulmonary edema ... Pulmonary edema is often caused by congestive heart failure . When the heart is not able to pump efficiently, blood ...

  12. Thoracoscopic plication for idiopathic eventration of the bilateral diaphragm: Report of a case

    Directory of Open Access Journals (Sweden)

    Yoshinobu Ichiki

    2015-01-01

    Conclusion: We experienced a case in which thoracoscopic plication of the unilateral diaphragm resulted in adequate objective improvements in the pulmonary function in a patient with idiopathic eventration of the bilateral diaphragm.

  13. Agenesis of third molars in students from the Dentistry Academic Program of Universidad del Valle, 16 to 25 years old

    OpenAIRE

    Sarmiento Pedro

    2004-01-01

    Background: Third molars are the teeth reported in the literature, that present more variability in number, shape and size. There is no information reported in the Colombian literature concerning the agenesis of the third molar in Colombians. This data may changes constantly from one population to another. This study was looking for agenesis of the third molars and performed on dental students at the Universidad del Valle. Objectives: Determine dental agenesis of the third molars and descri...

  14. Agenesis of the Dorsal Pancreas with Chronic Calcific Pancreatitis. Case Report, Review of the Literature and Genetic Basis

    OpenAIRE

    Vallath Balakrishnan; Venkateswara A Narayanan; Ismail Siyad; Lakshmi Radhakrishnan; Prem Nair

    2006-01-01

    Context Agenesis of the dorsal pancreas is a rare developmental anomaly. This anomaly may be complicated by recurrent acute and chronic pancreatitis.Case report We report the case of a 28-year-old female with agenesis of the dorsal pancreas and chronic calcific pancreatitis. The diagnosis of agenesis of the dorsal pancreas is discussed and the genetic changes leading to it are reviewed. The possibility of the patient having tropical pancreatitis is mentioned.Conclusions This is probably the f...

  15. Agenesis of the Dorsal Pancreas with Chronic Calcific Pancreatitis. Case Report, Review of the Literature and Genetic Basis

    Directory of Open Access Journals (Sweden)

    Vallath Balakrishnan

    2006-11-01

    Full Text Available Context Agenesis of the dorsal pancreas is a rare developmental anomaly. This anomaly may be complicated by recurrent acute and chronic pancreatitis.Case report We report the case of a 28-year-old female with agenesis of the dorsal pancreas and chronic calcific pancreatitis. The diagnosis of agenesis of the dorsal pancreas is discussed and the genetic changes leading to it are reviewed. The possibility of the patient having tropical pancreatitis is mentioned.Conclusions This is probably the first report of chronic calcific pancreatitis complicating agenesis of the dorsal pancreas

  16. Pulmonary manifestations revealing Rosai-Dorfman disease.

    Science.gov (United States)

    Goupil de Bouillé, J; de Muret, A; Diot, E; Dumont, P; Plantier, L; Diot, P; Marchand-Adam, Sylvain

    2015-01-01

    Rosai-Dorfman disease is a rare non-Langerhans cell histiocytosis, mainly involving cervical nodes. We present the case of a patient with a pulmonary form of Rosai-Dorfman disease without peripheral or intra-thoracic lymph nodes, characterized by the presence of pulmonary nodules and cysts associated with bilateral pleural effusions. PMID:26422575

  17. Pregnancy following bilateral salpingectomy

    DEFF Research Database (Denmark)

    Oturai, Annette Bang

    2008-01-01

    This report presents a rare case of spontaneous pregnancy following bilateral salpingectomy. A woman with a history of bilateral salpingectomy was admitted to hospital because of abdominal pain and positive urine HCG. Surprisingly, ultrasound confirmed a live intrauterine fetus. The pregnancy was...... unwanted, and the woman decided to terminate the pregnancy. She was offered diagnostic examination to localise a potential fistula, but she declined. In a MEDLINE search of English literature this is only the second case of spontaneous pregnancy following bilateral salpingectomy Udgivelsesdato: 2008/4/21...

  18. Primary pulmonary cryptococcosis

    OpenAIRE

    Asadi Gharabaghi, Mehrnaz; Allameh, Seyyed Farshad

    2014-01-01

    Here, we report a case of primary cryptococcal pneumonia in a 25-year-old woman who presented with several weeks’ history of cough, dyspnoea and night sweating. These symptoms started in the third trimester of her pregnancy. She was being treated for infertility and got pregnant with in vitro fertilisation. On chest imaging, there were bilateral air space consolidation and cavitary lesions. Fungal pulmonary infection was diagnosed after surgical lung biopsy. She received fluconazole 400 mg pe...

  19. Idiopathic Pulmonary Haemosiderosis

    OpenAIRE

    Gaurav Vishal; Ekta Dalal; Neelam Raval

    2013-01-01

    We report a case of idiopathic pulmonary hemosiderosis (IPH) in a 8year old female who presented with anaemia, breathlessness, puffiness of face and grade 4 clubbing. She had been investigated a number of times at multiple hos-pitals and had received multiple blood transfusions considering iron deficiency anaemia. Diagnosis of IPH was sus-pected on basis of clinical history of persistent respiratory distress, hypoxemia, repeated anaemia episodes and diffuse bilateral infiltrates on chest X ra...

  20. Pulmonary Hyalinizing Granuloma Mimicking Metastatic Lung Cancer

    OpenAIRE

    Nuri Düzgün; Ercan Kurtipek; Hıdır Esme; Meryem İlkay Eren Karanis; İsmet Tolu

    2015-01-01

    Pulmonary hyalinizing granuloma is a very rare benign condition, which usually manifests as solitary and sometimes as multiple pulmonary nodules. Deposition of immune complexes in the lung parenchyma due to hypersensitivity reactions is implicated in the etiology of pulmonary hyalinizing granuloma. A 59-year-old female patient who presented to our clinic with complaints of chest pain and cough had bilateral, multiple, and rounded lesions with regular margins suggesting metastatic lung disease...

  1. Bilateral Primary Intraocular Lymphoma

    OpenAIRE

    Mehrdad Karimi; Masoud Soheilian; Mozhgan Rezaei Kanavi

    2011-01-01

    Purpose: To report a case of bilateral primary intraocular lymphoma. Case report: A 33-year-old man presented with bilateral blurred vision since two years ago. Examination revealed large keratic precipitates, anterior chamber reaction, posterior subcapsular cataracts, and vitreous infiltration. After a short trial of topical and periocular steroids, diagnostic 25-gauge pars plana vitrectomy was performed and cytologic evaluation of the aspirate confirmed a diagnosis of intraocular lympho...

  2. Radiological case: idiopathic pulmonary hemosiderosis

    OpenAIRE

    Nascimento , J.; Morais, L; Macedo, F.

    2012-01-01

    ABSTRACT We present a case of a four year-old child with a second episode of iron deficiency anaemia. Following fever, cough and hemoptysis a chest X-ray and chest CT were performed. Bilateral and patchy areas of ground glass and consolidation were noted in the setting of intrapulmonary hemorrhage. The final diagnosis was idiopathic pulmonary hemosiderosis.

  3. Tracheal Agenesis: A Challenging Prenatal Diagnosis—Contribution of Fetal MRI

    Directory of Open Access Journals (Sweden)

    Charline Bertholdt

    2015-01-01

    Full Text Available Tracheal agenesis is a rare congenital anomaly. The prevalence is less than 1 : 50 000 with a male to female ratio of 2 : 1. This anomaly may be isolated but, in 93% of cases, it is part of polymalformative syndrome. The most evocative diagnosis situation is the ultrasonographic congenital high airway obstruction syndrome. Dilated airways, enlarged lungs with flattened diaphragm, fetal ascites and severe nonimmune hydrops can be observed. In the absence of a congenital high airway obstruction syndrome, the antenatal diagnosis of tracheal agenesis is difficult. Tracheal agenesis should be suspected in the presence of an unexplained polyhydramnios associated with congenital malformations. The fetal airway exploration should then be systematically performed by fetal thoracic magnetic resonance imaging. A case of Floyd’s type II tracheal agenesis, detected during the postnatal period, is reported here. The retrospective reexamination of fetal magnetic resonance images showed that the antenatal diagnosis would have been easy if a systematical examination of upper airways had been performed. Prenatal diagnosis of tracheal agenesis is possible with fetal MRI but the really challenge is to think about this pathology.

  4. Novel GATA6 mutations in patients with pancreatic agenesis and congenital heart malformations.

    Directory of Open Access Journals (Sweden)

    Christina S Chao

    Full Text Available Patients with pancreatic agenesis are born without a pancreas, causing permanent neonatal diabetes and pancreatic enzyme insufficiency. These patients require insulin and enzyme replacement therapy to survive, grow, and maintain normal blood glucose levels. Pancreatic agenesis is an uncommon condition but high-throughput sequencing methods provide a rare opportunity to identify critical genes that are necessary for human pancreas development. Here we present the clinical history, evaluation, and the genetic and molecular analysis from two patients with pancreatic agenesis. Both patients were born with intrauterine growth restriction, minor heart defects and neonatal diabetes. In both cases, pancreatic agenesis was confirmed by imaging studies. The patients are clinically stable with pancreatic enzymes and insulin therapy. In order identify the etiology for their disease, we performed whole exome sequencing on both patients. For each proband we identified a de novo heterozygous mutation in the GATA6 gene. GATA6 is a homeobox containing transcription factor involved in both early development of the pancreas and heart. In vitro functional analysis of one of the variants revealed that the mutation creates a premature stop codon in the coding sequence resulting in the production of a truncated protein with loss of activity. These results show how genetic mutations in GATA6 may lead to functional inactivity and pancreatic agenesis in humans.

  5. Bilateral persistent primitive trigeminal arteries associated with trigeminal neuralgia.

    Science.gov (United States)

    Son, B; Yang, S; Sung, J; Lee, S

    2013-03-01

    Persistent carotid-vertebrobasilar anastomoses (PCVBA) include the primitive trigeminal artery (PTA), the primitive otic artery (POA), the primitive hypoglossal artery and proatlantal arteries (ProAs). The PTA is the most commonly seen of these accounting for approximately 80-85% of PCVBAs. The PTA which connects the internal carotid artery (ICA) to the basilar artery (BA) may occasionally connect to the superior or posterior inferior cerebellar arteries without interposition to the BA. It is then referred to as a persistent trigeminal artery variant (PTAV), an anomalous carotid-cerebellar anastomosis. Bilateral occurrence of PTA is extremely rare. During vertebral artery (VA) development the anterior radicular artery of segment C1 from the proatlantal artery of Padget evolves into the intradural component of the VA (V4 segment) plus a short extradural segment (distal V3 segment). Agenesis of a single anterior radicular artery of ProA results in the absence of one distal VA associated with an unremarkable contralateral VA and the BA. Absence or hypoplasia of the terminal portion of one VA is a commonly observed anatomic variant. However, absence of the terminal portions of both VAs is exceptional. A rare case of bilateral PTAs is presented with unilateral PTA and a contralateral PTAV causing trigeminal neuralgia. Furthermore, the bilateral PTAs were associated with the absence of the proximal portion of the BA in addition to the bilateral lack of a distal VA. This finding comes as a logical consequence of the developmental anatomy of the vertebrobasilar junction and is consistent with the assumed congenital nature of the anatomic variant. PMID:22113402

  6. Anesthetic dilemma in planning bilateral cataract surgery for an infant associated with congenital cardiac anomaly

    Directory of Open Access Journals (Sweden)

    Devalina Goswami

    2015-01-01

    Full Text Available In a patient with tetralogy of Fallot (TOF and pulmonary atresia, treating the cardiac problem or the associated congenital illness is always a challenge. We describe the challenges and successful initial management of bilateral cataract to prevent visual loss in an infant with TOF with pulmonary atresia.

  7. Bilateral acute acquired toxoplasmic retinochoroiditis after steroid therapy for hantavirus pulmonary syndrome: case report Retinite toxoplásmica bilateral adquirida após terapia com corticoesteróide para síndrome pulmonar por hantavírus: relato de caso

    Directory of Open Access Journals (Sweden)

    Rubens Camargo Siqueira

    2007-06-01

    Full Text Available Description of a case of acute acquired ocular toxoplasmosis following hantavirus pulmonary syndrome. A 41-year-old man presenting hantavirus pulmonary syndrome, confirmed in the laboratory by detection of IgM antibodies to the virus, was submitted to high doses of intravenous corticosteroids for two months. After clinical improvement of hantavirus pulmonary syndrome the patient presented visual loss in both eyes that was secondary to a toxoplasmosis retinitis. The retinitis resolved with anti-toxoplasma therapy. Acquired toxoplasmic retinochoroiditis can occur following steroid therapy for hantavirus pulmonary syndrome.Descrição de um caso de toxoplasmose ocular adquirida pós-síndrome pulmonar por hantavírus. Paciente com 41 anos do sexo masculino apresentando síndrome pulmonar por hantavírus, confirmado no laboratório pela detecção de anticorpos IGM para o vírus, foi submetido a altas doses de corticosteróides intravenosos durante dois meses. Após melhora clínica da síndrome pulmonar por hantavírus, apresentou perda visual em ambos os olhos secundária a retinite por toxoplasmose confirmada com sorologia (IGG e IGM positivo A retinite resolveu após terapia sistêmica específica. A retinite por toxoplasmose pode ocorrer após terapia sistêmica com esteróide para síndrome pulmonar por hantavírus.

  8. [Bilateral caudate head infarcts].

    Science.gov (United States)

    Kuriyama, N; Yamamoto, Y; Akiguchi, I; Oiwa, K; Nakajima, K

    1997-11-01

    We reported a 67-year-old woman with bilateral caudate head infarcts. She developed sudden mutism followed by abulia. She was admitted to our hospital 2 months after ictus for further examination. She showed prominent abulia and was inactive, slow and apathetic. Spontaneous activity and speech, immediate response to queries, spontaneous word recall and attention and persistence to complex programs were disturbed. Apparent motor disturbance, gait disturbance, motor aphasia, apraxia and remote memory disturbance were not identified. She seemed to be depressed but not sad. Brain CT and MRI revealed bilateral caudate head hemorrhagic infarcts including bilateral anterior internal capsules, in which the left lesion was more extensive than right one and involved the part of the left putamen. These infarct locations were thought to be supplied by the area around the medial striate artery including Heubner's arteries and the A1 perforator. Digital subtraction angiography showed asymptomatic right internal carotid artery occlusion. She bad had hypertension, diabetes mellitus and atrial fibrillation and also had a left atrium with a large diameter. The infarcts were thought to be caused by cardioembolic occlusion to the distal portion of the left internal carotid artery. Although some variations of vasculature at the anterior communicating artery might contribute to bilateral medial striate artery infarcts, we could not demonstrate such abnormalities by angiography. Bilateral caudate head infarcts involving the anterior internal capsule may cause prominent abulia. The patient did not improve by drug and rehabilitation therapy and died suddenly a year after discharge. PMID:9503974

  9. Bilateral popliteal arterial dissection.

    Science.gov (United States)

    Chen, Po-Liang; Ko, Shih-Yu; Tan, Ken-Hing

    2012-01-01

    A clinical feature of bilateral popliteal arterial dissection without involving the descending aorta, bilateral iliac, as well as femoral arteries has never been reported in the past literature. We report a 56-year-old man with hypertension and coronary artery disease who presented to our emergency department with complaints of bilateral knee pain after long-distance walking. Physical examination was notable for elevated blood pressure, but there was no palpable pulsation over dorsalis pedis arteries on his feet. Laboratory evaluation revealed a d-dimer level of 35.2 mg/L (FEU) on the day of the test and 1.2 mg/L one and a half months ago (normal level, <0.55). These findings were suggestive of a recent-onset peripheral arterial occlusive disorder. Computed tomography of the aorta showed bilateral popliteal arterial dissection with arterial intimal flap. Abdominal aorta, bilateral iliac, and femoral arteries remained intact with only arteriosclerotic change. Minimally invasive endovascular stent grafting was then performed. The patient had an uneventful recovery. PMID:21106320

  10. Recessive mutations in a distal PTF1A enhancer cause isolated pancreatic agenesis.

    Science.gov (United States)

    Weedon, Michael N; Cebola, Inês; Patch, Ann-Marie; Flanagan, Sarah E; De Franco, Elisa; Caswell, Richard; Rodríguez-Seguí, Santiago A; Shaw-Smith, Charles; Cho, Candy H-H; Lango Allen, Hana; Houghton, Jayne A L; Roth, Christian L; Chen, Rongrong; Hussain, Khalid; Marsh, Phil; Vallier, Ludovic; Murray, Anna; Ellard, Sian; Ferrer, Jorge; Hattersley, Andrew T

    2014-01-01

    The contribution of cis-regulatory mutations to human disease remains poorly understood. Whole-genome sequencing can identify all noncoding variants, yet the discrimination of causal regulatory mutations represents a formidable challenge. We used epigenomic annotation in human embryonic stem cell (hESC)-derived pancreatic progenitor cells to guide the interpretation of whole-genome sequences from individuals with isolated pancreatic agenesis. This analysis uncovered six different recessive mutations in a previously uncharacterized ~400-bp sequence located 25 kb downstream of PTF1A (encoding pancreas-specific transcription factor 1a) in ten families with pancreatic agenesis. We show that this region acts as a developmental enhancer of PTF1A and that the mutations abolish enhancer activity. These mutations are the most common cause of isolated pancreatic agenesis. Integrating genome sequencing and epigenomic annotation in a disease-relevant cell type can thus uncover new noncoding elements underlying human development and disease. PMID:24212882

  11. Agenesis of the corpus callosum in a newborn with Turner mosaicism

    Directory of Open Access Journals (Sweden)

    Ester Pereira

    2014-05-01

    Full Text Available The agenesis of the corpus callosum results from a failure in the development of the largest fiber bundle that connects cerebral hemispheres. Patient’s outcome is influenced by etiology and associated central nervous system malformations. We describe a child with Turner syndrome (TS mosaicism, with particular phenotype features and a complete agenesis of the corpus callosum. To our knowledge, this is the second case report of TS mosaicism associated with complete agenesis of the corpus callosum. Anatomical brain magnetic resonance imaging and diffusion tensor imaging were useful to confirm the complete absence of the corpus callosum, evaluate associated central nervous system malformations, visualize abnormal white matter tracts (Probst bundles and assess the remaining commissures.

  12. Escleritis posterior bilateral Bilateral posterior scleritis

    Directory of Open Access Journals (Sweden)

    A. Zurutuza

    2011-08-01

    Full Text Available La escleritis posterior es un proceso inflamatorio de la parte posterior de la esclera. Su prevalencia es muy baja y el diagnóstico puede resultar complicado por la ausencia de signos oculares externos. Es más frecuente en mujeres. Cuando aparece en pacientes jóvenes no suele tener otras patologías asociadas, pero en mayores de 55 años hasta un tercio de los casos tienen relación con alguna enfermedad sistémica, sobre todo la artritis reumatoide. El diagnóstico de esta patología puede requerir un abordaje multidisciplinar y la colaboración de oftalmólogos con neurólogos, internistas o reumatólogos. En este artículo se describe un caso de escleritis posterior bilateral idiopática.Posterior scleritis is an inflammatory process of the posterior part of the sclera. Its prevalence is very low and its diagnosis can be complicated due to the absence of external ocular signs. It is more frequent in women. In young patients it does not usually have other associated pathologies, but in those over 55 years nearly one-third of the cases have a relation with some systemic disease, above all rheumatoid arthritis. The diagnosis of this pathology can require a multidisciplinary approach and the collaboration of ophthalmologists with neurologists, internists or rheumatologists. This article describes a case of idiopathic bilateral posterior scleritis.

  13. Bilaterally Incarcerated Morgagni Hernia

    Directory of Open Access Journals (Sweden)

    Zuhal Demirhan Yananli

    2013-06-01

    Full Text Available Morgagni hernia is a rare congenital diaphragmatic hernia. It is seen rarely bilaterally. Patients are usually asymptomatic. Therefore, diagnosis may be delayed until adulthood. Significant morbidity can occur in case complications arise and diagnosis is delayed. The patient, a 74 year-old female, presented in this article, was admitted to the emergency department with abdominal pain, vomiting, and shortness of breath. The plain abdominal radiograph of the patient revealed bowel obstruction and suspicious appearence in favor of the diaphragmatic hernia on both sides of the sternum. Computed tomography revealed bilaterally incarcerated Morgagni hernia with strangulated omentum in the right side of the sternum and a part of colon in the left side of sternum. Incarcerated organs were withdrawn to peritoneal cavity and defects of hernia were sutured primarily on laparatomy. Because bilateral incarcerated Morgagni hernia can be seen rarely, this case was reported.

  14. Atypical Radiological Manifestation of Pulmonary Metastatic Calcification

    Energy Technology Data Exchange (ETDEWEB)

    Kang, Eun Hae; Kim, Eun Sun; Kim, Chul Hwan; Ham, Soo Youn; Oh, Yu Whan [Korea University College of Medicine, Seoul (Korea, Republic of)

    2008-04-15

    Metastatic pulmonary calcification is a condition of calcium deposition in the normal pulmonary parenchyma, and this is secondary to abnormal calcium metabolism without any prior soft tissue damage. The predisposing factors for this condition include chronic renal failure, hypercalcemia and increased tissue alkalinity. The most common radiologic manifestation consists of poorly defined nodular opacities in the upper lung zone. These opacities reflect the deposition of calcium salts in the pulmonary interstitium. We present here a case of metastatic pulmonary calcification in a patient who recovered from pneumonia with sepsis and whose high-resolution CT (HRCT) images demonstrated localized parenchymal airspace calcification that was limited to the bilateral lower lobes. These lower lobes had been involved with pneumonic consolidation without calcification, as seen on the previous CT scan. In summary, we report here on an atypical presentation of metastatic pulmonary calcification that showed dense airspace consolidation localized to the bilateral lower lobes in a patient with primary hyperparathyroidism and pneumonia.

  15. Efficacy of bilateral pallidotomy.

    Science.gov (United States)

    Kim, R; Alterman, R; Kelly, P J; Fazzini, E; Eidelberg, D; Beric, A; Sterio, D

    1997-03-15

    Unilateral pallidotomy is a safe and effective treatment for medically refractory bradykinetic Parkinson's disease, especially in those patients with levodopa-induced dyskinesia and severe on-off fluctuations. The efficacy of bilateral pallidotomy is less certain. The authors completed 11 of 12 attempted bilateral pallidotomies among 150 patients undergoing pallidotomy at New York University. In all but one patient, the pallidotomies were separated by at least 9 months. Patients were selected for bilateral pallidotomy if they exhibited bilateral rigidity, bradykinesia, or levodopa-induced dyskinesia prior to treatment or if they exhibited disease progression contralateral to their previously treated side. The Unified Parkinson's Disease Rating Scale (UPDRS) and timed upper-extremity tasks of the Core Assessment Protocol for Intracerebral Transplantation (CAPIT) were administered to all 12 patients in the "off" state (12 hours without receiving medications) preoperatively and again at 6 and 12 months after each procedure. The median UPDRS and contralateral CAPIT scores improved 60% following the initial procedure (p = 0.008, Wilcoxon rank sums test). The second pallidotomy generated only an additional 10% improvement in the UPDRS and CAPIT scores ipsilateral to the original procedure (p = 0.05). Worsened speech was observed in two cases. In the 12th case, total speech arrest was noted during test stimulation. Speech returned within minutes after stimulation was halted. Lesioning was not performed. These results indicate that bilateral pallidotomy has a narrow therapeutic window. Motor improvement ipsilateral to the first lesion leaves little room for further improvement from the second lesion and the risk of speech deficit is greatly enhanced. Chronic pallidal stimulation contralateral to a previously successful pallidotomy may prove to be a safer alternative for the subset of patients who require bilateral procedures. PMID:15096015

  16. Primary Pulmonary Epithelioid Hemangioendothelioma: A Rare Cause of PET-Negative Pulmonary Nodules

    Directory of Open Access Journals (Sweden)

    Riccardo Cazzuffi

    2011-01-01

    Full Text Available We report here a case of primary pulmonary epithelioid hemangioendothelioma diagnosed in a 67-year-old Caucasian man, presenting with exertion dyspnoea, dry cough, and multiple bilateral pulmonary nodules revealed by computed tomography. At the 18F-fluorodeoxyglucose positron emission tomography, these nodules were negative. The histopathological diagnosis was made on a pulmonary wedge resection (performed during video-thoracoscopic surgery.

  17. Bilateral Primary Intraocular Lymphoma

    Directory of Open Access Journals (Sweden)

    Mehrdad Karimi

    2011-01-01

    Full Text Available Purpose: To report a case of bilateral primary intraocular lymphoma. Case report: A 33-year-old man presented with bilateral blurred vision since two years ago. Examination revealed large keratic precipitates, anterior chamber reaction, posterior subcapsular cataracts, and vitreous infiltration. After a short trial of topical and periocular steroids, diagnostic 25-gauge pars plana vitrectomy was performed and cytologic evaluation of the aspirate confirmed a diagnosis of intraocular lymphoma. The patient was subsequently managed with intravitreal methotrexate in both eyes and responded favorably. Central nervous system workup for lymphoma was negative. Conclusion: Primary intraocular lymphoma should be considered in young adults suffering from chronic recalcitrant panuveitis.

  18. Bilateral neck paragangliomas.

    Science.gov (United States)

    Mumoli, N; Cei, M; Pauletti, M; Ferrito, G; Scazzeri, F

    2009-10-01

    Paragangliomas of the head and neck are rare neoplasms presented as cervical mass, generally bilateral, that arise from chemoreceptors located at the carotid bifurcation (carotid body tumors), along the vagus nerve (vagal paragangliomas), and in the jugular fossa and tympanic cavity (jugulotympanic paragangliomas). They are typically asymptomatic at the beginning, highly vascular, slow-growing and compressing the surrounding anatomic structures. Only radical surgery is the curative treatment for paragangliomas. We present a case of a 62- year-old woman with a diagnosis of bilateral neck paragangliomas where surgical removal was judged burdened by excessive risk because of the size of the tumor. PMID:19622673

  19. BILATERAL IMMATURE OVARIAN TERATOMA

    OpenAIRE

    Vinay; Aditya Pratap; Chetan; Ramesh; Rajlaxmi Jaysing

    2014-01-01

    : Immature teratoma (IMT) is tumor composed of tissues from ectoderm, mesoderm and endoderm and is considered the second most common germ cell tumor. IMT account for 10-20% of all ovarian neoplasias in women less than 20 years of age, with peak incidence between 15 and 19 years old. IMT rarely occurs during menopause. We herein reporting a rare case in a 3 years old girl with bilateral immature ovarian teratoma which is very rare in bilateralism of tumor as well as the fac...

  20. BILATERAL IMMATURE OVARIAN TERATOMA

    Directory of Open Access Journals (Sweden)

    Vinay

    2014-10-01

    Full Text Available : Immature teratoma (IMT is tumor composed of tissues from ectoderm, mesoderm and endoderm and is considered the second most common germ cell tumor. IMT account for 10-20% of all ovarian neoplasias in women less than 20 years of age, with peak incidence between 15 and 19 years old. IMT rarely occurs during menopause. We herein reporting a rare case in a 3 years old girl with bilateral immature ovarian teratoma which is very rare in bilateralism of tumor as well as the fact that the patient age is below the average for the occurrence of these tumors.

  1. Uterus didelphys with unilateral obstructed hemivagina and haematocolpos with ipsilateral renal agenesis - a case report.

    Science.gov (United States)

    Chowdhury, S; Ara, R; Begum, S A; Chowdhury, S B; Hussain, M A; Mirza, T T

    2015-01-01

    Mullerian anomalies are a relatively uncommon occurrence with implication for adolescents and adults as they may result in specific gynaecologic, fertility and obstetrical issues. Uterus didelphys with blind hemivagina and ipsilateral renal agenesis is a rare congenital anomaly. Patient may be asymptomatic and unaware of having double uterus or may present with severe dysmenorrhoea or dyspareunia or a palpable mass due to unilateral haematocolpos. We report a case of 12 year old girl with this condition who was diagnosed as uterus didelphys with unilateral haematocolpos with ipsilateral renal agenesis on the basis of clinical association, physical examination and sonography and intravenous urogram. PMID:25725693

  2. Uterus didelphys with unilateral obstructed hemivagina with hematometrocolpos and hematosalpinx with ipsilateral renal agenesis

    Directory of Open Access Journals (Sweden)

    Gaurav Jindal

    2009-01-01

    Full Text Available Uterus didelphys with blind hemivagina and ipsilateral renal agenesis (Herlyn Werner-Wunderlich Syndrome is a rare congenital anomaly. It mostly presents with severe dysmenorrhea and a palpable mass due to unilateral hematocolpos. A patient with dysmenorrhea from a double uterus and an obstructed hemivagina is a diagnostic dilemma because the menses are regular. We report a case of a 14-year-old girl with this condition who was diagnosed as uterus didelphys with unilateral hematocolpos and hydrosalpinx with ipsilateral renal agenesis on the basis of sonography and confirmed by laparoscopic examination.

  3. MR diagnosis of penile agenesis: is it just absence of a phallus?

    Energy Technology Data Exchange (ETDEWEB)

    Goenka, Ajit; Jain, Vaibhav; Sharma, Raju; Gupta, Arun K. [All India Institute of Medical Sciences, Department of Radiodiagnosis, New Delhi (India); Bajpai, Minu [All India Institute of Medical Sciences, Department of Paediatric Surgery, New Delhi (India)

    2008-10-15

    Penile agenesis is an extremely rare anomaly that results from absence of the genital tubercle or its failure to develop into a penis during embryonic life. Associated anomalies of the genitourinary and distal gastrointestinal tracts are frequently present. Imaging modalities, particularly MRI and cystography, play a crucial role in establishing the diagnosis and guiding further management. We report a child with penile agenesis with a urethroanal fistula and a blind-ending anterior urethra who had been reared as a male until presentation. (orig.)

  4. Bart’s Syndrome Associated Corpus Callosum Agenesis and Choanal Atresia

    OpenAIRE

    Saeed, Muhammad; Anwar ul HAQ; Khaqan QADIR

    2014-01-01

    How to Cite This Article: Saeed M, Haq A, Qadir Kh.Bart’s Syndrome Associated Corpus Callosum Agenesis and Choanal Atresia. Iran J Child Neurol. 2014 Autumn;8(4): 76-79.AbstractObjectiveBart’s syndrome is defined as congenital localized absence of skin, and associated with epidermolysis bullosa. A newborn with Bart’s syndrome is reported because it is a very rare condition, especially when associated with corpus callosum agenesis and concomitant choanal atresia. Clinically it is characterized...

  5. Sacral Agenesis with Neurogenic Bladder Dysfunction—A Case Report and Review of the Literature

    Science.gov (United States)

    Sharma, Seema; Awasthi, Bhanu; Sehgal, Manik; Singla, Deeksha A

    2015-01-01

    Sacral agenesis (part of the caudal regression syndrome) is a rare and severe sacral developmental abnormality. It is a congenital malformation of unknown aetiology with possible involvement of genetic and teratogenic factors. It is described by various degrees of developmental failure, the most extreme and rare being sirenomelia or mermaid syndrome. The associated malformations comprise anorectal, vertebral, urological, genital, and lower limb anomalies. Approximately 15-20% mothers of these children have insulin dependent diabetes mellitus. The case is being reported for its rarity and educative value because prognosis is good in isolated sacral agenesis. PMID:26266174

  6. Sacral Agenesis with Neurogenic Bladder Dysfunction-A Case Report and Review of the Literature.

    Science.gov (United States)

    Sharma, Seema; Sharma, Vipin; Awasthi, Bhanu; Sehgal, Manik; Singla, Deeksha A

    2015-06-01

    Sacral agenesis (part of the caudal regression syndrome) is a rare and severe sacral developmental abnormality. It is a congenital malformation of unknown aetiology with possible involvement of genetic and teratogenic factors. It is described by various degrees of developmental failure, the most extreme and rare being sirenomelia or mermaid syndrome. The associated malformations comprise anorectal, vertebral, urological, genital, and lower limb anomalies. Approximately 15-20% mothers of these children have insulin dependent diabetes mellitus. The case is being reported for its rarity and educative value because prognosis is good in isolated sacral agenesis. PMID:26266174

  7. Left lung agenesis discovered by a spontaneous pneumothorax in a 20-year-old girl

    Directory of Open Access Journals (Sweden)

    Abdessalem Hentati

    2016-01-01

    Full Text Available Lung agenesis is a rare condition which prognosis widely depends on associated malformations. Clinical presentation is so variable and diagnosis is often made in childhood. Here, we present a case of a 20-year-old girl who was admitted because of a spontaneous pneumothorax. Explorations concluded at a left lung agenesis, a hyperinflated right lung crossing the midline with a corresponding pneumothorax. There was no malformation else. This congenital condition and treatment for this rare presentation are discussed in detail.

  8. MR diagnosis of penile agenesis: is it just absence of a phallus?

    International Nuclear Information System (INIS)

    Penile agenesis is an extremely rare anomaly that results from absence of the genital tubercle or its failure to develop into a penis during embryonic life. Associated anomalies of the genitourinary and distal gastrointestinal tracts are frequently present. Imaging modalities, particularly MRI and cystography, play a crucial role in establishing the diagnosis and guiding further management. We report a child with penile agenesis with a urethroanal fistula and a blind-ending anterior urethra who had been reared as a male until presentation. (orig.)

  9. Pancreas-specific deletion of mouse Gata4 and Gata6 causes pancreatic agenesis

    OpenAIRE

    Xuan, Shouhong; Borok, Matthew J.; Decker, Kimberly J.; Battle, Michele A.; Duncan, Stephen A.; Hale, Michael A.; MacDonald, Raymond J.; Sussel, Lori

    2012-01-01

    Pancreatic agenesis is a human disorder caused by defects in pancreas development. To date, only a few genes have been linked to pancreatic agenesis in humans, with mutations in pancreatic and duodenal homeobox 1 (PDX1) and pancreas-specific transcription factor 1a (PTF1A) reported in only 5 families with described cases. Recently, mutations in GATA6 have been identified in a large percentage of human cases, and a GATA4 mutant allele has been implicated in a single case. In the mouse, Gata4 a...

  10. Chronic renal failure due to unilateral renal agenesis and total renal dysplasia (=aplasia)

    International Nuclear Information System (INIS)

    Three adult patients with unilateral renal agenesis/total dysplasia (= aplasia) and with an early chronic renal failure are presented. One patient had renal agenesis without ureter bud and ureteric ostium on one side, and reflux pyelonephritis on the other; one had small compact total renal dysplasia (= aplasia) on one side, while chronic uric acid nephropathy (chronic renal disease as a cause of gout) was diagnosed on the other; the third patient had a total large multicystic dysplasia on one side, and on the other a segmental large multicystic dysplasia. Radiological steps and radiodiagnostic criteria are discussed and the combination of urogenital and extraurogenital anomalies is referred to. (orig.)

  11. Chronic renal failure due to unilateral renal agenesis and total renal dysplasia (=aplasia)

    Energy Technology Data Exchange (ETDEWEB)

    Kroepelin, T.; Ziupa, J.; Wimmer, B.

    1983-05-01

    Three adult patients with unilateral renal agenesis/total dysplasia (= aplasia) and with an early chronic renal failure are presented. One patient had renal agenesis without ureter bud and ureteric ostium on one side, and reflux pyelonephritis on the other; one had small compact total renal dysplasia (= aplasia) on one side, while chronic uric acid nephropathy (chronic renal disease as a cause of gout) was diagnosed on the other; the third patient had a total large multicystic dysplasia on one side, and on the other a segmental large multicystic dysplasia. Radiological steps and radiodiagnostic criteria are discussed and the combination of urogenital and extraurogenital anomalies is referred to.

  12. Effect of Vestibulo-Proprioceptive Stimulations in a Child with Agenesis of the Corpus Callosum

    OpenAIRE

    Hamid Dalvand; Leila Dehghan; Dr Hossein Bagheri

    2010-01-01

    Background and Aim: The purpose of the present study was to investigate the effect of vestibulo-proprioceptive stimulations of sensory integration theory on the development of gross and fine motor, language and personal-social functions in a child with agenesis of the corpus callosum.Case: We report a 10.5 month old boy with agenesis of the corpus callosum. The intervention was administered based on sensory integration theory an hour a week for 20 weeks. The exercise intervention consisted of...

  13. Pancreatic Agenesis with Congenital Diaphragmatic Hernia and Congenital Heart Disease: A Case Report

    Directory of Open Access Journals (Sweden)

    Atsushi Nakao

    2013-10-01

    Full Text Available Pancreatic agenesis is a rare disease that causes neonatal diabetes mellitus and exocrine pancreatic insufficiency. We report the case of a very low birth weight infant with congenital diaphragmatic hernia, and congenital heart disease (ventricular septal defect and patent ductus arteriosus. Failure to gain weight, despite well-managed respiratory and heart failure, was improved by infusion of subcutaneous insulin, administration of pancreatic enzyme, and nutrition of medium-chain-triglyceride -enriched formula. This is the first case of pancreatic agenesis with both malformations where the patient is discharged from the hospital. Early diagnosis and adequate treatments to compensate pancreatic function may prevent mortality and improve growth.

  14. Complicated Varicella Infection at 8-year-old Boy with Pulmonary Agenesis

    Science.gov (United States)

    Hadzovic–Cengic, Meliha; Baljic, Rusmir; Hadzic, Amir; Lukovac, Enra; Mehanic, Snjezana; Ahmetspahic-Begic, Aida

    2012-01-01

    SUMMARY CONFLICT OF INTEREST: none declared. Introduction Varicella or chickenpox is highly contagious, childhood infectious disease caused by primary infection with varicella – zoster virus from the herpes family of viruses. Usually it has a mild clinical course, rarely with described complication, mostly affecting respiratory tract and rarely the central nervous system. Case report The case present 8 year old boy hospitalized eighth day of disease with clinical pictures of varicella complication. Upon receipt tachydyspnea, high fever, tachycardia, hypotensive with positive findings on lung auscultation in the sense of pneumonia. Extremely high values of non-specific inflammatory parameters are implied on bacterial infection which is treated using triple antimicrobial therapy and antiviral. A detailed clinical, laboratory and radiological evaluation is determined of clinical disease complication under a picture of MODS that required prolonged multidisciplinary treatment in ICU. Conclusion The disease had a favorable clinical outcome in terms of training completely without consequences but, with the detected congenital absence lower lobe of right lung and transposition of the brachiocephalic trunk. PMID:24493991

  15. Bilateral akillesseneruptur hos nyretransplanterede

    DEFF Research Database (Denmark)

    Skovgaard, D; Feldt-Rasmussen, B F; Nimb, L;

    1996-01-01

    Increased incidence of tendinitis and tendon ruptures is reported in recipients of a kidney transplant. Two cases of bilateral achilles tendon rupture after minimal trauma are described. Tendon ruptures are more frequent in individuals with kidney disease in dialysis or after transplantation comp...

  16. Bilateral endogenous fungal endophthalmitis

    OpenAIRE

    Michal, Wilczynski; Olena, Wilczynska; Wojciech, Omulecki

    2013-01-01

    Endogenous endophthalmitis is a rare and severe intraocular infection which can be vision-threatening. We describe a case of bilateral fungal endogenous endophthalmitis in a 64-year-old male which was successfully treated with systemic administration of fluconazole followed by pars plana vitrectomy with an intravitreous injection of amphotericin B.

  17. Bilateral chronic subdural hematoma

    DEFF Research Database (Denmark)

    Andersen-Ranberg, Nina Christine; Poulsen, Frantz Rom; Bergholt, Bo;

    2016-01-01

    ) surgery. The overall retreatment rate was 21.6% (57 of 264 patients). Cases treated with unilateral surgery had twice the risk of retreatment compared with cases undergoing bilateral surgery (28.7% vs 14.1%, respectively, p = 0.002). In accordance with previous studies, the data also showed that a...

  18. Bilateral lunate intraosseous ganglia

    International Nuclear Information System (INIS)

    An intraosseous ganglion is a relatively uncommon, benign, cyst-like lesion that occurs in young and middle-aged adults. Most commonly seen adjacent to the hip, ankle, knee, or wrist, they are histologically identical to their soft tissue counterparts. A review of the literature revealed only two previously reported examples of bilateral symmetrical ganglia of the lunate bones. (orig.)

  19. Pulmonary alveolar proteinosis

    Directory of Open Access Journals (Sweden)

    Hadice Selimoğlu Şen

    2011-06-01

    Full Text Available Pulmonary alveolar proteinosis is a rare disease characterized by the accumulation of lipoproteinosis material within the alveoli. A 36 age male patient applied to our hospital with dyspnea that began 5 months ago. Bilateral diffuse infiltration on postero-anterior chest graphy was seen. Arterial blood gas measurements (ABG was: pH:7.44, pCO2: 36 mmHg, pO2: 49 mmHg, SaO2: 85%, HCO3: 24 mmol/L. High resolution computed tomography (HRCT reported as bilateral diffuse ground glass areas and interlobuler septal thickening on this areas. Asido resistant bacillus was negative in sputum at three times and was also negative in post bronchoscopic sputum and bronchoalveoler lavage material. Wedge resection was made by video-assisted thoracoscopic surgery. Histopathology report of biopsy material revealed “Pulmonary alveolar proteinosis (PAP”. Massive lung lavage under general anesthesia was planned for patient. Clinical improvement was seen in patient beginning from admission and ABG measurements entered the recovery trend in follow-up. Partial pressure of oxygen was increased to 65 mmHg and patient was followed without lung lavage. Control chest radiography and HRCT showed significant radiological improvement. After three months, radiological lesions had completely regressed at control HRCT. As a result, in patients with symptoms and radiologically bilateral diffuse infiltration physicians should consider PAP as a rare disease in the differential diagnosis.

  20. Bilateral adrenal histoplasmosis in an immunocompetent man Histoplasmose adrenal bilateral em um homem imunocompetente

    Directory of Open Access Journals (Sweden)

    Carlos Frederico Lopes Benevides

    2007-04-01

    Full Text Available Histoplasmosis is a fungal disease that is endemic in Brazil. It may present as chronic pulmonary infection or in disseminated form. Disseminated histoplasmosis frequently affects the adrenal gland; however, unilateral involvement in immunosuppressed patients is the usual presentation. We report a case of an elderly immunocompetent male with history of weight loss, fever and bilateral adrenal mass who was successfully treated with itraconazole.Histoplasmose é uma doença fúngica endêmica no Brasil que pode se apresentar como infecção pulmonar crônica ou na forma disseminada. A histoplasmose disseminada freqüentemente acomete a glândula adrenal; entretanto, ocorre mais em pacientes imunossuprimidos e de forma unilateral. Relatamos um caso de um homem idoso imunocompetente com história de perda de peso, febre e massa adrenal bilateral que foi tratada com itraconazol, com sucesso.

  1. Development of remnant kidney in the chick embryos with unilateral renal agenesis

    Czech Academy of Sciences Publication Activity Database

    Zemanová, Zdeňka; Jirsová, Z.; Murphy, M. J.

    Berlin : Institute of biology, 2005. s. 61-61. [Combined workshop of fundamental physiology of the european working group of physiology and perinatal development poultry /2./. 23.09.2005-25.09.2005, Berlin] R&D Projects: GA ČR GA304/04/0972 Keywords : chick embryo * renal agenesis * mesonephros Subject RIV: EA - Cell Biology

  2. Professional and patient-based evaluation of oral rehabilitation in patients with tooth agenesis

    DEFF Research Database (Denmark)

    Dueled, Erik; Gotfredsen, Klaus; Trab Damsgaard, Mogens;

    2009-01-01

    OBJECTIVES: The outcome of oral rehabilitation is usually monitored with clinical tests rather than by patient's perception of change. The aim of this study was to describe the objective measure and subjective perception of oral rehabilitation in patients with tooth agenesis. MATERIAL AND METHODS...

  3. Novel PAX9 gene polymorphisms and mutations and susceptibility to tooth agenesis in the Czech population

    Czech Academy of Sciences Publication Activity Database

    Hloušková, A.; Bonczek, Ondřej; Izakovičová Hollá, L.; Lochman, J.; Šoukalová, J.; Štembírek, Jan; Míšek, Ivan; Černochová, P.; Krejčí, P.; Vaněk, J.; Šerý, Omar

    2015-01-01

    Roč. 36, č. 5 (2015), s. 101-106. ISSN 0172-780X R&D Projects: GA MZd(CZ) NT11420 Institutional support: RVO:67985904 Keywords : odontogenesis * tooth agenesis * PAX9 gene Subject RIV: FF - HEENT, Dentistry Impact factor: 0.799, year: 2014

  4. Non-syndromic tooth agenesis associated with a nonsense mutation in ectodysplasin-A (EDA).

    Science.gov (United States)

    Nikopensius, T; Annilo, T; Jagomägi, T; Gilissen, C; Kals, M; Krjutškov, K; Mägi, R; Eelmets, M; Gerst-Talas, U; Remm, M; Saag, M; Hoischen, A; Metspalu, A

    2013-06-01

    Mutations in the ectodysplasin-A (EDA) gene have been generally associated with X-linked hypohidrotic ectodermal dysplasia (XLHED). Recently, missense mutations in EDA have been reported to cause familial non-syndromic tooth agenesis. In this study, we report a novel EDA mutation in an Estonian family segregating non-syndromic tooth agenesis with variable expressivity. Affected individuals had no associated defects in other ectodermal organs. Using whole-exome sequencing, we identified a heterozygous nonsense mutation c.874G>T (p.Glu292X) in the TNF homology domain of EDA in all affected female patients. This protein-altering variant arose de novo, and the potentially causative allele was transmitted to affected offspring from the affected mother. We suggest that the dental phenotype variability described in heterozygous female carriers of EDA mutation may occur because of the differential pattern of X-chromosome inactivation, which retains reduced levels of EDA-receptor signaling in tissues involved in tooth morphogenesis. This results in selective tooth agenesis rather than XLHED phenotype. The present study broadens the mutation spectrum for this locus and demonstrates that EDA mutations may result in non-syndromic tooth agenesis in heterozygous females. PMID:23603338

  5. Unilateral lacrimal fistula in a patient with uterus didelphys and renal agenesis.

    Science.gov (United States)

    Turan-Vural, Ece; Atmaca, Fatih; Unlu, Cihan; Erdogan, Gurkan; Bayramlar, Huseyin; Bayramalar, Huseyin

    2012-02-01

    A 30-year-old female patient presented to our clinic because of lacrimation from an orifice close to the left lower eyelid. Ocular examinations and analyses revealed uterus didelphys and unilateral renal agenesis associated with a left lacrimal fistula. The patient underwent fistulectomy and external dacryocystorhinostomy. We decided to report on this patient owing to the unusual concurrent systemic abnormalities. PMID:22258183

  6. Bilateral intraocular dirofilariasis

    Directory of Open Access Journals (Sweden)

    Viney Gupta

    2014-01-01

    Full Text Available Ocular dirofilariasis mostly presents as a subconjunctival or eyelid lesion. [1] Intraocular dirofilarial infestation is rare. [2],[3] We report a case of a young woman who was accidentally detected to have a live motile worm in the anterior segment in one eye and a cystic lesion on the optic disc in the other eye. To our knowledge, bilateral intraocular dirofilariasis has never been reported.

  7. Bilateral ocular osseous choristomas

    Energy Technology Data Exchange (ETDEWEB)

    Moon, Jeung Hee; Yoon, Dae Young; Choi, Chul Soon; Yoon, Eun Joo; Park, Sang Joon; Seo, Young Lan [Hallym University, Department of Radiology, Kangdong Sacred Heart Hospital, Seoul (Korea); Kim, Byoung Jin [Hallym University, Department of Ophthalmology, Kangdong Sacred Heart Hospital, Seoul (Korea)

    2005-11-01

    Choristoma is a benign tumour defined as normal tissue in an ectopic location. Osseous choristoma, one subtype of this entity, occurring within the orbit has rarely been reported in the world literature. We report a 6-year-old girl with bilateral ocular osseous choristomas who presented with palpable nodules protruding from both upper lids. The radiological and clinical findings are described and previous reports are reviewed. (orig.)

  8. Bilateral Integrative Medicine, Obviously

    OpenAIRE

    Shapiro, Simon J.; Stumpf, Steven H.

    2006-01-01

    Unstated and unacknowledged bias has a profound impact on the nature and implementation of integrative education models. Integrative education is the process of training conventional biomedical and traditional Chinese medicine practitioners in each tradition such that patient care may be effectively coordinated. A bilateral education model ensures that students in each tradition are cross-taught by experts from the ‘other’ tradition, imparting knowledge and values in unison. Acculturation is ...

  9. Bilateral lunate intraosseous ganglia

    Energy Technology Data Exchange (ETDEWEB)

    Pablos, J.M. [Department of Radiology, Hospital San Juan de Dios, Seville (Spain); Valdes, J.C. [Department of Radiology, Cemedi, Seville (Spain); Gavilan, F. [Department of Pathology, Hospital Universitario Virgen del Rocio, Seville (Spain)

    1998-12-01

    An intraosseous ganglion is a relatively uncommon, benign, cyst-like lesion that occurs in young and middle-aged adults. Most commonly seen adjacent to the hip, ankle, knee, or wrist, they are histologically identical to their soft tissue counterparts. A review of the literature revealed only two previously reported examples of bilateral symmetrical ganglia of the lunate bones. (orig.) With 3 figs., 10 refs.

  10. Compressive Bilateral Filtering.

    Science.gov (United States)

    Sugimoto, Kenjiro; Kamata, Sei-Ichiro

    2015-11-01

    This paper presents an efficient constant-time bilateral filter that produces a near-optimal performance tradeoff between approximate accuracy and computational complexity without any complicated parameter adjustment, called a compressive bilateral filter (CBLF). The constant-time means that the computational complexity is independent of its filter window size. Although many existing constant-time bilateral filters have been proposed step-by-step to pursue a more efficient performance tradeoff, they have less focused on the optimal tradeoff for their own frameworks. It is important to discuss this question, because it can reveal whether or not a constant-time algorithm still has plenty room for improvements of performance tradeoff. This paper tackles the question from a viewpoint of compressibility and highlights the fact that state-of-the-art algorithms have not yet touched the optimal tradeoff. The CBLF achieves a near-optimal performance tradeoff by two key ideas: 1) an approximate Gaussian range kernel through Fourier analysis and 2) a period length optimization. Experiments demonstrate that the CBLF significantly outperforms state-of-the-art algorithms in terms of approximate accuracy, computational complexity, and usability. PMID:26068315

  11. Idiopathic Pulmonary Haemosiderosis

    Directory of Open Access Journals (Sweden)

    Gaurav Vishal

    2013-06-01

    Full Text Available We report a case of idiopathic pulmonary hemosiderosis (IPH in a 8year old female who presented with anaemia, breathlessness, puffiness of face and grade 4 clubbing. She had been investigated a number of times at multiple hos-pitals and had received multiple blood transfusions considering iron deficiency anaemia. Diagnosis of IPH was sus-pected on basis of clinical history of persistent respiratory distress, hypoxemia, repeated anaemia episodes and diffuse bilateral infiltrates on chest X ray. [Natl J Med Res 2013; 3(3.000: 292-293

  12. Pulmonary alveolar proteinosis

    International Nuclear Information System (INIS)

    Since the original description of pulmonary alveolar proteinosis (PAP) as bilaterally symmetric, confluent, perihilar infiltrates, no large series evaluating the relative frequency of radiographic findings has been published. The authors reviewed 53 histologically proved cases and found a predominantly confluent pattern in 77.4%; of these, 61% had a nodular component. A predominantly nodular pattern was seen in 22.6% often with some coalescence or reticular shadows. Distribution was perhilar 56.6%, diffuse in 26.4%, basilar in 26.4%, and asymmetric in 35.8%. The classic pattern was seen in 18.9%. The authors conclude that the spectrum of patterns in pulmonary alveolar proteinosis is wider than generally appreciated

  13. Bilateral primary adrenal lymphoma presenting with adrenal insufficiency

    DEFF Research Database (Denmark)

    Holm, Jakob; Breum, Leif; Stenfeldt, Katrine; Friberg Hitz, Mette

    2012-01-01

    surgery was performed. A new computerized tomography scan showed rapid progression of disease with further enlargement of the adrenal masses and both pulmonary and hepatic metastasis. Needle biopsy was performed but the patient refused further treatment and died before a diagnosis was obtained. The...... immuneohistochemical diagnosis was large B-cell lymphoma. This case should remind clinicians that PAL may be a cause of bilateral adrenal incidentaloma especially if the patient presents with adrenal insufficiency....

  14. Neuralgic Amyotrophy: A Rare Cause of Bilateral Diaphragmatic Paralysis

    OpenAIRE

    Neil Shinder; Alasdair Polson; Elizabeth Pringle; Denis E O'Donnell

    1998-01-01

    Neuralgic amyotrophy, also known as brachial neuritis, is a well described clinical entity. Diaphragmatic dysfunction, as a result of phrenic nerve root involvement (cervical roots 3 to 5), is an uncommon, but increasingly recognized association. The case of a previously healthy 61-year-old woman who, after a prodrome of neck and shoulder discomfort, presented with severe orthopnea is described. Pulmonary function and electrophysiological studies led to a diagnosis of bilateral diaphragmatic ...

  15. Sonographic findings of the uterus didelphys associated with obstructed hemivagina and ipsilateral renal agenesis: Report of 4 cases

    International Nuclear Information System (INIS)

    The association of uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis is rare anomaly. It is caused by failure in the fusion of the caudal portion of the M llerian duct, secondary to Wolffian duct anomaly. We report 4 cases of uterus didelphys with an obstructed hemivagina and ipsilateral renal agenesis. All 4 cases are diagnosed by ultrasonogram, IVU, CT and MRI findings and confirmed by surgery or diagnostic laparoscopy and vaginal examination.

  16. Sonographic findings of the uterus didelphys associated with obstructed hemivagina and ipsilateral renal agenesis: Report of 4 cases

    Energy Technology Data Exchange (ETDEWEB)

    Kim, Jung Hae; Lee, Hyeon Keong; Choi, Dae Seob; Lee, Sung Woo; Bae, Chul Sung; Yoon, Hye Won; Kim, Mi Woon; Kim, Sung Ja [Dongguk University College of Medicine, Kyungju (Korea, Republic of)

    2001-09-15

    The association of uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis is rare anomaly. It is caused by failure in the fusion of the caudal portion of the M llerian duct, secondary to Wolffian duct anomaly. We report 4 cases of uterus didelphys with an obstructed hemivagina and ipsilateral renal agenesis. All 4 cases are diagnosed by ultrasonogram, IVU, CT and MRI findings and confirmed by surgery or diagnostic laparoscopy and vaginal examination.

  17. Compensatory adaptations of the remnant kidney to the overload in the chick model of the unilateral renal agenesis

    Czech Academy of Sciences Publication Activity Database

    Zemanová, Zdeňka; Jirsová, Z.; Hubičková, L.; Klusoňová, Petra; Tůma, P.

    Berlin : Freie Universität, 2007. s. 47-47. [Combined workshop on fundamental physiology and perinatal development in poultry /3./. 05.10.2007-07.10.2007, Berlin] R&D Projects: GA ČR(CZ) GA304/04/0972 Institutional research plan: CEZ:AV0Z50110509 Keywords : spr2 * chick embryo * unilateral renal agenesis * compensatory renal agenesis Subject RIV: EA - Cell Biology

  18. Involvement of and interaction between WNT10A and EDA mutations in tooth agenesis cases in the Chinese population.

    Directory of Open Access Journals (Sweden)

    Huiying He

    Full Text Available BACKGROUND: Dental agenesis is the most common, often heritable, developmental anomaly in humans. Although WNT10A gene mutations are known to cause rare syndromes associated with tooth agenesis, including onycho-odontodermal dysplasia (OODD, Schöpf-Schulz-Passarge syndrome (SSPS, hypohidrotic ectodermal dysplasia (HED, and more than half of the cases of isolated oligodontia recently, the genotype-phenotype correlations and the mode of inheritance of WNT10A mutations remain unclear. The phenotypic expression with WNT10A mutations shows a high degree of variability, suggesting that other genes might function with WNT10A in regulating ectodermal organ development. Moreover, the involvement of mutations in other genes, such as EDA, which is also associated with HED and isolated tooth agenesis, is not clear. Therefore, we hypothesized that EDA mutations interact with WNT10A mutations to play a role in tooth agenesis. Additionally, EDA, EDAR, and EDARADD encode signaling molecules in the Eda/Edar/NF-κB signaling pathways, we also checked EDAR and EDARADD in this study. METHODS: WNT10A, EDA, EDAR and EDARADD were sequenced in 88 patients with isolated oligodontia and 26 patients with syndromic tooth agenesis. The structure of two mutated WNT10A and two mutated EDA proteins was analyzed. RESULTS: Digenic mutations of both WNT10A and EDA were identified in 2 of 88 (2.27% isolated oligodontia cases and 4 of 26 (15.38% syndromic tooth agenesis cases. No mutation in EDAR or EDARADD gene was found. CONCLUSIONS: WNT10A and EDA digenic mutations could result in oligodontia and syndromic tooth agenesis in the Chinese population. Moreover, our results will greatly expand the genotypic spectrum of tooth agenesis.

  19. A case of single atrium and single ventricle physiology with bilateral cleft lip and palate for lip repair surgery

    OpenAIRE

    Rakhee Goyal; Ravinder Kumar Batra; Avinash Jangde; Gaurav Kumar

    2012-01-01

    Bilateral cleft lip and palate may occasionally be associated with complex congenital cyanotic heart disease. An infant with common atrium and single ventricle with infundibular pulmonary stenosis (Blalock-Taussig shunt done recently) presented for lip repair surgery. Balanced general anesthesia was administered using sevoflurane along with a regional nerve block to maintain optimal pulmonary and systemic vascular resistance.

  20. Multiple myeloma with pulmonary embolism: a case report

    Institute of Scientific and Technical Information of China (English)

    YING Ke-jing; ZHOU Yong; JIANG Hao; CHEN En-guo; ZHOU Pan

    2006-01-01

    @@ We report a rare case of a patient who died suddenly, in whom bilateral pulmonary artery thrombosis with multiple myeloma was found at autopsy. An estimate of the incidence of pulmonary embolism in myeloma patients based on postmortem examinations is about 3.2%. Hypercoagulability and decreased fibrinolytic capacity due to multiple myeloma were the probable causes of multiple thromboses.

  1. Agenesis of the corpus callosumina premature infant associated with Langdon-Down syndrome

    Directory of Open Access Journals (Sweden)

    Velisavljev-Filipović Gordana

    2005-01-01

    Full Text Available Introduction Agenesis of the corpus callosum is an abnormality of the part of the brain connecting the two cerebral hemispheres. It can be partial, complete or atypical. The fibers from the cerebral cortex project towards the homotypical region of the contra-lateral cortex passing through the corpus callosum, and crossing the middle line. The absence of corpus callosum causes failure of information transfer from one hemisphere to the other. Children with this anomaly present with learning disabilities and trouble with memorizing facts. Agenesis of corpus callosum may be of syndromic or non-syndromic type. The more common form is the one not associated with any syndrome. The agenesis of corpus callosum is more frequent in male children. Case report This paper presents a child from a twin pregnancy with partial absence of corpus callosum. The pregnancy was not controlled. It ended in premature birth. The afflicted twin is a boy, second in birth order. Apart from agenesis of corpus callosum, he also suffers from Down syndrome. The first twin is healthy, with corpus callosum and with normal karyotype. There was no consanguinity. In the 6th month of pregnancy the mother suffered from infection of the upper respiratory tract that might be the etiological factor of this anomaly. The child was born with hypotrophy, and all anthropometric parameters were below the third percentile. In the neonatal period, the agenesis of corpus callosum was diagnosed by ultrasonic examination and confirmed by CT and MR examinations. The child is now three and a half month old. Active monitoring of the psychical and motor development will show whether there will be any retardation in the psycho-motor development and later deficiency of the higher cortical functions and intelligence. Discussion and Conclusion The clinical characteristics of this anomaly are numerous. They range from asymptomatic cases, with normal intellectual capacity, to severe mental retardation

  2. Bilateral Testicular Epidermoid Cysts

    OpenAIRE

    Norman Loberant; Shweta Bhatt; Edward Messing; Dogra, Vikram S.

    2011-01-01

    Testicular epidermoid cysts are the most common benign tumors of the testes, but account for only 1-2% of all testicular tumors. In a young man presenting with a testicular mass, a high index of suspicion must be maintained for the malignant testicular germ cell tumor, which is 50-times more common than testicular epidermoid cyst. Bilateral testicular epidermoid cysts are a very rare condition, with only a few reports in the literature. It is extremely important in this condition to make a co...

  3. Pulmonary Rehabilitation

    Science.gov (United States)

    ... programs are certified by the American Association of Cardiovascular and Pulmonary Rehabilitation. These programs can be found on the AACVPR ... at 1-800-586-4872. American Association of Cardiovascular and Pulmonary Rehabilitation ... ...

  4. Pulmonary Fibrosis

    Science.gov (United States)

    Pulmonary fibrosis is a condition in which the tissue deep in your lungs becomes scarred over time. This tissue ... may not get enough oxygen. Causes of pulmonary fibrosis include environmental pollutants, some medicines, some connective tissue ...

  5. Pulmonary edema

    Science.gov (United States)

    Pulmonary edema is an abnormal buildup of fluid in the lungs. This buildup of fluid leads to shortness of ... Pulmonary edema is often caused by congestive heart failure . When the heart is not able to pump efficiently, blood ...

  6. Bilateral matrix-exponential distributions

    DEFF Research Database (Denmark)

    Bladt, Mogens; Esparza, Luz Judith R; Nielsen, Bo Friis

    2012-01-01

    In this article we define the classes of bilateral and multivariate bilateral matrix-exponential distributions. These distributions have support on the entire real space and have rational moment-generating functions. These distributions extend the class of bilateral phasetype distributions of [1......] and the class of multivariate matrix-exponential distributions of [9]. We prove a characterization theorem stating that a random variable has a bilateral multivariate distribution if and only if all linear combinations of the coordinates have a univariate bilateral matrix-exponential distribution. As...... an application we demonstrate that certain multivariate disions, which are governed by the underlying Markov jump process generating a phasetype distribution, have a bilateral matrix-exponential distribution at the time of absorption, see also [4]....

  7. Pulmonary rehabilitation

    OpenAIRE

    Spruit, Martijn A.

    2001-01-01

    Pulmonary rehabilitation is a therapy that offers benefits to patients with chronic obstructive pulmonary disease that are complementary to those obtained by pharmacotherapy. The main objective of pulmonary rehabilitation is to restore muscle function and exercise tolerance, reverse other nonrespiratory consequences of the disease, and help patients to self-manage chronic obstructive pulmonary disease and its exacerbations and symptoms. To do so, a multidisciplinary program tailored to the pa...

  8. Pulmonary Embolism.

    Science.gov (United States)

    Rali, Parth; Gandhi, Viral; Malik, Khalid

    2016-01-01

    Pulmonary embolism covers a wide spectrum of presentation from an asymptomatic individual to a life-threatening medical emergency. It is of paramount importance to appropriately risk stratify patients with pulmonary embolism, particularly with those who present without hypotension. Right ventricular dysfunction can evolve after a patient has received a diagnosis of pulmonary embolism, necessitating aggressive measures rather than simple anticoagulation. In this review, we discuss definition, risk stratification, pathogenesis, diagnostic approach, and management, with particular focus on massive pulmonary embolism. PMID:26919674

  9. Achondroplasia Associated with Bilateral Keratoconus

    OpenAIRE

    Samar A. Al-Swailem; Al Bin Ali, Ghada Y.; Al Katan, Hind M.; Al Mahmood, Ammar M.

    2012-01-01

    We report a rare case of bilateral keratoconus in association with achondroplasia. A 26-year-old male, with a known case of achondroplasia, complained of bilateral gradual deterioration in vision for the past few years. Slit lamp biomicroscopy showed bilateral central corneal protrusion and stromal thinning at the apex consistent with keratoconus. a trial of hard contact lens fitting failed to improve VA in the left eye (LE). Right eye (RE) improved to 20/25. The patient underwent penetrating...

  10. Learn About Pulmonary Fibrosis

    Science.gov (United States)

    ... www.lung.org > Lung Health and Diseases > Lung Disease Lookup > Pulmonary Fibrosis Learn About Pulmonary Fibrosis Pulmonary fibrosis is ... while processing XML file."); } }); } } --> Blank Section Header Lung Disease Lookup Pulmonary Fibrosis Learn About Pulmonary Fibrosis Pulmonary Fibrosis Symptoms, ...

  11. The combination of thermal dysregulation and agenesis of corpus callosum: Shapiro′s or/and reverse Shapiro′s syndrome

    Directory of Open Access Journals (Sweden)

    Yasemin Topcu

    2013-01-01

    Full Text Available Shapiro syndrome is an extremely rare condition consisting the clinical triad of recurrent hypothermia, hyperhydrosis and agenesis of the corpus callosum. On the other hand, reverse Shapiro′s sydrome is characterized periodic hyperthermia and agenesis of the corpus callosum. Here, we describe a 3.5-year-old girl with complete agenesis of corpus callosum presenting with recurrent fever and vomiting. She also had hypothermia attacks with accompanying diaphoresis. To the best of our knowledge, there is no described case with episodes of hyperthermia, hypothermia, and vomiting associated with agenesis of the corpus callosum. Recurrent vomiting may be a newly defined symptom associated with these syndromes.

  12. A rare case of Turner′s syndrome presenting with Mullerian agenesis

    Directory of Open Access Journals (Sweden)

    Suresh Vaddadi

    2013-01-01

    Full Text Available Turner′s syndrome also called as Ullrich Turner′s syndrome, is a disease of unclear pathogenesis characterized by complete or partial absence of one sex chromosome, with or without cell line mosaicism in a phenotypic female with short stature. Various anomalies result in a constellation of features, of which the most disturbing is primary amenorrhea due to gonadal dysgenesis. Hormone therapy in these patients can often result in successful menstruation, and scope for subsequent pregnancy because of anatomically normal uterus and vagina. Coexisting Mullerian agenesis in these patients can jeopardize the chances of future pregnancy as they have associated structural abnormalities of the uterus and vagina. We report a rare case of middle-aged female with Turner′s syndrome and Mullerian agenesis having absent secondary sexual characters and missing uterus with incompletely formed vagina.

  13. Segmental aplasia of the uterine horn with ipsilateral renal agenesis in a cat.

    Science.gov (United States)

    Chang, Jinhwa; Jung, Joo-hyun; Yoon, Junghee; Choi, Min-cheol; Park, Jae Hak; Seo, Kang-Moon; Jeong, Seong Mok

    2008-06-01

    A nine-month-old domestic short haired cat was admitted with the history of acute vomiting, depression and shivering. Abdominal ultrasonography revealed minimum enlargement of the right uterine horn filled with anechoic fluid. On excretory urography, functionally and anatomically normal, enlarged left kidney was found, but right kidney was absent. It was preliminary diagnosed as hydrometra with right renal agenesis. Aiming at the correction of hydrometra, we performed ovariohysterectomy. During spaying, we found a missing segment of distal part of the right uterine horn and absence of ipsilateral kidney and ureter. Compressed uterine structure and segmental aplasia of right uterine horn were found in histopathological investigation. Taken together, it was diagnosed as a segmental aplasia of uterine horn with ipsilateral renal agenesis. PMID:18628611

  14. A case of corpus callosum agenesis presenting with recurrent brief depression

    Directory of Open Access Journals (Sweden)

    Bhattacharyya Ranjan

    2009-01-01

    Full Text Available Agenesis of corpus callosum can have various neuropsychiatric manifestations. Following case report highlights the case of a young man presenting with features of recurrent brief depressive disorder, each lasting for about 3 to 7 days, for over a year. He had history of occasional headache and episodes of swooning attack in between, usually precipitated by emotional events. His neuroimaging revealed agenesis of corpus callosum. He was experiencing swooning attacks as he became aware that some ′unusual′ findings were present in his reports. Recurrent brief depression can be a manifestation of this congenital anomaly, and conversion disorder can be present as comorbid diagnosis perhaps due to ignorance and fear of this apparently innocuous congenital malformation.

  15. Changes in bilateral bispectral index VISTA monitoring system during Wada test.

    Science.gov (United States)

    Pacreu, S; Vila, E; Rodríguez, C; Arroyo, R; Fernández, S; Fernández, J L

    2014-12-01

    The Wada test is a procedure used in the preoperative assessment before epilepsy surgery in order to determine language lateralization, to assess the post-operative risk of an amnesia syndrome, and to evaluate the risk of material-specific memory deficits, in particular verbal memory deficits. This test involves inserting a cannula into the internal carotid artery via the femoral artery, and then to inject amobarbital to shut down brain function, usually in one of the brain hemispheres. The bilateral bispectral index (BIS) VISTA™ monitoring system (BVMS) was used to detect changes in EEG, and in the power spectrum distribution using the density spectral array (DSA) of both hemispheres. We describe a patient with an agenesis of the A1 segment of the right anterior cerebral artery, scheduled for a Wada test, in whom the BVMS demonstrated its potential value. PMID:24657004

  16. Uterus didelphys with partial vaginal septum and distal vaginal agenesis: an unusual anomaly

    International Nuclear Information System (INIS)

    Complex malformations of female genital tract are not so common. Their correct identification is of paramount importance for appropriate management. A thorough knowledge of embryology, pre-operative imaging with MRI and examination under anaesthesia is essential to identify accurately the constellation of anomalies and to plan appropriate management. This case reports the coexistence of Mullerian abnormality with vaginal agenesis in an 18 years old girl which was managed by dissecting the lower half of vagina and pull-through vaginoplasty. (author)

  17. Uterus didelphys with unilateral obstructed hemivagina with hematometrocolpos and hematosalpinx with ipsilateral renal agenesis

    OpenAIRE

    Gaurav Jindal; Satish Kachhawa; G L Meena; Gopal Dhakar

    2009-01-01

    Uterus didelphys with blind hemivagina and ipsilateral renal agenesis (Herlyn Werner-Wunderlich Syndrome) is a rare congenital anomaly. It mostly presents with severe dysmenorrhea and a palpable mass due to unilateral hematocolpos. A patient with dysmenorrhea from a double uterus and an obstructed hemivagina is a diagnostic dilemma because the menses are regular. We report a case of a 14-year-old girl with this condition who was diagnosed as uterus didelphys with unilateral hematocolpos and h...

  18. Uterus Didelphys with Obstructed Right Hemivagina, Ipsilateral Renal Agenesis and Right Pyocolpos: A Case Report

    OpenAIRE

    Dhar, Hansa; Yasser A Razek; Hamdi, Ilham

    2011-01-01

    Uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis (OHVIRA Syndrome) is a rare congenital anomaly of the female genital tract. Uterus didelphys occurs when the midline fusion of the mullerian ducts is arrested, either completely or incompletely. Women with didelphic uterus may be asymptomatic and unaware of having a double uterus. They may present with complaints of dysmenorrhoea and dyspareunia. A 25 year old woman attending the infertility clinic at Nizwa regional re...

  19. Uterus didelphys with an obstructed unilateral vagina and ipsilateral renal agenesis: A rare cause of dysmenorrhoea

    OpenAIRE

    ATTAR, Rukset; Yıldırım, Gazi; Inan, Yücel; Küzılkale, Özge; Karateke, Ateş

    2013-01-01

    Didelphic uterus with obstructed hemivagina and ipsilateral renal agenesis is a rare condition. It usually presents with pelvic pain following the menarche, dysmenorrhoea, and an increase in abdominal volume or a palpable mass due to unilateral haematocolpos. We present the case of a 13-year-old girl who referred with recurrent pelvic pain, mainly at the time of menses, and irregular menstrual cycle complaints in this report. The patient underwent ultrasonography and magnetic resonance (MR) i...

  20. OHVIRA: Uterus didelphys, blind hemivagina and ipsilateral renal agenesis: Advantage MRI

    OpenAIRE

    Bajaj, Sunil K.; Ritu Misra; Thukral, Brij B; Rohini Gupta

    2012-01-01

    We present here a case of an uncommon complex uterine anomaly - Obstructed HemiVagina with Ipsilateral Renal Agenesis (OHVIRA), also known as Herlyn-Werner-Wunderlich syndrome in a 14-year-old girl along with sonographic (trans-abdominal and trans labial), and MRI findings. The patient underwent surgery wherein imaging findings were confirmed. An MRI has proved to be of great help in correct diagnosis avoiding surgical interventions/ laparoscopy, which were needed in past to diagnose this rar...

  1. The Structural Connectome of the Human Brain in Agenesis of the Corpus Callosum

    OpenAIRE

    Owen, Julia P.; Li, Yi-Ou; Ziv, Etay; Strominger, Zoe; Gold, Jacquelyn; Bukhpun, Polina; Wakahiro, Mari; Friedman, Eric J.; Sherr, Elliott H; Mukherjee, Pratik

    2012-01-01

    Adopting a network perspective, the structural connectome reveals the large-scale white matter connectivity of the human brain, yielding insights into cerebral organization otherwise inaccessible to researchers and clinicians. Connectomics has great potential for elucidating abnormal connectivity in congenital brain malformations, especially axonal pathfinding disorders. Agenesis of the corpus callosum (AgCC) is one of the most common brain malformations and can also be considered a prototypi...

  2. Sacral Agenesis with Neurogenic Bladder Dysfunction—A Case Report and Review of the Literature

    OpenAIRE

    Sharma, Seema; Sharma, Vipin; Awasthi, Bhanu; Sehgal, Manik; Deeksha A Singla

    2015-01-01

    Sacral agenesis (part of the caudal regression syndrome) is a rare and severe sacral developmental abnormality. It is a congenital malformation of unknown aetiology with possible involvement of genetic and teratogenic factors. It is described by various degrees of developmental failure, the most extreme and rare being sirenomelia or mermaid syndrome. The associated malformations comprise anorectal, vertebral, urological, genital, and lower limb anomalies. Approximately 15-20% mothers of these...

  3. Unilateral renal agenesis: case review of ambulatory pediatric nephrology clinics in Cali

    Directory of Open Access Journals (Sweden)

    Consuelo Restrepo de Rovetto

    2010-03-01

    Full Text Available Introduction: Unilateral renal agenesis is a frequent renal malformation with incidence of 1 per 1000 live born children. There are no statistics nor protocols to manage and follow-up these patients. Usually asymptomatic, it can be early detected by prenatal ultrasound, allowing opportune detection and adequate follow up. The goal is to describe main features found in children with renal agenesis at four ambulatory pediatric nephrology clinics in Cali, Colombia: Hospital Universitario del Valle (HUV, Club Noel Children’s hospital and the private practice of two pediatric nephrologists.Methods: This is a retrospective descriptive study in a series of cases with congenital renal agenesis diagnosed between January, 1995 and December 2007, of patients under 18 years of age and based on a review of clinical records.Results: 43 patients were found, 51.2% males, 88% from the department of Valle del Cauca. Prenatal diagnoses were conducted in only 21% of these patients; agenesis was right in 48.8% and left in 51.2%. In 46.5%, association with other pathologies was found: occult spinal bifida, congenital scoliosis, and Klippel-Feil sequence. Compensatory renal hypertrophy was reported in 39.5% of the patients, 42% had episodes of urinary tract infection, 31% vesicoureteral reflux and 10% proteinuria. Four male patients (9.3% developed renal failure. The average follow-up was 6.6 years.Conclusions: Prenatal diagnose was not very frequent in our set of cases. Urinary tract infection and vesicoureteral reflux were common, increasing the risk of renal deterioration. Prolonged follow up is suggested, with unified protocols to prevent renal failure.

  4. Unilateral renal agenesis: case review of ambulatory pediatric nephrology clinics in Cali

    Directory of Open Access Journals (Sweden)

    Consuelo Restrepo de Rovetto

    2010-09-01

    Full Text Available Introduction: Unilateral renal agenesis is a frequent renal malformation with incidence of 1 per 1000 live born children. There are no statistics nor protocols to manage and follow-up these patients. Usually asymptomatic, it can be early detected by prenatal ultrasound, allowing opportune detection and adequate follow up. The goal is to describe main features found in children with renal agenesis at four ambulatory pediatric nephrology clinics in Cali, Colombia: Hospital Universitario del Valle (HUV, Club Noel Children’s hospital and the private practice of two pediatric nephrologists. Methods: This is a retrospective descriptive study in a series of cases with congenital renal agenesis diagnosed between January, 1995 and December 2007, of patients under 18 years of age and based on a review of clinical records. Results: 43 patients were found, 51.2% males, 88% from the department of Valle del Cauca. Prenatal diagnoses were conducted in only 21% of these patients; agenesis was right in 48.8% and left in 51.2%. In 46.5%, association with other pathologies was found: occult spinal bifida, congenital scoliosis, and Klippel-Feil sequence. Compensatory renal hypertrophy was reported in 39.5% of the patients, 42% had episodes of urinary tract infection, 31% vesicoureteral reflux and 10% proteinuria. Four male patients (9.3% developed renal failure. The average follow-up was 6.6 years. Conclusions: Prenatal diagnose was not very frequent in our set of cases. Urinary tract infection and vesicoureteral reflux were common, increasing the risk of renal deterioration. Prolonged follow up is suggested, with unified protocols to prevent renal failure.

  5. Agenesis of the corpus callosumina premature infant associated with Langdon-Down syndrome

    OpenAIRE

    Velisavljev-Filipović Gordana

    2005-01-01

    Introduction Agenesis of the corpus callosum is an abnormality of the part of the brain connecting the two cerebral hemispheres. It can be partial, complete or atypical. The fibers from the cerebral cortex project towards the homotypical region of the contra-lateral cortex passing through the corpus callosum, and crossing the middle line. The absence of corpus callosum causes failure of information transfer from one hemisphere to the other. Children with this anomaly present with learning dis...

  6. Agenesis of Ductus Venosus Presenting with Dilated Inferior Vena Cava with Favorable Outcome

    OpenAIRE

    Omer Dai; Harun Egemen Tolunay; Mehmet Murat Seval; Alper Kahraman; Korhan Kahraman; Acar Koç

    2014-01-01

    The ductus venosus regulates the distribution of oxygen and placental nutrients by restricting the centralization of blood flow in fetal circulation. The ductus venosus is a small vein transmitting oxygen-rich blood from the umbilical vein to the fetal heart.. Increasing numbers of case reports are being published about ductus venosus agenesis with cardiac structural abnormalities, other malformations, chromosomal abnormalities, and stillbirth. Heart failure, hydrops and unexplained polyhydra...

  7. Agenesis of the internal carotid artery with a large hemangioma of the tongue

    International Nuclear Information System (INIS)

    Total developmental absence of the internal carotid artery is relatively rare, with only 54 cases previously reported. Most, being without neurological symptoms, were found by chance. For this patient with cavernous hemangiomas in the facial, oral and cervical regions, conventional treatment would be embolization of the feeding vessels, combined with ligation of the external carotid artery. Angiography, however, revealed agenesis of the left internal carotid artery, abnormal origin of the aortic arch and azygos anterior cerebral artery. (orig./MG)

  8. 5q11.2 deletion in a patient with tracheal agenesis

    OpenAIRE

    de Jong, Elisabeth; Douben, Hannie; Eussen, Bert; Felix, Janine; Wessels, Marja; Poddighe, Pino; Nikkels, Peter; De Krijger, Ronald; Tibboel, Dick; De Klein, Annelies

    2010-01-01

    textabstractTracheal agenesis (TA) is a rare congenital anomaly of the respiratory tract. Many patients have associated anomalies, suggesting a syndromal phenotype. In a cohort of 12 patients, we aimed to detect copy number variations. In addition to routine cytogenetic analysis, we applied oligonucleotide array comparative genomic hybridization. Our patient cohort showed various copy number variations, of which many were parentally inherited variants. One patient had, in addition to an inher...

  9. Pancreatic Agenesis with Congenital Diaphragmatic Hernia and Congenital Heart Disease: A Case Report

    OpenAIRE

    Atsushi Nakao; Tomohiro Takeda; Yoshiya Hisaeda; Atsushi Hirota; Syusuke Amagata; Yuko Sakurai; Tadashi Kawakami

    2013-01-01

    Pancreatic agenesis is a rare disease that causes neonatal diabetes mellitus and exocrine pancreatic insufficiency. We report the case of a very low birth weight infant with congenital diaphragmatic hernia, and congenital heart disease (ventricular septal defect and patent ductus arteriosus). Failure to gain weight, despite well-managed respiratory and heart failure, was improved by infusion of subcutaneous insulin, administration of pancreatic enzyme, and nutrition of medium-chain-triglyceri...

  10. Uterus Didelphys with Obstructed Hemivagina and Ipsilateral Renal Agenesis: The Radiologic Findings of Five Cases

    International Nuclear Information System (INIS)

    To evaluate the imaging findings of uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis. From March 2003 to December 2008, five patients with uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis were evaluated as part of this study. We retrospectively reviewed the CT, ultrasound, and MRI findings as well as the medical records of each patient. The patients initially underwent an imaging study for abdominal pain (n=3), recurrent vaginal bleeding (n=1), and prenatal evaluation (n=1). Of the five patients that underwent US, four had hematocolpos and two of them had hematometra. Moreover, three patients underwent a CT examination. The MR examination of four patients revealed hematocolpos (n=3), hematometra (n=1), and a tubular structure resembling an ectopic ureter (n=2). The gynecologic examination of a patient without hematocolpos revealed a pinpoint hole in the vaginal septum. Two of four patients with hematocolpos underwent a vaginal septectomy, which resulted in an improvement of the symptoms. The most common finding of patients with uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis is vaginal fluid collection. Hematometra is not a consistent finding and can be transient according to the menstrual cycle. MR is the most useful imaging modality for the diagnosis of an ectopic ureter

  11. Uterus Didelphys with Obstructed Hemivagina and Ipsilateral Renal Agenesis: The Radiologic Findings of Five Cases

    Energy Technology Data Exchange (ETDEWEB)

    Rhee, Sun Jung; Yi, Boem Ha; Park, Seong Jin; Lee, Hae Kyung; Lee, Kwon Hae; Lee, Hae Hyeog; Kim, Tae Hee [Soonchunhyang University Bucheon Hospital, Bucheon (Korea, Republic of)

    2010-08-15

    To evaluate the imaging findings of uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis. From March 2003 to December 2008, five patients with uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis were evaluated as part of this study. We retrospectively reviewed the CT, ultrasound, and MRI findings as well as the medical records of each patient. The patients initially underwent an imaging study for abdominal pain (n=3), recurrent vaginal bleeding (n=1), and prenatal evaluation (n=1). Of the five patients that underwent US, four had hematocolpos and two of them had hematometra. Moreover, three patients underwent a CT examination. The MR examination of four patients revealed hematocolpos (n=3), hematometra (n=1), and a tubular structure resembling an ectopic ureter (n=2). The gynecologic examination of a patient without hematocolpos revealed a pinpoint hole in the vaginal septum. Two of four patients with hematocolpos underwent a vaginal septectomy, which resulted in an improvement of the symptoms. The most common finding of patients with uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis is vaginal fluid collection. Hematometra is not a consistent finding and can be transient according to the menstrual cycle. MR is the most useful imaging modality for the diagnosis of an ectopic ureter.

  12. Cerebellar contributions to self-motion perception: evidence from patients with congenital cerebellar agenesis.

    Science.gov (United States)

    Dahlem, Kilian; Valko, Yulia; Schmahmann, Jeremy D; Lewis, Richard F

    2016-05-01

    The cerebellum was historically considered a brain region dedicated to motor control, but it has become clear that it also contributes to sensory processing, particularly when sensory discrimination is required. Prior work, for example, has demonstrated a cerebellar contribution to sensory discrimination in the visual and auditory systems. The cerebellum also receives extensive inputs from the motion and gravity sensors in the vestibular labyrinth, but its role in the perception of head motion and orientation has received little attention. Drawing on the lesion-deficit approach to understanding brain function, we evaluated the contributions of the cerebellum to head motion perception by measuring perceptual thresholds in two subjects with congenital agenesis of the cerebellum. We used a set of passive motion paradigms that activated the semicircular canals or otolith organs in isolation or combination, and compared results of the agenesis patients with healthy control subjects. Perceptual thresholds for head motion were elevated in the agenesis subjects for all motion protocols, most prominently for paradigms that only activated otolith inputs. These results demonstrate that the cerebellum increases the sensitivity of the brain to the motion and orientation signals provided by the labyrinth during passive head movements. PMID:26888100

  13. Pediatric neurofunctional intervention in agenesis of the corpus callosum: a case report☆

    Science.gov (United States)

    Pacheco, Sheila Cristina da Silva; Queiroz, Ana Paula Adriano; Niza, Nathália Tiepo; da Costa, Letícia Miranda Resende; Ries, Lilian Gerdi Kittel

    2014-01-01

    Objective: To describe a clinical report pre- and post-neurofunctional intervention in a case of agenesis of the corpus callosum. Case description: Preterm infant with corpus callosum agenesis and hypoplasia of the cerebellum vermis and lateral ventricles, who, at the age of two years, started the proposed intervention. Functional performance tests were used such as the neurofunctional evaluation, the Gross Motor Function Measure and the Gross Motor Function Classification System. In the initial evaluation, absence of equilibrium reactions, postural transfers, deficits in manual and trunk control were observed. The intervention was conducted with a focus on function, prioritizing postural control and guidance of the family to continue care in the home environment. After the intervention, there was an improvement of body reactions, postural control and movement acquisition of hands and limbs. The intervention also showed improvement in functional performance. Comments: Postural control and transfers of positions were benefited by the neurofunction intervention in this case of agenesis of the corpus callosum. The approach based on function with activities that involve muscle strengthening and balance reactions training, influenced the acquisition of a more selective motor behavior. PMID:25479858

  14. Effect of Vestibulo-Proprioceptive Stimulations in a Child with Agenesis of the Corpus Callosum

    Directory of Open Access Journals (Sweden)

    Hamid Dalvand

    2010-06-01

    Full Text Available Background and Aim: The purpose of the present study was to investigate the effect of vestibulo-proprioceptive stimulations of sensory integration theory on the development of gross and fine motor, language and personal-social functions in a child with agenesis of the corpus callosum.Case: We report a 10.5 month old boy with agenesis of the corpus callosum. The intervention was administered based on sensory integration theory an hour a week for 20 weeks. The exercise intervention consisted of proprioceptive and linear, sustained and low frequency vestibular stimulations on suspension device and physio roll. A Denver Developmental Screening- II and milestones skill testing was completed pre-intervention and monthly. Post-intervention, age of gross motor, fine motor adaptive, language, and personal-social functions significantly improved. Based on milestones skills, maintenance of gross motor functions (e.g. sitting and quadruped position improved. The child could roll from side to side and released objects voluntarily. The reaction time to auditory stimulations became less than 2 seconds.Conclusion: vestibulo-proprioceptive stimulations using the neuroplasticity ability of the central nervous system is effective for development of gross and fine motor, language, and personal-social functions. These exercises can be administered for a child with agenesis of the corpus callosum.

  15. Bilateral Antepartum Mastitis

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    Peyman Alibeigi

    2010-12-01

    Full Text Available Antepartum mastitis is a rare condition, whereas postpartum orlactation mastitis is a common problem. This report introducesa case of complicated bilateral antepartum mastitis, which wastreated successfully by drain insertion and antibiotic therapy.The patient was a 23-year-old woman in the 23rd week of herfirst pregnancy. Her chief complaint was progressive swelling,redness and radicular pain in both breasts, which had beenstarted gradually from the 18th week of pregnancy. The patientwas admitted to hospital, and received oral and intravenous antibioticsempirically, which was not effective. The patient wastreated by drainage and oral antibiotic therapy. Based on theapproaches employed and the outcomes achieved it is suggestedthat early surgical insertion in the presence of fluid collection inantepartum mastitis will shorten hospitalization and course ofintravenous antibiotic therapy.Iran J Med Sci 2010; 35(4: 327-330.

  16. Idiopathic Bilateral Bloody Tearing

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    Emrullah Beyazyıldız

    2015-01-01

    Full Text Available Bloody tear is a rare and distinct clinic phenomenon. We report a case presenting with the complaint of recurrent episodes of bilateral bloody tearing. A 16-year-old girl presented to our clinic with complaint of bloody tearing in both eyes for 3 months. Bloody tearing was not associated with her menses. A blood-stained discharge from the punctum was not observed during the compression of both nasolacrimal ducts. Nasolacrimal passage was not obstructed. Imaging studies such as dacryocystography and gradient-echo magnetic resonance imaging (MRI of nasolacrimal canal were normal. Intranasal endoscopic evaluation was normal. We collected samples from bloody tears two times and pathological examination was performed. Pathological analysis showed lots of squamous cells and no endometrial cells; dysplastic cells were found. Further evaluations for underlying causes were unremarkable. No abnormalities were found in ophthalmologic, radiologic, and pathologic investigations. This condition is likely a rare abnormality and the least recognized aetiology for the idiopathic phenomenon.

  17. Isolated bilateral ectopic microspherophakia

    Directory of Open Access Journals (Sweden)

    André Omgbwa Eballe

    2010-02-01

    Full Text Available André Omgbwa Eballe1, Godefroy Koki2, Emilienne Epée2, Didier Owono2, Lucienne Bella Asumpta2, Côme Ebana Mvogo21Faculty of Medicine and Pharmaceuticals Sciences, University of Douala Cameroon; 2Faculty of Medicine and Biomedical Sciences, University of Yaoundé CameroonSummary: An eight-year-old school girl presented to us with a bilateral ectopic microspherophakia. She was surgically managed in the ophthalmology unit of the Gynaeco-Obstetric and Pediatric Hospital of Yaoundé in Cameroon. The postoperative follow up was uneventful, and the patient returned to school with a best corrected visual acuity of 4/10 in both eyes to the great satisfaction of her parents.Keywords: ectopic, microspherophakia, surgery

  18. Silicosis with bilateral spontaneous pneumothorax

    OpenAIRE

    Fotedar Sanjay; Chaudhary Dhruva; Singhla Vikas; Narang Rajat

    2010-01-01

    Presentation with simultaneous bilateral pneumothorax is uncommon and usually in the context of secondary spontaneous pneumothorax. The association of pneumothorax and silicosis is infrequent and most cases are unilateral. Bilateral pneumothorax in silicosis is very rare with just a few reports in medical literature.

  19. Silicosis with bilateral spontaneous pneumothorax

    Directory of Open Access Journals (Sweden)

    Fotedar Sanjay

    2010-01-01

    Full Text Available Presentation with simultaneous bilateral pneumothorax is uncommon and usually in the context of secondary spontaneous pneumothorax.The association of pneumothorax and silicosis is infrequent and most cases are unilateral. Bilateral pneumothorax in silicosis is very rare with just a few reports in medical literature.

  20. Unusual ventilation perfusion scintigram in a case of immunologic pulmonary edema clinically simulating pulmonary embolism

    International Nuclear Information System (INIS)

    A case of immunologic pulmonary edema secondary to hydrochlorothiazide allergy developed in a 55-year-old woman that clinically simulated pulmonary embolism. The patient had abnormal washin images with normal washout images on an Xe-133 ventilation study. On the perfusion study, large bilateral central and posterior perfusion defects were present that showed an unusual mirror image pattern on the lateral and posterior oblique views. Resolution of radiographic and scintigraphic abnormalities occurred over a 3-day period in conjunction with corticosteroid therapy

  1. Bilateral, independent juvenile nasopharyngeal angiofibroma

    DEFF Research Database (Denmark)

    Mørkenborg, Marie-Louise; Frendø, M; Stavngaard, T;

    2015-01-01

    BACKGROUND: Juvenile nasopharyngeal angiofibroma is a benign, vascular tumour that primarily occurs in adolescent males. Despite its benign nature, aggressive growth patterns can cause potential life-threatening complications. Juvenile nasopharyngeal angiofibroma is normally unilateral, originati...... with bilateral symptoms. Our management, including successful pre-operative planning, enabled one-step total removal of both tumours and rapid patient recovery....... from the sphenopalatine artery, but bilateral symptoms can occur if a large tumour extends to the contralateral side of the nasopharynx. This paper presents the first reported case of true bilateral extensive juvenile nasopharyngeal angiofibroma involving clinically challenging pre-surgical planning...... bilateral embolisation. Radical removal performed as one-step, computer-assisted functional endoscopic sinus surgery was performed. The follow-up period was uncomplicated. CONCLUSION: This case illustrates the importance of suspecting bilateral juvenile nasopharyngeal angiofibroma in patients presenting...

  2. Pulmonary alveolar microlithiasis

    International Nuclear Information System (INIS)

    Pulmonary alveolar microlithiasis (PAM) is a rare disease characterized by the diffuse and bilateral presence of calcium phosphate microlite in the alveolar spaces. The progression of this potentially lethal disease is show and most of the patients remain asymptomatic during years or decades, resulting in a show deterioration of the pulmonary function. The typical finding of the sand storm in the chest X-ray is characteristic of this entity. Mutations in the SLC34A2 gene that does the coding for the type II co-transporter of sodium phosphate were identified as responsible for this disease. Of the almost 600 cases, only 6 have been reported in Colombia. We are presenting a case of pulmonary alveolar microlite in a 27 year old man, with progressive respiratory distress whose diagnosis was made by the X-ray findings and confirmed by trans bronchial biopsy. In the 2 years follow-up, shows evolution towards deterioration of his respiratory function making him a candidate for lung transplantation.

  3. Agenesis of third molars in students from the Dentistry Academic Program of Universidad del Valle, 16 to 25 years old

    Directory of Open Access Journals (Sweden)

    Sarmiento Pedro

    2004-09-01

    Full Text Available Background: Third molars are the teeth reported in the literature, that present more variability in number, shape and size. There is no information reported in the Colombian literature concerning the agenesis of the third molar in Colombians. This data may changes constantly from one population to another. This study was looking for agenesis of the third molars and performed on dental students at the Universidad del Valle. Objectives: Determine dental agenesis of the third molars and describe characteristics such as gender, arcade, and dental quadrants. Method: A descriptive study using 1824 periapical radiographs of 456 people aged 16 to 25 years old. Results: The results showed that 96 students didn’t developed some or none of third molars, which corresponds to 21% agenesis of the studied population. No significant statistical differences were found on gender, in the mandible and maxilla, neither in the left and right sides Conclusion: The percentage of agenesis in this study is consistent with most of the reports in the literature, in populations from different parts of the world.

  4. Acute silicosis with bilateral pneumothorax.

    Science.gov (United States)

    Srivastava, G N; Prasad, Rajniti; Meena, Manoj; Hussain, Moosa

    2014-01-01

    We present a case of acute silicosis with bilateral pneumothorax of a 28-year-old man working at a stone crusher factory for 1 year. He presented to the emergency department with cough, respiratory distress and diffuse chest pain. The patient was managed with bilateral intercostal tube drainage under water seal, oxygen inhalation and conservative therapy. On follow-up he showed improvement of resting dyspnoea and was doing well. This case is being reported because of the rare complications of acute silicosis as bilateral pneumothorax. PMID:24862410

  5. [A paradigmatic case of partial agenesis of the pericardium].

    Science.gov (United States)

    Scapellato, F; Imparato, A; Brunetto, A; Temporelli, P L; Giannuzzi, P

    1996-10-01

    Complete or partial absence of the pericardium is an uncommon congenital abnormality. Since its first description, several cases have been reported. Although the clinical and instrumental features of this defect are well described, at present it is often overlooked. We report a case of a 19-year old boy referred to our Echocardiography laboratory because of a suspected interatrial septal defect. For this reason it has been recommended to undergo cardiac catheterization. The patient, completely asymptomatic, had a negative physical examination. The echocardiogram excluded the presence of an interatrial septal defect, but some abnormalities, regarding overall right-sided heart, were found (paradoxical interventricular septal movement, apparent right-sided heart enlargement, with unusual bulging of the apex of the right ventricle, an excessive basculant heart movement). In the absence of any other heart disease, we thought that these abnormalities reflected a congenital absence of the pericardium. Indeed, the chest X-ray showed some features, considered patognomonic for congenital absence of the pericardium (levoposition of the heart, lung interposition between the diaphragm and the base of the heart and between the aorta and pulmonary artery). Nuclear magnetic resonance study showed the absence of the pericardium in the posterior and posterolateral wall, confirming our suspicion. Furthermore, the posterior wall of the heart leaned to herniate through the pericardial defect. Since the partial absence of the pericardium can lead to severe complications, surgery was recommended. The correct diagnosis of this disease is very important because of its prognostic implications; this case represents a further contribution to the understanding of this defect. PMID:9005163

  6. Living with Pulmonary Fibrosis

    Science.gov (United States)

    ... www.lung.org > Lung Health and Diseases > Lung Disease Lookup > Pulmonary Fibrosis Living With Pulmonary Fibrosis What to Expect ... while processing XML file."); } }); } } --> Blank Section Header Lung Disease Lookup Pulmonary Fibrosis Learn About Pulmonary Fibrosis Pulmonary Fibrosis Symptoms, ...

  7. Prune belly syndrome associated with bilateral multicystic dysplastic kidneys and urethral obstruction: A case report

    Directory of Open Access Journals (Sweden)

    Arzu Akdag

    2015-06-01

    Full Text Available Prune belly syndrome is a rare congenital disorder defined by a characteristic clinical triad: Abdominal muscle deficiency, severe urinary tract abnormalities, and bilateral cryptorchidism. We describe a preterm neonate of Prune Belly syndrome who had abdominal muscle deficiency, multicystic dysplastic kidney, urethral hypoplasia and pulmonary hypoplasia. We presented this rare case with the data gathered from the literatüre.

  8. "Recurrent Bilateral Spontaneous Pneumothorax in Early Infancy: A Case of Langerhans Cell Histiocytosis"

    OpenAIRE

    S. Alavi; N. Hemmati; M.T. Arzanian; Ashena, Z

    2005-01-01

    Langerhans cell histiocytosis (LCH) is a rare disorder characterized by infiltration of either single or multiple organs by S100 and CD1a positive cells. Patients with pulmonary LCH are predisposed to pneumothorax due to destructive changes in the lung parenchyma. Here, we report a case of multisystem LCH who presented at 2 months of age with simultaneous bilateral spontaneous pneumothorax.

  9. MURCS (Müllerian duct aplasia-renal agenesis-cervicothoracic somite dysplasia): a rare cause of primary amenorrhoea.

    Science.gov (United States)

    Kumar, Sunil; Sharma, Shruti

    2016-04-01

    The agenesis of the Müllerian duct is the second most common cause of primary amenorrhoea after Turner syndrome. The abnormal development of Müllerian duct often associates with the urinary tract and skeletal abnormalities. MURCS (Müllerian duct aplasia-renal agenesis-cervicothoracic somite dysplasia) association is a unique and rare developmental disorder with four common features of uterine hypoplasia or aplasia, renal agenesis or ectopy, vertebral anomalies and short stature. We report a case of young female with primary amenorrhoea. She had well-developed secondary sexual characteristics along with multiple congenital developmental abnormalities such as the absence of uterus, ectopic kidney, cervical vertebral fusion, hemivertebrae, scoliosis, cervical rib, facial asymmetry and growth retardation. Our case highlights the rarity and clinical importance of this syndrome. For the evaluation of primary amenorrhoea in a female with well-developed secondary sexual characteristics, congenital anomalies should be ruled out before hormone and karyotype analyses. PMID:27099773

  10. A successful laparoscopic neovaginoplasty using peritoneum in Müllerian agenesis with inguinal ovaries accompanied by primary ovarian insufficiency

    Science.gov (United States)

    Gweon, Seonghye; Lee, Jisun; Hwang, Suna; Hwang, Kyoung Joo

    2016-01-01

    The combination of Müllerian agenesis with inguinal ovaries accompanied by primary ovarian insufficiency is extremely rare. A 21-year-old Korean woman was referred to our center with primary amenorrhea. The patient was diagnosed with Müllerian agenesis with inguinal ovaries. Her hormonal profile showed hypergonadotrophic hypogonadism suggesting primary ovarian insufficiency. We performed laparoscopic neovaginoplasty using modified Davydov's procedure and reposition inguinal ovaries in the pelvic cavity. Oral estrogen replacement was applied for the treatment of primary ovarian insufficiency. This is a rare case report on Mayer-Rokitansky-Kuster-Hauser syndrome accompanied not only by inguinal ovaries but also with primary ovarian insufficiency. We present our first experience on the laparoscopic neovaginoplasty performed on the patient with müllerian agenesis accompanied by inguinal ovaries and primary ovarian insufficiency. PMID:27462606

  11. Bilateral support programmes

    International Nuclear Information System (INIS)

    In the Russian Federation, serious consideration is being given to the safety of nuclear facilities and to security of nuclear material. One important factor in ensuring security of nuclear material and facilities is physical protection. The Ministry of Atomic Energy of the Russian Federation is making great efforts to improve physical protection of nuclear facilities. Some of the measures which are being implemented to improve physical protection are being carried out in the framework of international co-operation in this field. To date, the Russian Federation has established bilateral co-operation with the United States of America, Germany, Belgium, France and Finland, and has studied and taken account of the experience of foreign countries in the field of physical protection and nuclear material accounting and control. At present, over 20 enterprises of the Ministry of Atomic Energy of the Russian Federation representing all the main branches of the nuclear activities of the Ministry are involved in international co-operation on physical protection and nuclear material accounting and control. The main branches of this co-operation are as follows: Scientific and technical co-operation; production of a conceptual design to improve physical protection systems directly at nuclear risk facilities; swift improvement of physical protection (without developing design documentation for the nuclear risk facility as a whole); reinforcement of physical protection during the transport of nuclear material; and training of staff at nuclear risk facilities. During this work, many facilities were equipped with modern physical protection and with nuclear material accounting and control equipment. The Ministry of Atomic Energy of the Russian Federation is pleased with the results of this international co-operation and feels that it should continue. (author)

  12. Bilateral internal laryngoceles mimicking asthma.

    Science.gov (United States)

    Aksoy, Elif A; Elsürer, Cağdaş; Serin, Gediz M; Unal, O Faruk

    2013-05-01

    Laryngocele is an air-filled, abnormal dilation of the laryngeal saccule that extends upward within the false vocal fold, in communication with the laryngeal lumen. A case of 43-year-old male with bilateral internal laryngoceles, who has been treated as asthma for 4 years, is presented. The patient had dyspnea, cough, and excessive phlegm for a month and a late onset stridor. Flexible nasopharyngolaryngoscopy showed bilateral cystic enlargements of the false vocal folds and true vocal folds could not be visualized. Laryngeal CT without contrast enhancement showed bilateral internal laryngoceles. Submucosal total excision of bilateral cystic masses including parts of false vocal folds was performed. The symptoms resolved immediately after surgery. Although the incidence of internal laryngocele is rare, it should be remembered in the differential diagnosis of upper airway problems and diagnostic flexible nasopharnygolaryngoscopy is routinely indicated for airway evaluation in at-risk patients. PMID:24174956

  13. Bilateral internal laryngoceles mimicking asthma

    Directory of Open Access Journals (Sweden)

    Elif A Aksoy

    2013-01-01

    Full Text Available Laryngocele is an air-filled, abnormal dilation of the laryngeal saccule that extends upward within the false vocal fold, in communication with the laryngeal lumen. A case of 43-year-old male with bilateral internal laryngoceles, who has been treated as asthma for 4 years, is presented. The patient had dyspnea, cough, and excessive phlegm for a month and a late onset stridor. Flexible nasopharyngolaryngoscopy showed bilateral cystic enlargements of the false vocal folds and true vocal folds could not be visualized. Laryngeal CT without contrast enhancement showed bilateral internal laryngoceles. Submucosal total excision of bilateral cystic masses including parts of false vocal folds was performed. The symptoms resolved immediately after surgery. Although the incidence of internal laryngocele is rare, it should be remembered in the differential diagnosis of upper airway problems and diagnostic flexible nasopharnygolaryngoscopy is routinely indicated for airway evaluation in at-risk patients.

  14. Bilateral Polydactyly in a foal

    OpenAIRE

    Carstanjen, Bianca; Abitbol, Marie; Desbois, Christophe

    2007-01-01

    The following case report describes the diagnosis and surgery of bilateral polydactyly of unknown origin in a colt. A 7-month-old Berber colt was referred for cosmetic and curative excision of supernumerary digits. Radiographic examination revealed bilateral polydactyly and well-developed first carpal bones. Surgery consisted of an osteotomy of both second metacarpal bones combined with an amputation of the supernumerary digits. The follow-up at 18 months after surgery revealed a sound horse ...

  15. Bilateral Symmetrical Parietal Extradural Hematoma

    OpenAIRE

    Agrawal, Amit

    2011-01-01

    The occurrence of bilateral extradural hematomas (EDH) is an uncommon consequence of craniocerebral trauma, and acute symmetrical bilateral epidural hematomas are extremely rare. We discuss the technique adopted by us for the management of this rare entity. A 55-year-old patient presented with history of fall of branch of tree on her head. She had loss of consciousness since then and had multiple episodes of vomiting. Examination of the scalp was suggestive of diffuse subgaleal hematoma. Her ...

  16. Bilateral cleft lip nasal deformity

    OpenAIRE

    Singh Arun; Nandini R.

    2009-01-01

    Bilateral cleft lip nose deformity is a multi-factorial and complex deformity which tends to aggravate with growth of the child, if not attended surgically. The goals of primary bilateral cleft lip nose surgery are, closure of the nasal floor and sill, lengthening of the columella, repositioning of the alar base, achieving nasal tip projection, repositioning of the lower lateral cartilages, and reorienting the nares from horizontal to oblique position. The multiplicity of procedures in the li...

  17. Late Bilateral Renal Metastasis of Choriocarcinoma: A Case Report

    Directory of Open Access Journals (Sweden)

    M. Tahmasebi

    2008-01-01

    Full Text Available Introduction: Late bilateral renal metastasis of chori-ocarcinoma is a rare condition. Herein we report a case of late renal metastasis of choriocarcinoma."nCase Presentation: A 28 years old woman presented with gross hematuria since three months ago. A mo-bile palpable mass in her right flank was detected. She was not pregnant but had a history of abortion at six weeks of gestation in seven years age. Her gyneco-logic examination was normal. She had severe anemia (Hb=7.2, elevated ESR (52 with normal renal and liver function testes. Ultrasonography showed bilat-eral large kidney masses by solid-cystic components, with predominant solid parts. Bilateral lobulated het-erogeneous renal masses were found at non-enhancing axial CT-Scan, with notable enhancement of solid parts after administration of contrast agent. Angiography revealed bilateral hypovascular renal masses. Right radical nephrectomy performed for the patient and pathology reported choriocarcinoma of kidney. Finally, she was referred for chemotherapy of contralateral renal mass, but she delayed that for three months. She died following a seizure attack and cardiopulmonary arrest without response to prompt cardio-pulmonary resuscitation. Autopsy not permit-ted by relevants. "nConclusion: Late metastases of choriocarcinoma may appear in kidney or brain after a long time following the primary source of malignancy, when the primary lesion has disappeared.

  18. A rare case of cerebellar agenesis: a probabilistic Constrained Spherical Deconvolution tractographic study.

    Science.gov (United States)

    Mormina, Enricomaria; Briguglio, Marilena; Morabito, Rosa; Arrigo, Alessandro; Marino, Silvia; Di Rosa, Gabriella; Micalizzi, Alessia; Valente, Enza Maria; Salpietro, Vincenzo; Vinci, Sergio Lucio; Longo, Marcello; Granata, Francesca

    2016-03-01

    Aim of this study is to show the potential of probabilistic tractographic techniques, based on the Constrained Spherical Deconvolution (CSD) algorithms, in recognizing white matter fiber bundle anomalies in patients with complex cerebral malformations, such as cerebellar agenesis. The morphological and tractographic study of a 17-year-old male patient affected by cerebellar agenesis was performed by using a 3Tesla MRI scanner. Genetic and neuropsychological tests were carried out. An MRI morphological study showed the absence of both cerebellar hemispheres and the flattening of the anterior side of the pons. Moreover, it showed a severe vermian hypoplasia with a minimal vermian residual. The study recognized two thin cerebellar remnants, medially in contact with the small vermian residual, at the pontine level. The third ventricle, morphologically normal, communicated with a permagna cerebello-medullary cistern. Probabilistic CSD tractography identified some abnormal and aberrant infratentorial tracts, symmetrical on both sides. In particular, the transverse pontine fibers were absent and the following tracts with aberrant trajectories have been identified: "cerebello-thalamic" tracts; "fronto-cerebellar" tracts; and ipsilateral and contralateral "spino-cerebellar" tracts. Abnormal tracts connecting the two thin cerebellar remnants have also been detected. There were no visible alterations in the main supratentorial tracts in either side. Neuropsychiatric evaluation showed moderate cognitive-motor impairment with discrete adaptive compensation. Probabilistic CSD tractography is a promising technique that overcome reconstruction biases of other diffusion tensor-based approaches and allowed us to recognize, in a patient with cerebellar agenesis, abnormal tracts and aberrant trajectories of normally existing tracts. PMID:25832852

  19. Hernia of the bladder through the broad ligament with renal agenesis and ipsilateral ureter blind ending.

    Science.gov (United States)

    El Madi, Aziz; Khattala, Khalid; Rami, Mohammed; Bouabdallah, Youssef

    2014-01-01

    Hernia through a defect of the broad ligament is extremely rare in children. These defects can result from a developmental defect or a spontaneous rupture of cystic formations remnants of mesonephric ducts or Müller. Genital anomalies associated with unilateral renal agenesis are more common in females. We report the case of a 13 months girl allowed for assessment of recurrent urinary tract infection; abdominal examination did not objectified palpable mass, the external genitalia were without abnormalities. Abdominal ultrasound revealed a left renal space is empty with a retrovesical cyst. Cystography was requested that objectified a large pelvic cystic mass retrovesical communicating with the bladder, there was also a left vesicoureteral reflux. Uro- MRI showed a cystic formation retrovesical communicating with the bladder, the right pelvic kidney; uterus is normal size for age. DMSA scintigraphy confirmed the absence of the left kidney with the right kidney that ensures 100% of total renal function. To surgical exploration we found a hernia of the bladder through the left broad ligament, the uterus was dislocated on the right side; left ovary was hypoplasic; the ipsilateral ureter was blind with renal agenesis, we performed by reduction of the bladder then closing the hernial orifice, dissection of the ureter with its ligation and section at the vesical stoma. The postoperative course was uneventful. Evolution is favorable. This observation illustrates a hernia of the bladder through the broad ligament associated with ovarian hypoplasia, renal agenesis and ipsilateral ureter blind ending; this association was not described to our knowledge in the literature. PMID:25422693

  20. Tooth agenesis in patients referred to an Irish tertiary care clinic for the developmental dental disorders.

    LENUS (Irish Health Repository)

    Hashem, Atef A

    2010-01-01

    PURPOSE: This study was carried out to determine the prevalence, severity and pattern of hypodontia in Irish patients referred to a tertiary care clinic for developmental dental disorders. MATERIALS AND METHODS: Details of 168 patients with hypodontia referred during the period 2002-2006 were entered in a database designed as a national record. Tooth charting was completed using clinical and radiographic examinations. The age of patients ranged from 7-50 years, with a median age of 20 years (Mean: 21.79; SD: 8.005). RESULTS: Hypodontia referrals constituted 65.5% of the total referrals. Females were more commonly affected than males with a ratio of 1.3:1. The number of referrals reflected the population density in this area; the majority were referrals from the public dental service. Mandibular second premolars were the most commonly missing teeth, followed by maxillary second premolars and maxillary lateral incisors; maxillary central incisors were the least affected. Symmetry of tooth agenesis between the right and left sides was an evident feature. Slightly more teeth were missing on the left side (n = 725) than on the right side (n = 706) and in the maxillary arch (n = 768) as compared to the mandibular arch (n = 663). Some 54% of patients had severe hypodontia with more than six teeth missing; 32% had moderate hypodontia, with four to six teeth missing. The most common pattern of tooth agenesis was four missing teeth. CONCLUSION: Hypodontia was a common presentation in a population referred to this tertiary care clinic. The pattern and distribution of tooth agenesis in Irish patients appears to follow the patterns reported in the literature.

  1. Agenesis and not ectopia is common in North Indian children with thyroid dysgenesis

    Directory of Open Access Journals (Sweden)

    Devi Dayal

    2014-01-01

    Full Text Available Context: Ectopic Thyroid Gland (ETG is known to be the most common form of thyroid dysgenesis in children with permanent congenital hypothyroidism (CH. Recent reports indicate that agenesis or hypoplasia of thyroid gland may be commoner as compared to ETG in thyroid dysgenesis (TD. There is limited information available on the proportion of different variants of TD in Indian children. Aim: To characterize the different TD variants in a cohort of North Indian children with TD. Settings and Design: Endocrinology Unit of a large Multispecialty Pediatrics Center located in North India. Retrospective review of clinical records of children with CH due to TD diagnosed between April 2004 and March 2014. Results: Diagnoses of TD in 94 children (48 boys and 46 girls were based on combined scanning with high-resolution ultrasonography, and technetium-99m pertechnetate thyroid scintigraphy. Thyroid agenesis, ectopia and hypoplasia were diagnosed in 74 (78.7%, 14 (14.8% and 6 (6.4% patients respectively. The mean initial serum total T4 and thyroid stimulating hormone concentrations at diagnosis were 3.03 ± 2.88 μg/dL (range 0.01-8.9 and 284.52 ± 300.67 mIU/L (range 10.03-1159.0 respectively. Patients with ETG were older at the time of diagnosis as compared to patients with hypoplasia or ectopia. The mean duration of follow-up was 3.7 ± 2.85 years (range 3 months-10 years. Conclusions : Thyroid agenesis was the most common form of TD in our children with permanent CH. Hypoplasia and ectopia were uncommon. Female preponderance, noted in many previous reports, was not seen in our patients with TD.

  2. Laparoscopic and gasless laparoscopic sigmoid colon vaginoplasty in women with vaginal agenesis

    Institute of Scientific and Technical Information of China (English)

    ZHONG Chen-xi; WU Ji-xiang; LIANG Jie-xiong; WU Qing-hua

    2012-01-01

    Background In the past several decades we have seen multiple advances in the reconstruction for girls born with vaginal agenesis.This study aimed to evaluate the technical feasibility,anatomical and functional outcomes of one-stage laparoscopic and gasless laparoscopic vaginoplasty with sigmoid colon for the patients of vaginal agenesis (Mayer-Rokitansky-Kuster-Hauser syndrome).Methods We did a retrospective review of a total of 150 women with Mayer-Rokitansky-Kuster-Hauser syndrome treated at Beijing Anzhen Hospital,Capital Medical University from March 2006 to August 2010.The patients were divided into the CO2 pneumoperitoneum laparoscopic group and the abdominal wall lift of gasless laparoscopic group.Sigmoid colon vaginoplasty approaches were performed in all of the patients.The surgical techniques,perioperative results,complications,anatomical and functional outcomes of vaginoplasty were recorded.Results All procedures were performed successfully.Significant differences in the operative time and intraoperative blood loss existed in the laparoscopic vaginoplasty group compared with the gasless laparoscopic vaginoplasty group.The patients who underwent sigmoid colon vaginoplasty had good cosmetic results without the problem of excessive mucus production.The postoperative complications were minimal.During a mean follow-up of 15.6 months,no stenosis or shrinkage was encountered.The subjective sexual satisfaction rate with the surgical outcomes in all patients was 83.3%.Conclusions Laparoscopic or gasless laparoscopic vaginoplasty with sigmoid colon are effective and feasible approaches for women with congenital vaginal agenesis.The procedures have satisfactory anatomical and functional results.

  3. Pulmonary studies

    International Nuclear Information System (INIS)

    Radionuclide studies of the lung are described, as regards perfusion studies, ventilation studies and physiological considerations. The four principal applications for radionuclide studies of the lungs are outlined and the uses of these discussed in relation to particular entities including pulmonary embolic disease, congestive heart failure, asthma, acute, nonasthmatic, bronchial obstruction, chronic pulmonary disease and cancer. (Auth./C.F.)

  4. Simultaneous Bilateral Ophthalmic Artery Chemosurgery for Bilateral Retinoblastoma (Tandem Therapy)

    Science.gov (United States)

    Abramson, David H.; Marr, Brian P.; Francis, Jasmine H.; Dunkel, Ira J.; Fabius, Armida W. M.; Brodie, Scott E.; Mondesire-Crump, Ijah; Gobin, Y. Pierre

    2016-01-01

    Objective Report on the 7-year experience with bilateral ophthalmic artery chemosurgery (OAC-Tandem therapy) for bilateral retinoblastoma. Design Retrospective, single institution study. Subjects 120 eyes of 60 children with bilateral retinoblastoma treated since March 2008. Methods Retrospective review of all children treated at Memorial Sloan Kettering with bilateral ophthalmic artery chemosurgery (Melphalan, Carboplatin, Topotecan, Methotrexate) delivered in the same initial session to both naïve and previously treated eyes. Main Outcome Measures Ocular survival, metastatic disease, patient survival from metastases, second cancers, systemic adverse effects, need for transfusion of blood products, electroretinogram before and after treatment. Results 116 eyes were salvaged (4 eyes were enucleated: 3 because of progressive disease, 1 family choice). Kaplan Meier ocular survival was 99.2% at one year, 96.9% at 2 and 3 years and 94.9% for years 4 through 7. There were no cases of metastatic disease or metastatic deaths with a mean follow-up of 3.01 years. Two children developed second cancers (both pineoblastoma) and one of them died. Transfusion of blood products was required in 3 cases (4 transfusions), 1.9%. Two children developed fever/neutropenia requiring hospitalization (0.95%). ERGs were improved in 21.6% and unchanged after treatment in 52.5% of cases (increase or decrease of less than 25μV). Conclusions Bilateral ophthalmic artery chemosurgery is a safe and effective technique for managing bilateral retinoblastoma-even when eyes are advanced bilaterally, and if both eyes have progressed after systemic chemotherapy. Ocular survival was excellent (94.9% at 8 years), there were no cases of of metastatic disease and no deaths from metastatic disease, but children remain at risk for second cancers. In 21.6% of cases ERG function improved. Despite using chemotherapy in both eyes in the same session, systemic toxicity was low. PMID:27258771

  5. OHVIRA: Uterus didelphys, blind hemivagina and ipsilateral renal agenesis: Advantage MRI

    Directory of Open Access Journals (Sweden)

    Sunil K Bajaj

    2012-01-01

    Full Text Available We present here a case of an uncommon complex uterine anomaly - Obstructed HemiVagina with Ipsilateral Renal Agenesis (OHVIRA, also known as Herlyn-Werner-Wunderlich syndrome in a 14-year-old girl along with sonographic (trans-abdominal and trans labial, and MRI findings. The patient underwent surgery wherein imaging findings were confirmed. An MRI has proved to be of great help in correct diagnosis avoiding surgical interventions/ laparoscopy, which were needed in past to diagnose this rare anomaly. We also discuss the development of this anomaly with the help of a relatively new theory of uro-genital development by Acien and review the literature.

  6. Solitary Median Maxillary Central Incisor Versus Agenesis of the Maxillary Central Incisor.

    Science.gov (United States)

    de Carvalho, Fabrício Kitazono; Arid, Juliana; De Rossi, Andiara; Paula-Silva, Francisco W G; Nelson-Filho, Paulo

    2016-01-01

    A solitary median maxillary central incisor (SMMCI) is rare and affected individuals may carry a potentially serious condition known as SMMCI syndrome. However, many of these cases do not receive proper attention because they are misdiagnosed as agenesis of the maxillary central incisor. The purpose of this manuscript is to report two cases of children with only one maxillary central incisor and draw diagnostic differences between the entities. A correct diagnosis is very important because if an SMMCI is confirmed, the patient should be referred for genetic counseling. PMID:27098718

  7. Coexistence of Left Internal Carotid Agenesis, Klippel-Feil Syndrome and Postaxial Polydactyly

    International Nuclear Information System (INIS)

    Internal carotid artery agenesis is a rare anomaly that can be clinically asymptomatic. Klippel-Feil syndrome is a skeletal malformation characterized by vertebral fusion. Presence of postaxial polydactyly is suggestive of an underlying syndrome. We report a rare case of a 44-year-old patient with non-specific symptoms and an association between these three rare abnormalities. Vascular anomalies were found using intracranial MR angiography and multi-detector CT angiography of the supraaortic arteries. Presence of a single aforementioned anomaly requires cautious imaging assessment in order to detect possible associated anomalies and avoid diagnostic pitfalls. A possible common genetic background could explain the coexistence of these three anomalies

  8. Unilateral agenesis of the abdominal wall musculature: An early muscle deficiency.

    Science.gov (United States)

    Gerard-Blanluet, Marion; Port-Lis, Marylin; Baumann, Clarisse; Perrin-Sabourin, Laurence; Ebrad, Patrick; Audry, Georges; Delezoide, Anne-Lise; Verloes, Alain

    2010-11-01

    Prune-belly sequence (PBS) usually results from early urethral obstruction. In rare cases, PBS seems to be due to a faulty primary development of the parietal mesenchyme leading to underdevelopment of the abdominal wall musculature, and disorganization of the smooth muscles in the urinary tract. We report on two patients with segmental, unilateral wall musculature deficiency associated with homolateral agenesis of ribs. One patient also had hemivertebrae and the other one ipsilateral diaphragmatic eventration and aplasia cutis. This combination of anomalies may represent a localized deficiency in the development of somitic mesoderm mesenchyme during early embryogenesis. PMID:20949627

  9. Uterus didelphys with partial vaginal septum and distal vaginal agenesis: an unusual anomaly.

    Science.gov (United States)

    Singhal, Savita Rani; Lakra, Pinkey; Bishnoi, Pushpa; Rohilla, Seema; Dahiya, Pushpa; Nanda, Smiti

    2013-02-01

    Complex malformations of female genital tract are not so common. Their correct identification is of paramount importance for appropriate management. A thorough knowledge of embryology, pre-operative imaging with MRI and examination under anaesthesia is essential to identify accurately the constellation of anomalies and to plan appropriate management. This case reports the coexistence of Müllerian abnormality with vaginal agenesis in an 18 years old girl which was managed by dissecting the lower half of vagina and pull-through vaginoplasty. PMID:23374522

  10. Bilateral hemimelia of radio in canine – case reportHemimelia bilateral de rádio em canino – relato de caso

    Directory of Open Access Journals (Sweden)

    Thalita Priscila da Silva Peres

    2013-10-01

    Full Text Available Hemimelia or agenesis is a congenital morphological change that may affect the appendicular skeleton. This rare anomaly is the total or partial absence of one or more bones matched. It may present unilaterally or bilaterally, with the most frequent unilateral. In dogs, the most common form of the radial hemimelia is that causes deformity of the affected limb and severe functional limitations. Was Veterinary Hospital of the Federal University of Mato Grosso (HOVET/UFMT, a canine, female mongrel, with 30 days of age, presenting deformity in thoracic limbs since birth, observed signs of lameness and difficulty support. After complete bone development the animal returned for re-evaluation and establishment of appropriate treatment. On radiographic examination, there was bilateral agenesis of radios, angulation of the ulnar cranially, fusion of carpal bones proximal and distal to the ranks, bilateral scapulohumeral dislocation and the presence of a bone structure with well-defined boundaries and rectangular articulated with the scapula. Due to good quality of life of the animal and reserved prognosis of surgery, we opted for conservation of members. The owner was recommended to keep the animal in an environment with soft ground and castration. Hemimelia ou agenesia é uma alteração morfológica congênita que pode acometer o esqueleto apendicular. Esta anomalia rara consiste na ausência total ou parcial de um ou mais ossos pareados. Pode se apresentar de forma uni ou bilateral, sendo a unilateral mais frequente. Em cães, a forma mais comum de hemimelia é a radial, que provoca deformidade do membro afetado e severas limitações funcionais. Foi atendido no Hospital Veterinário da Universidade Federal de Mato Grosso (HOVET/UFMT, um canino, fêmea, sem raça definida, com 30 dias de idade, apresentando deformidade nos membros torácicos desde o nascimento, claudicação e dificuldade de apoio. Após o completo desenvolvimento ósseo o animal

  11. Flash pulmonary edema in patients with renal artery stenosis--the Pickering Syndrome

    DEFF Research Database (Denmark)

    Pelta, Anna; Andersen, Ulrik B; Just, Sven;

    2010-01-01

    We report the prevalence of flash pulmonary edema in patients consecutively referred for balloon angioplasty of uni- or bilateral renal artery stenosis (PTRA), and describe the characteristics of this special fraction of the patients. We further report two unusual cases.......We report the prevalence of flash pulmonary edema in patients consecutively referred for balloon angioplasty of uni- or bilateral renal artery stenosis (PTRA), and describe the characteristics of this special fraction of the patients. We further report two unusual cases....

  12. Functional Benefits of Bilateral Emphysema Reduction Surgery in an Adolescent.

    Science.gov (United States)

    Julian, Valérie; Moreau de Bellaing, Anne; Filaire, Marc; Richard, Ruddy; Labbé, André

    2016-09-01

    Acquired emphysema is a rare pathology in pediatrics. We report the case of a patient born at term with a neonatal respiratory distress, which had required mechanical ventilation. She developed gradually chronic obstructive pulmonary disease with severe emphysematous lesions, respiratory failure and functional impairments. Bilateral emphysema resection, performed at 16 years old, allowed major functional benefits at rest and during exercise. We present the results of respiratory functional evaluations, walk tests and maximal exercise tests (including measure of dynamic hyperinflation) before and after surgery, which highlights that surgery is a successful option in the treatment of compressive emphysema in childhood. PMID:27549540

  13. Neuralgic Amyotrophy: A Rare Cause of Bilateral Diaphragmatic Paralysis

    Directory of Open Access Journals (Sweden)

    Neil Shinder

    1998-01-01

    Full Text Available Neuralgic amyotrophy, also known as brachial neuritis, is a well described clinical entity. Diaphragmatic dysfunction, as a result of phrenic nerve root involvement (cervical roots 3 to 5, is an uncommon, but increasingly recognized association. The case of a previously healthy 61-year-old woman who, after a prodrome of neck and shoulder discomfort, presented with severe orthopnea is described. Pulmonary function and electrophysiological studies led to a diagnosis of bilateral diaphragmatic paralysis. The patient's clinical course and the exclusion of other nerve entrapment syndromes and neurological disorders strongly favoured the diagnosis of neuralgic amyotrophy.

  14. The radiological findings of pulmonary paragonimiasis

    International Nuclear Information System (INIS)

    Authors analysed chest X-ray findings in 36 cases of proven pulmonary paragonimiasis which sputum smears were positive for ova of Paragonimus Westermani from October 1978 to January 1987 at Chonbuk National University Hospital, PMC and Korean Institute of tuberculosis. The results are as follows: 1. Roentgenographic findings are cyst formation, pulmonary infiltration, fibrosis, pleural thickening, pleural effusion and pneumothorax. 2. The characteristic cystic shadows were observed in 19 cases (58%). They were multiple aggregated cysts surrounded with pericystic haziness and ranged in size from 1cm to 4cm in diameter. 3. Pulmonary infiltrations showed multiple, variable ill defined area of consolidation, predominantly in periphery of mid and lower lung field, which has migratory tendency. 4. Distribution of pulmonary lesions were bilateral (72%), mid lung fields (56%), and there were tendency of peripheral lung predominance. 5. 4 cases (11%) showed normal chest X-ray findings

  15. Paralisia facial bilateral Bilateral facial paralysis: a case report

    Directory of Open Access Journals (Sweden)

    J. Fortes-Rego

    1976-03-01

    Full Text Available É apresentado um caso de diplegia facial surgida após meningite meningocócica e infecção por herpes simples. Depois de discutir as diversas condições que o fenômeno pode apresentar-se, o autor inclina-se por uma etiologia herpética.A case of bilateral facial paralysis following meningococcal meningitis and herpes simplex infection is reported. The author discusses the differential diagnosis of bilateral facial nerve paralysis which includes several diseases and syndromes and concludes by herpetic aetiology.

  16. Bilateral spontaneous hemotympanum: Case report

    Directory of Open Access Journals (Sweden)

    Economou Nicolas C

    2006-10-01

    Full Text Available Abstract Background The most common causes of hemotympanum are therapeutic nasal packing, epistaxis, blood disorders and blunt trauma to the head. Hemotympanum is characterized as idiopathic, when it is detected in the presence of chronic otitis media. A rare case of spontaneous bilateral hemotympanum in a patient treated with anticoagulants is presented herein. Case presentation A 72-year-old male presented with acute deterioration of hearing. In the patient's medical history aortic valve replacement 1 year before presentation was reported. Since then he had been administered regularly coumarinic anticoagulants, with INR levels maintained between 3.4 and 4.0. Otoscopy revealed the presence of bilateral hemotympanum. The audiogram showed symmetrical moderately severe mixed hearing loss bilaterally, with the conductive component predominating. Tympanograms were flat bilaterally with absent acoustic reflexes. A computerized tomography scan showed the presence of fluid in the mastoid and middle ear bilaterally. Treatment was conservative and consisted of a 10-day course of antibiotics, anticongestants and temporary interruption of the anticoagulant therapy. After 3 weeks, normal tympanic membranes were found and hearing had returned to previous levels. Conclusion Anticoagulant intake should be included in the differential diagnosis of hemotympanum, because its detection and appropriate treatment may lead to resolution of the disorder.

  17. MKS1 mutations cause Joubert syndrome with agenesis of the corpus callosum.

    Science.gov (United States)

    Bader, Ingrid; Decker, E; Mayr, J A; Lunzer, V; Koch, J; Boltshauser, E; Sperl, W; Pietsch, P; Ertl-Wagner, B; Bolz, H; Bergmann, C; Rittinger, O

    2016-08-01

    Joubert syndrome (JS) is a clinically and genetically heterogeneous ciliopathy characterized by episodic hyperpnea and apnea, hypotonia, ataxia, cognitive impairment and ocular motor apraxia. The "molar tooth sign" is pathognomonic of this condition. Mutations in the MKS1 gene are a major cause of Meckel-Gruber syndrome (MKS), the most common form of syndromic neural tube defects, frequently resulting in perinatal lethality. We present the phenotype and genotype of a child with severe JS and agenesis of the corpus callosum (ACC). In our patient, a next generation sequencing (NGS) approach revealed the following two variants of the MKS1 gene: first, a novel missense variant [ c.240G > T (p.Trp80Cys)], which affects a residue that is evolutionarily highly conserved in mammals and ciliates; second, a 29 bp deletion in intron 15 [c.1408-35_1408-7del29], a founder mutation, which in a homozygous state constitutes the major cause of MKS in Finland. We review the MKS1-variants in all of the eleven JS patients reported to date and compare these patients to our case. To our knowledge, this is the first patient with Joubert syndrome and agenesis of the corpus callosum where a potentially causal genotype is provided. PMID:27377014

  18. PSYCHO-MOTRIC REHABILITATION IN CHILDREN WITH AGENESIS OF CORPUS CALLOSUM (Case study

    Directory of Open Access Journals (Sweden)

    Zărnica I. Manuela Mirela

    2011-12-01

    Full Text Available Agenesis of corpus callosum is a new challenge for the puluridisciplinary team, in their efforts for the rehabilitation of childrens with this diagnose, because is a rare congenital desease with an unknown frequence of emergence. Scope. This paperwork aims to present the approach modalities of the child with agenesis of corpus callousum and the means of physical therapy that may help. Methods. This is a case study of a 5 years old girl with asenesis of corpus callosum. For assessment we used motor assessment, range of motion, reflexes. Exercises from different positions were done as games and motric songs. Sensory stimulation was also done. Rezults. By direct observation of the patient we observed significant improvement in motor function. We also foud unexpected improvement of quiet stance and dynamic balance, sensory-motor coordination. Conclusions. Game, tha main therapeutic method through its form, diversity and characteristics - atractivity, directness, creativity, movement freedom – is the main supplement of child psicho-motric development. A balanced distribution of influences upon motricity, psychic, thinking and action, nonverbal and expression language, represent the qualitative peculiarity of specialist’s and parents efforts.

  19. Uterus didelphys with an obstructed unilateral vagina and ipsilateral renal agenesis: A rare cause of dysmenorrhoea.

    Science.gov (United States)

    Attar, Rukset; Yıldırım, Gazi; Inan, Yücel; Küzılkale, Ozge; Karateke, Ateş

    2013-01-01

    Didelphic uterus with obstructed hemivagina and ipsilateral renal agenesis is a rare condition. It usually presents with pelvic pain following the menarche, dysmenorrhoea, and an increase in abdominal volume or a palpable mass due to unilateral haematocolpos. We present the case of a 13-year-old girl who referred with recurrent pelvic pain, mainly at the time of menses, and irregular menstrual cycle complaints in this report. The patient underwent ultrasonography and magnetic resonance (MR) imaging of the pelvis was performed. The diagnosis was uterus didelphys with obstructed hemivagina and ipsilateral renal agenesia. Laparotomy was performed for diagnosis and treatment purposes. Two separated hemiuteri and two cervices with hematometra and hematocolpos on the right side and ipsilateral renal agenesis were detected. The vaginal septum was excised completely and Strassman metroplasty was performed. Her complaints were resolved and she was absolutely asymptomatic after surgery. Diagnosis and management of this congenital anomaly is challenging due to the complexity of the anatomic structures, nonspecific complaints, and heterogenic presentation. These anomalies must always be considered while working-up female patients presenting with episodic abdominal pain and abdominopelvic mass. PMID:24592115

  20. Concomitant bilateral testicular epidermoid cysts

    International Nuclear Information System (INIS)

    Epidermoid cyst of the testis is a rare benign germ tumor, comprising 1-2% of all resected benign testicular masses. Approximately 300 cases have been reported to date. Unilateral involvement has often been reported in the English literature. However, bilateral occurrence is very rare and to the best of our knowledge, there only 3 previous reports of bilateral testicular epidermoid cysts. The fact that they are completely benign makes them amenable to treatment by local excision, thereby saving patient from orchidectomy. Recognition of their characteristic ulatrsonographic features is very important to avoid unnecessary intervention. We present here, a case of bilateral epidermoid cyst in which characteristic ultrasound (US) findings allowed testis-sparing enucleation instead of radical orchiectomy. (author)

  1. Pulmonary Atresia

    Science.gov (United States)

    ... to repair the defect. Return to main topic: Congenital Heart Disease See on other sites: MedlinePlus https://medlineplus.gov/ency/article/001091.htm Pulmonary atresia American Heart Association www. ...

  2. [Pulmonary rehabilitation].

    Science.gov (United States)

    Senjyu, Hideaki

    2016-05-01

    Pulmonary rehabilitation commenced in Japan in 1957. However, the development of pulmonary rehabilitation took a long time due to the lack of the necessary health and medical services. Pulmonary rehabilitation is a comprehensive intervention based on a thorough patient assessment followed by patient-tailored therapies that include, but are not limited to, exercise training, education, and behavior change, designed to improve the physical and psychological condition of people with chronic respiratory disease and to promote the long-term adherence to health-enhancing behaviors. The benefits of pulmonary rehabilitation include a decrease in breathlessness and an improvement in exercise tolerance. It is important that the gains in exercise tolerance lead to an increase in daily physical activity. PMID:27254948

  3. Pulmonary sequestration

    International Nuclear Information System (INIS)

    Pulmonary sequestration is a congenital affection consisting in the presence of a cystic mass of no-functional pulmonary tissue without an obvious communication with tracheobronchial tree and that receives all or most of its bloodstream of the anomalous vessels from systemic circulation. Taking into account that presentation of this affection is rare compared to other pulmonary affections (between the 1% and the 2% of all pulmonary resections) and that also the more usual is its definitive treatment before adulthood. The case of man aged 44 is presented coming to consultation due to frequent episodes of pneumonias from more 10 years ago diagnosed as a bronchiectasis. The more significant facts of embryology origin of this affection including: anatomical and pathological features, imaging diagnosis, surgical treatment details, and postoperative course. (author)

  4. PULMONARY SCEDOSPORIOSIS

    Directory of Open Access Journals (Sweden)

    SEVERO Luiz Carlos

    1998-01-01

    Full Text Available A case of a solitary pulmonary nodule due to Scedosporium apiospermum (Pseudallescheria boydii is related. A review of the pertinent literature was done and, in addition, similar lesions caused by other opportunistic fungi are commented.

  5. Pulmonary manifestation of leptospirosis

    International Nuclear Information System (INIS)

    Authors analysed and present chest X-ray findings of serologically proven leptospirosis from Seoul National University Hospital, either admitted or referred for serological verification, during recent 2 years. Radiological findings were correlated with the lung specimen findings of experimentally induced leptospirosis in guinea pig. The results are as follows: 1. 24 cases (56%) showed positive X-ray findings. 2. Predominant radiological patterns of involved lung were tiny dot, small nodule, rosette density in 11 cases, massive confluent consolidation in 4 cases, and diffuse ill-defined velly increased density in 9 cases. 3. Distribution of pulmonary lesions were bilateral (100%), non-lobar, non-segmental (95%), and there were conspicuous tendency of peripheral lung predominance. 4. Extrapulmonary manifestation, such as pleural effusion or cardiomegaly was rate. 5. Pulmonary lesions resolved completely usually 5 to 10 days after their appearance. 6. From the gross and microscopic findings of serially sacrificed guinea pig's lung and a case of autopsy, authors concluded that fine dot-like density in chest X-ray was due to paleolithic hemorrhage in intraalveolar space at initial stage, growing up to ressette density or confluent consolidation as the pathetic extends to surrounding lung forming purpura and coalescent hemorrhage.

  6. Pulmonary manifestation of leptospirosis

    Energy Technology Data Exchange (ETDEWEB)

    Im, Jung Gi; Yeon, Kyung Mo; Han, Man Chung; Kim, Chu Wan; Lee, Jung Sang; Kim, Suhng Gwon; Han, Yong Chol; Chang, Woo Hyun; Chi, Je Geun [Seoul National University College of Medicine, Seoul (Korea, Republic of)

    1986-02-15

    Authors analysed and present chest X-ray findings of serologically proven leptospirosis from Seoul National University Hospital, either admitted or referred for serological verification, during recent 2 years. Radiological findings were correlated with the lung specimen findings of experimentally induced leptospirosis in guinea pig. The results are as follows: 1. 24 cases (56%) showed positive X-ray findings. 2. Predominant radiological patterns of involved lung were tiny dot, small nodule, rosette density in 11 cases, massive confluent consolidation in 4 cases, and diffuse ill-defined velly increased density in 9 cases. 3. Distribution of pulmonary lesions were bilateral (100%), non-lobar, non-segmental (95%), and there were conspicuous tendency of peripheral lung predominance. 4. Extrapulmonary manifestation, such as pleural effusion or cardiomegaly was rate. 5. Pulmonary lesions resolved completely usually 5 to 10 days after their appearance. 6. From the gross and microscopic findings of serially sacrificed guinea pig's lung and a case of autopsy, authors concluded that fine dot-like density in chest X-ray was due to paleolithic hemorrhage in intraalveolar space at initial stage, growing up to ressette density or confluent consolidation as the pathetic extends to surrounding lung forming purpura and coalescent hemorrhage.

  7. Pulmonary Edema

    OpenAIRE

    Tanser, Paul H.

    1981-01-01

    The physician who deals with pulmonary edema from a pathophysiologic basis will seldom make a diagnostic or therapeutic error. Recent additions to preload and afterload therapy have greatly helped in the emergency and ambulatory treatment of pulmonary edema due to left heart failure. Careful follow-up and patient self-monitoring are the most effective means of reducing hospitalization of chronic heart failure patients.

  8. Pulmonary hypertension

    OpenAIRE

    Anton Vonk Noordegraaf; Joanne A. Groeneveldt; Harm Jan Bogaard

    2016-01-01

    In 2015, more than 800 papers were published in the field of pulmonary hypertension. A Clinical Year in Review article cannot possibly incorporate all this work and needs to be selective. The recently published European guidelines for the diagnosis and treatment of pulmonary hypertension contain an inclusive summary of all published clinical studies conducted until very recently. Here, we provide an overview of papers published after the finalisation of the guideline. In addition, we summaris...

  9. Treatment to pulmonary alveolar proteinosis with total bronchoalveolar lavage

    International Nuclear Information System (INIS)

    The pulmonary alveolar proteinosis is a rare disease with variable course and characterized by the accumulation of surfactant in the alveoli. By the treatment it was used the alveolar lavage with good results. We present the experience accumulated in the Las Americas clinic of Medellin city with the treatment of three patients with diagnosis of pulmonary alveolar proteinosis by alveolar lavage. We realized seven lavages, two of them bilateral and sequential. We didn't 't have important complications and in all cases we got good clinical and physiological results. Two patients had relapsed six and seven months after the first lavage and they needed a second lavage. We conclude that the pulmonary lavage is a secure and effective procedure in the treatment of the pulmonary alveolar proteinosis and the modality of bilateral sequential lavage is a good alternative and less expensive

  10. Herlyn-Werner-Wunderlich Syndrome with Unilateral Hemivaginal Obstruction, Ipsilateral Renal Agenesis, and Contralateral Renal Thin GBM Disease: A Case Report with Radiological Follow Up

    International Nuclear Information System (INIS)

    Herlyn-Werner-Wunderlich syndrome is a rare Mullerian ductal anomaly that is characterized by the presence of a hemivaginal septum, a didelphic uterus and ipsilateral renal agenesis. It is generally difficult to diagnose the uterine malformation before menarche owing to its small size. Therefore, a follow-up study is very important for confirming the uterine malformation in girls with renal agenesis. We report a patient with renal agenesis and microscopic hematuria, who showed symptoms before menarche. A follow-up study eventually revealed uterine didelphys with a hemivaginal obstruction. A biopsy proved that the microscopic hematuria was caused by thin glomerular basement membrane disease of the contralateral kidney

  11. Herlyn-Werner-Wunderlich Syndrome with Unilateral Hemivaginal Obstruction, Ipsilateral Renal Agenesis, and Contralateral Renal Thin GBM Disease: A Case Report with Radiological Follow Up

    Energy Technology Data Exchange (ETDEWEB)

    Park, Noh Hyuck; Park, Hee Jin; Park, Chan Sup [Myongji Hospital, Kwandong University, Koyang (Korea, Republic of); Park, Sung Il [Bucheon Hospital, Soonchunhyang University, Bucheon (Korea, Republic of)

    2010-08-15

    Herlyn-Werner-Wunderlich syndrome is a rare Mullerian ductal anomaly that is characterized by the presence of a hemivaginal septum, a didelphic uterus and ipsilateral renal agenesis. It is generally difficult to diagnose the uterine malformation before menarche owing to its small size. Therefore, a follow-up study is very important for confirming the uterine malformation in girls with renal agenesis. We report a patient with renal agenesis and microscopic hematuria, who showed symptoms before menarche. A follow-up study eventually revealed uterine didelphys with a hemivaginal obstruction. A biopsy proved that the microscopic hematuria was caused by thin glomerular basement membrane disease of the contralateral kidney

  12. A Triad of Complete Dorsal Pancreatic Agenesis, Pancake Kidney and Bicornuate Uterus. An Association or an Incidental Finding: First Case in Literature

    Directory of Open Access Journals (Sweden)

    Anupam Lal

    2015-05-01

    Full Text Available Context Agenesis of the dorsal pancreas is a rare congenital anomaly which can be detected either incidentally during imaging or encountered during investigation for pancreas related pathologies such as pancreatitis or diabetes mellitus. Few associations of this condition with heterotaxy syndrome, pancreatic tumors and congenital heart disease have been described. Case report We report a novel association of genitourinary anomalies in a case of complete dorsal pancreatic agenesis not previously described in the literature. Conclusion Knowledge about dorsal pancreatic agenesis is essential for radiologist in patients presenting with unexplained pain abdomen and its association with various conditions may help radiologist in assisting clinician in management of these patients.

  13. Sequential bilateral retinal artery occlusion

    OpenAIRE

    Padrón-Pérez N; Aronés JR; Muñoz S; Arias-Barquet L; Arruga J

    2014-01-01

    Noel Padrón-Pérez,1 Janny Rosario Aronés,2 Silvia Muñoz,1 Luis Arias-Barquet,1 Jorge Arruga1,31Department of Ophthalmology, Hospital Universitari de Bellvitge, 2Hospital de l'Esperança – Parc de Salut Mar, 3Institut Català de Retina, Barcelona, SpainAbstract: An 86 year old woman experienced a sequential bilateral loss of vision over a period of less than 24 hours. Clinical findings and complementary studies suggested a bilateral atherogenic...

  14. Acute silicosis with bilateral pneumothorax

    OpenAIRE

    Srivastava, G. N.; Prasad, Rajniti; Meena, Manoj; Hussain, Moosa

    2014-01-01

    We present a case of acute silicosis with bilateral pneumothorax of a 28-year-old man working at a stone crusher factory for 1 year. He presented to the emergency department with cough, respiratory distress and diffuse chest pain. The patient was managed with bilateral intercostal tube drainage under water seal, oxygen inhalation and conservative therapy. On follow-up he showed improvement of resting dyspnoea and was doing well. This case is being reported because of the rare complications of...

  15. Different and Unpredictable Clinical Outcome of Ruptured Pulmonary Hydatid Cysts

    OpenAIRE

    Sheikhy, Kambiz; Abbasidezfouli, Mehregan; Daneshvar Kakhaki, Abolghasem; Saghebi, Seyed Reza; Malekzadegan, Alireza

    2015-01-01

    Most authors believe that the best treatment for pulmonary hydatid disease is surgical evacuation. Although albendazole has been used prophylactically before surgery, there are many reports about increased incidence of the rupture of cyst after albendazole therapy, which can cause some complications. In this case report we present a patient with bilateral pulmonary hydatid cyst that was ruptured after using albendazole and different strategies were used for management of each cyst.

  16. Acute respiratory distress syndrome: Pulmonary and extrapulmonary not so similar

    OpenAIRE

    Inderpaul Singh Sehgal; Sahajal Dhooria; Digambar Behera; Ritesh Agarwal

    2016-01-01

    Acute respiratory distress syndrome (ARDS) is characterized by acute onset respiratory failure with bilateral pulmonary infiltrates and hypoxemia. Current evidence suggests different respiratory mechanics in pulmonary ARDS (ARDSp) and extrapulmonary ARDS (ARDSexp) with disproportionate decrease in lung compliance in the former and chest wall compliance in the latter. Herein, we report two patients of ARDS, one each with ARDSp and ARDSexp that were managed using real-time esophageal pressure m...

  17. Sudden death of a patient with pulmonary Langerhans cell histiocytosis.

    Science.gov (United States)

    Nakhla, Hassan; Jumbelic, Mary I

    2005-06-01

    We report a case of sudden death due to bilateral pneumothorax in a previously healthy 16-year-old adolescent white girl. She presented with sudden onset of shortness of breath followed by loss of consciousness. Postmortem chest radiograph showed bilateral pneumothoraces. Autopsy confirmed the bilateral pneumothorax and additionally showed emphysematous changes and bullae throughout the lung tissue. Microscopic sections of the lungs showed Langerhans cell histiocytosis. To the best of our knowledge, this is the first reported case of fatal presentation of pulmonary Langerhans cell histiocytosis. PMID:15913433

  18. Types of Pulmonary Hypertension

    Science.gov (United States)

    ... from the NHLBI on Twitter. Types of Pulmonary Hypertension The World Health Organization divides pulmonary hypertension (PH) ... are called pulmonary hypertension.) Group 1 Pulmonary Arterial Hypertension Group 1 PAH includes: PAH that has no ...

  19. Pulmonary malaria: high-resolution computed tomography findings - a case report

    International Nuclear Information System (INIS)

    We report the case of a 38-year-old man with pulmonary malaria. High-resolution computed tomography showed thickening of the peribronchovascular interstitium and interlobular septa, areas of consolidation and ground glass attenuation and bilateral pleural effusion suggesting pulmonary edema. The patient recovered well after receiving specific treatment and was discharged after 11 days of hospitalization. (author)

  20. Pulmonary embolism

    International Nuclear Information System (INIS)

    Pulmonary embolism is a common medical problem whose incidence is likely to increase in our aging population. Although it is life-threatening, effective therapy exists. The treatment is not, however, without significant complications. Thus, accurate diagnosis is important. Unfortunately, the clinical manifestations of pulmonary embolism are nonspecific. Furthermore, in many patients the symptoms of an acute embolism are superimposed on underlying chronic heart or lung disease. Thus, a high index of suspicion is needed to identify pulmonary emboli. Laboratory parameters, including arterial oxygen tensions and electrocardiography, are as nonspecific as the clinical signs. They may be more useful in excluding another process than in diagnosing pulmonary embolism. The first radiologic examination is the chest radiograph, but the clinical symptoms are frequently out of proportion to the findings on the chest films. Classic manifestations of pulmonary embolism on the chest radiograph include a wedge-shaped peripheral opacity and a segmental or lobar diminution in vascularity with prominent central arteries. However, these findings are not commonly seen and, even when present, are not specific. Even less specific findings include cardiomegaly, pulmonary infiltrate, elevation of a hemidiaphragm, and pleural effusion. Many patients with pulmonary embolism may have a normal chest radiograph. The chest radiograph is essential, however, for two purposes. First, it may identify another cause of the patient's symptoms, such as a rib fracture, dissecting aortic aneurysm, or pneumothorax. Second, a chest radiograph is essential to interpretation of the radionuclide V/Q scan. The perfusion scan accurately reflects the perfusion of the lung. However, a perfusion defect may result from a variety of etiologies. Any process such as vascular stenosis or compression by tumor may restrict blood flow. 84 references

  1. Bilateral posterior cerebral artery infarction

    OpenAIRE

    Ryan, Davinia; Murphy, Sinead M; Hennessey, Michael J

    2010-01-01

    We report the case of a 70-year-old man who presented with short-term memory impairment and a homonymous left inferior quadrantanopia secondary to simultaneous bilateral posterior cerebral artery (PCA) territory infarction. As in more than a quarter of cases of PCA

  2. Primary synchronous bilateral breast cancer

    Directory of Open Access Journals (Sweden)

    R Krishnappa

    2014-01-01

    Full Text Available Background: Primary synchronous bilateral breast cancer (PSBBC is a rare clinical entity. The reported incidence ranges between 0.3% and 12%. There are several controversial issues regarding PSBBC pertaining to the diagnostic criteria, nomenclature, and management policies. Materials and Methods: Fourteen cases of PSBBC treated between 2001 to 2010 at our institute were retrospectively analysed in regards to demographic data, management and follow up. Results: PSBBC constituted 0.19% of total breast cancer patients at our institute. Age ranged from 28 to 78 years. PSBBC were detected by clinical examination in eight cases and by mammography in six cases. Twelve patients underwent bilateral modified radical mastectomy, one had unilateral mastectomy on one side and breast conservation on the other side and one patient has bilateral breast conservation. Majority of patients belonged to stage 2 and stage 3. All patients were found to have invasive ductal carcinoma. Five cases were ER/PR positive and 8 patients were triple hormone receptor negative. Eight patients received unilateral and six received bilateral adjuvant radiotherapy. Nine patients received adjuvant chemotherapy. 5 patients received adjuvant hormonal therapy. Median follow up of patients was 15.4 months. Conclusion: PSBBC is a rare event warranting awareness and screening of the contralateral breast in patients with unilateral breast cancer. These patients require individualized treatment planning based on the tumor factors of the index lesion. Further multi institutional prospective studies are needed for adequate understanding of management of PSBBC.

  3. Lymphatic Endothelial Differentiation in Pulmonary Lymphangioleiomyomatosis Cells

    OpenAIRE

    Davis, Jennifer M.; Hyjek, Elizabeth; Husain, Aliya N.; Shen, Le; Jones, Jennifer; Schuger, Lucia A.

    2013-01-01

    Pulmonary lymphangioleiomyomatosis (LAM) is a rare, low-grade neoplasm affecting almost exclusively women of childbearing age. LAM belongs to the family of perivascular epithelioid cell tumors, characterized by spindle and epithelioid cells with smooth muscle and melanocytic differentiation. LAM cells infiltrate the lungs, producing multiple, bilateral lesions rich in lymphatic channels and forming cysts, leading to respiratory insufficiency. Here we used antibodies against four lymphatic end...

  4. Pulmonary imaging

    International Nuclear Information System (INIS)

    Studies in pulmonary imaging predominantly depict the distribution of pulmonary perfusion or the ventilation-perfusion balance for the detection of pulmonary embolism, obstructive airway disease, and lung carcinoma. Much work has been done using 133Xe to study both regional ventilation and blood flow. This work has led to a greater understanding of regional blood flow and ventilation, of lung anatomy and morphometry, of the mechanical factors that influence lung function, and of the biochemical and other nonrespiratory functions of the lung. The recent development of emission tomography and the potential for receptor- and function-oriented radiopharmaceuticals have allowed several important regional physiologic processes to be measured. The physical parameters measured in radionuclide emission studies are the temporal and spatial relations of the distribution of tracer in the lungs. Tracers that are diffusible will wash into and out of the lungs at a rate proportional not only to flow but also to membrane permeability. If the radionuclide label is attached to microspheres or macro-aggregates, the amount lodging in any portion of the lung can be related to relative flow in the lung. Metabolism of the lung is reflected by the rate of extraction and disappearance of various labeled radiopharmaceuticals. The problems encountered in assessing the value of ventilation-perfusion scans for the diagnosis of pulmonary embolism are concerned not only with improving specificity, but also with better estimation of the prior probability of pulmonary embolism in the patient being evaluated

  5. Whole lung lavage for pulmonary alveolar proteinosis

    Directory of Open Access Journals (Sweden)

    Jayaraman S

    2010-01-01

    Full Text Available A 26-year-old male presented with complaints of dry cough of six months and progressive breathlessness of three months duration. He was coughing out milky white sputum for two months and had lost 12 kg weight in two months. He had an evening rise in temperature of one month duration. Clinically, the patient was in respiratory distress and the respiratory system examination revealed bilateral velcro crackles. High resolution computed tomography chest showed bilateral diffuse reticulonodular opacities and "Crazy Paving" pattern suggestive of alveolar proteinosis. Broncho alveolar lavage showed eosinophilic granular material, which was periodic acid-Schiff positive. Open lung biopsy was done to confirm the diagnosis and the histopathologic examination revealed eosinophilic secretions with granular appearance suggestive of pulmonary alveolar proteinosis. Subsequently, patient underwent bilateral sequential whole lung lavage under general anesthesia. Patient showed marked clinical and radiological improvement after sequential whole lung lavage.

  6. Infectious mononucleosis presenting as bilateral acute dacryocystitis.

    OpenAIRE

    Atkinson, P L; Ansons, A M; Patterson, A.

    1990-01-01

    A case of infectious mononucleosis presenting as bilateral acute dacryocystitis in a 7-year-old girl is reported. Acute dacryocystitis is uncommon in this age group, and an underlying systemic illness should be suspected particularly when it is bilateral.

  7. Role of treatment for solitary pulmonary nodule in breast cancer patients

    OpenAIRE

    Miyokawa Naoyuki; Hayashi Satoshi; Matsuda Yoshinari; Sato Kazuhiro; Kitada Masahiro; Sasajima Tadahiro

    2011-01-01

    Abstract Background Metastatic pulmonary tumors secondary to breast cancer detected either before or after surgery are predominantly multiple and bilateral. However, in cases detected to have a solitary pulmonary nodule (SPN), determining whether the lesion represents a primary cancer, metastasis, or a benign pulmonary lesion can be difficult. Materials and methods Between January 2000 and December 2009, we performed breast cancer surgery on 1,226 patients, of which 49 cases (3.9%) were detec...

  8. Amyand’s hernia in a child with permanent neonatal diabetes due to pancreatic agenesis.

    Directory of Open Access Journals (Sweden)

    Alessandro Giannattasio

    2009-11-01

    Full Text Available Acute or perforated appendicitis within inguinal hernia is rarely encountered and it is known as Amyand’s hernia. We report on the first case occurring in a 4-year-old boy affected by permanent neonatal diabetes mellitus due to pancreatic agenesis, an extremely rare condition. The initial suspicion of inguinal hernia was confirmed by ultrasound examination of the right inguinal region which revealed omental layers inside a swollen inguinal canal; this finding and the clinical presentation allowed a prompt and appropriate surgical management. The careful evaluation of this patient and early recognition of this unique presentation of appendicitis allowed trans-hernial appendectomy and immediate herniorrhaphy. Ultrasonography played a pivotal role to reach the correct diagnosis and to start a prompt treatment.

  9. Agenesis of the corpus callosum: genetic, developmental and functional aspects of connectivity.

    Science.gov (United States)

    Paul, Lynn K; Brown, Warren S; Adolphs, Ralph; Tyszka, J Michael; Richards, Linda J; Mukherjee, Pratik; Sherr, Elliott H

    2007-04-01

    Agenesis of the corpus callosum (AgCC), a failure to develop the large bundle of fibres that connect the cerebral hemispheres, occurs in 1:4000 individuals. Genetics, animal models and detailed structural neuroimaging are now providing insights into the developmental and molecular bases of AgCC. Studies using neuropsychological, electroencephalogram and functional MRI approaches are examining the resulting impairments in emotional and social functioning, and have begun to explore the functional neuroanatomy underlying impaired higher-order cognition. The study of AgCC could provide insight into the integrated cerebral functioning of healthy brains, and may offer a model for understanding certain psychiatric illnesses, such as schizophrenia and autism. PMID:17375041

  10. Hymen Tearing in the Treatment of Vaginal Agenesis: Implications on Women in Turkey.

    Science.gov (United States)

    Yaman, Sengül; Ayaz, Sultan

    2014-02-11

    Rupture of the hymen before marriage is a significant problem for a woman and her family. The purpose of this article is to present a case report where hymen membrane perforation was required for vaginal agenesis in a 13-year-old adolescent, identify psychosocial issues related to the torn hymen in the Turkish culture, discuss gender inequality affecting health care of women with a torn hymen, identify the impact of a torn hymen on a Turkish woman's social status, and discuss health care implications for the woman and her family. The girl could not understand the situation and experienced feelings of guilt. The parents displayed an intense reaction of shock and denial after being informed about the disorder and treatment process and felt a sense of hopelessness about the future of their daughter. Nurses are expected to fulfill their training, counseling, and advocacy roles about this issue. PMID:24518059

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

    Directory of Open Access Journals (Sweden)

    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.

  12. An incidental finding of unicornuate uterus with unilateral ovarian agenesis during laparoscopy in patient who gave birth to eleven children: a case report.

    Science.gov (United States)

    Alvir, Ilija; Puljiz, Mario; Tomica, Darko; Danolić, Damir; Mamić, Ivica; Toth, Tibor

    2013-03-01

    Congenital uterine anomalies are often asymptomatic. They may present with infertility, recurrent miscarriage, preterm delivery, abnormal lie in pregnancy and other obstetric complications. We report the case of a 38-year old patient with unicornuate uterus without rudimentary horn and with unilateral left ovarian agenesis and unilateral left renal agenesis who gave birth to eleven children. Anomaly was incidentally diagnosed during laparoscopic sterilization. PMID:23697286

  13. Bilateral ovarian tumour in a young girl

    OpenAIRE

    2013-01-01

    Bilateral ovarian tumour in a girl presents the dilemma of conservative versus aggressive approach towards these tumours. When faced with suspicious tumour and complete replacement of the ovaries bilaterally, bilateral oophorectomy is a viable option, though the certain possibility of infertility and lifelong hormonal supplementation is unavoidable. We report a case of bilateral ovarian masses in a young girl, which on histopathological examination showed mature teratoma with aggregates of pr...

  14. Laparoscopic Adrenalectomy for Bilateral Metachronous Aldosteronomas

    OpenAIRE

    Rizek, Philippe; Gorecki, Piotr; Lindenmayer, Aristid; Moktan, Sabita

    2011-01-01

    Introduction: Primary aldosteronism affects 5% to 13% of patients with hypertension. Idiopathic bilateral hyperplasia (IHA) and unilateral aldosterone-producing adenoma (APA) are the most common types of primary aldosteronism. Bilateral APA is a very rare entity with only a few reports in the literature. We present the case of a patient with metachronous bilateral APA treated with metachronous bilateral total and near total adrenalectomy. Case Report: A 66-year-old female was evaluated for hy...

  15. Tratamiento conservador en pacientes con retinoblastoma bilateral

    OpenAIRE

    Juan C. Suárez; Mabel C. Ospina; Sandra A. Arias; González, María E

    2008-01-01

    OBJETIVO: comparar el tratamiento convencional del retinoblastoma bilateral, usado hasta hace algunos años, consistente en radioterapia o enucleación bilateral, con el tratamiento conservador actual que incluye termoterapia transpupilar (TTT) o TTT/quimioterapia al menos en un ojo, en niños con diagnóstico de retinoblastoma bilateral. DISEÑO: estudio retrospectivo descriptivo. MUESTRA: 20 pacientes con diagnóstico de retinoblastoma bilateral que consultaron al Hospital Universitario San Vicen...

  16. Simultaneous and staged bilateral total hip arthroplasty

    DEFF Research Database (Denmark)

    Lindberg-Larsen, Martin; Joergensen, Christoffer Calov; Husted, Henrik;

    2013-01-01

    Bilateral total hip arthroplasty (BTHA) and bilateral simultaneous total hip arthroplasty (BSTHA) are done increasingly. Previous studies evaluating outcomes after bilateral procedures have found different results. The aim of this study was to investigate length of hospital stay (LOS), 30 days...

  17. Development of bilateral coronary artery aneurysms in a child with Noonan syndrome

    International Nuclear Information System (INIS)

    Noonan syndrome is a constellation of congenital malformations including heart defects, facial anomalies and short stature. The cardiovascular defects are variable and extensive, with the most common being pulmonary stenosis and hypertrophic cardiomyopathy. Coronary artery anomalies have only been reported in a few cases. We report a child with Noonan syndrome status post pulmonary stenosis and atrial septal defect repair, who developed bilateral coronary artery aneurysms. The aneurysms were diagnosed with both cardiac magnetic resonance imaging and coronary computed tomography angiography. There had been no evidence of them on a cardiac MR exam 5 years previously. (orig.)

  18. Development of bilateral coronary artery aneurysms in a child with Noonan syndrome

    Energy Technology Data Exchange (ETDEWEB)

    Mauro, David M.; Flors, Lucia; Norton, Patrick T.; Hagspiel, Klaus D. [University of Virginia Health System, Department of Radiology and Medical Imaging, Charlottesville, VA (United States); Hoyer, Andrew W. [University of Virginia Health System, Department of Pediatrics, Division of Pediatric Cardiology, Charlottesville, VA (United States); Pediatric Cardiology Center of Oregon, Portland, OR (United States)

    2016-03-15

    Noonan syndrome is a constellation of congenital malformations including heart defects, facial anomalies and short stature. The cardiovascular defects are variable and extensive, with the most common being pulmonary stenosis and hypertrophic cardiomyopathy. Coronary artery anomalies have only been reported in a few cases. We report a child with Noonan syndrome status post pulmonary stenosis and atrial septal defect repair, who developed bilateral coronary artery aneurysms. The aneurysms were diagnosed with both cardiac magnetic resonance imaging and coronary computed tomography angiography. There had been no evidence of them on a cardiac MR exam 5 years previously. (orig.)

  19. Development of bilateral coronary artery aneurysms in a child with Noonan syndrome.

    Science.gov (United States)

    Mauro, David M; Flors, Lucia; Hoyer, Andrew W; Norton, Patrick T; Hagspiel, Klaus D

    2016-03-01

    Noonan syndrome is a constellation of congenital malformations including heart defects, facial anomalies and short stature. The cardiovascular defects are variable and extensive, with the most common being pulmonary stenosis and hypertrophic cardiomyopathy. Coronary artery anomalies have only been reported in a few cases. We report a child with Noonan syndrome status post pulmonary stenosis and atrial septal defect repair, who developed bilateral coronary artery aneurysms. The aneurysms were diagnosed with both cardiac magnetic resonance imaging and coronary computed tomography angiography. There had been no evidence of them on a cardiac MR exam 5 years previously. PMID:26515448

  20. Bilateral posterior cerebral artery infarction.

    Science.gov (United States)

    Ryan, Davinia; Murphy, Sinead M; Hennessey, Michael J

    2010-01-01

    We report the case of a 70-year-old man who presented with short-term memory impairment and a homonymous left inferior quadrantanopia secondary to simultaneous bilateral posterior cerebral artery (PCA) territory infarction. As in more than a quarter of cases of PCA infarction, no aetiological cause was identified. Unlike the transient nature of symptoms in some cases following unilateral infarction, his deficits persisted on 2-month follow-up. PMID:22798298

  1. China's Bilateral Currency Swap Lines

    OpenAIRE

    Zhitao, Lin; Wenjie, Zhan; Cheung, Yin-Wong

    2016-01-01

    We study the determinants of China’s bilateral local currency swap lines that were established since the recent global finance crisis. It is found that economic factors, political considerations, and institutional characteristics including trade intensity, economic size, strategic partnership, free trade agreement, corruption, and stability affect the decision of signing a swap line agreement. Once a swap line agreement decision is made, the size of the swap line is then mainly affected by tr...

  2. Bilateral monopolies and location choice

    OpenAIRE

    Brekke, Kurt R.; Straume, Odd Rune

    2002-01-01

    We analyse how equilibrium locations in location-price games à la Hotelling are affected when firms acquire inputs through bilateral monopoly relations with suppliers. Assuming a duopoly downstream market, we consider the case of two independent input suppliers bargaining with both downstream firms. We find that the presence of input suppliers changes the locational incentives of downstream firms in several ways, compared with the case of exogenous production costs. Bargaining induces downstr...

  3. Bilateral Molariform Mandibular Second Premolars

    OpenAIRE

    Sonu Acharya; Pradip Kumar Mandal; Chiranjit Ghosh

    2015-01-01

    Macrodontia is a rare dental anomaly that refers to teeth that appear larger than normal. Generalised macrodontia can be associated with certain medical conditions and syndromes. This case report presents clinical and radiographic findings of isolated bilateral macrodontia in a 14-year-old child. The patient was referred to the clinic with local crowding of maxillary and mandibular teeth. Radiographic findings revealed the presence of impacted macrodont mandibular second premolar on one side ...

  4. Pulmonary histiocytosis

    Directory of Open Access Journals (Sweden)

    Milenković Branislava

    2013-01-01

    Full Text Available Introduction. Langerhans cell histiocytosis encompasses a group of disorders of unknown origin with different clinical presentations and outcomes. It is characterized by infiltration of the involved tissues by large numbers of Langerhans cells, often organized into granulomas. The accumulation of these cells causes the classic lytic bone lesions, skin rashes, lymphadenopathy, splenomegaly, and dysfunction of organ such as the pituitary gland, lungs, liver, and bone marrow. Pulmonary histiocytosis. Adult pulmonary Langerhans cell histiocytosis is a rare disorder of unknown etiology. It occurs predominantly in male smokers, with an incidence peak between 20 and 40 years of age. High-resolution computed tomography of the chest can show nodules, cavitated nodules, and thickand thin-walled cysts. The definite diagnosis of pulmonary Langerhans cell histiocytosis requires identification of Langerhans’ cell granulomas infiltrating and destroying distal bronchioles, which is usually achieved by lung biopsy at a site selected by chest high-resolution computed tomography. Treatment. Treatment options for adults have never been clarified by a clinical trial. The published literature provides minimal data on the comparative efficacy of various treatment options which include surgery/curettage, steroids, radiation, and various chemotherapy regimens. The improved understanding of the mechanisms involved in the pathogenesis of pulmonary Langerhans cell histiocytosis should help in the development of specific therapeutic strategies and effective treatment.

  5. Bilateral Pleural Effusions as an Initial Presentation in Primary Sjögren’s Syndrome

    Directory of Open Access Journals (Sweden)

    Go Makimoto

    2012-01-01

    Full Text Available Sjögren’s syndrome (SS is a systemic autoimmune disease characterized by sicca symptoms. Interstitial pulmonary fibrosis and tracheobronchial sicca are the most common symptoms of pulmonary involvement in primary SjS, and they are rarely accompanied by serositis such as pleuritis or pericarditis. We report a case of SS presenting initially with bilateral pleural effusions. A 63-year old man was admitted to our hospital with a one-month history of cough, dyspnea, and right chest pain. Chest-computed tomography revealed bilateral pleural effusions. Serum anti-SS-A antibody titer was 1 : 256. Ophthalmological examination revealed a positive Schirmer test. Lip biopsy showed atrophy and plasmacytic infiltration of the salivary gland. Corticosteroid treatment was initiated. Pleural effusions were almost completely resolved by day 30. The patient has not experienced any recurrence.

  6. A fatal case of pulmonary nocardiosis

    Science.gov (United States)

    Patil, Mahantesh; C, Shivaprasad; Varghese, Jaicob; Rajagopalan, Natarajan

    2012-01-01

    Pulmonary nocardiosis is a serious, most often considered an opportunistic infection affecting the respiratory tract. Even though it is more common in immunocompromised hosts, it is not infrequently seen in immunocompetent patients as well. The aerosol route is the main portal of entry in to the body. Molecular techniques have revolutionised the identification of Nocardia species. However such tests are limited to referral laboratories. The radiographic appearances of Nocardia infection vary from a small nodule to bilateral infiltrates with cavitation. Traditionally sulphonamides have been considered the treatment of choice. However, resistance to sulphonamides is increasingly recognised. Carbapenems and linezolid have been found to be uniformly active against all the pathogenic species of Nocardia that affect human beings. The authors report a case of pulmonary nocardiosis in an immunocompetent patient, in whom the infection relentlessly progressed to florid sepsis despite prompt institution of right antibiotics. Florid sepsis relating to pulmonary nocardiosis is rare. PMID:22665550

  7. Complex malformations of the urogenital tract in a female dog: Gartner duct cyst, ipsilateral renal agenesis, and ipsilateral hydrometra.

    Science.gov (United States)

    Fujita, Atsushi; Tsuboi, Masaya; Uchida, Kazuyuki; Nishimura, Ryohei

    2016-05-01

    A 10-month-old female toy poodle was referred to the University of Tokyo Veterinary Medical Center with a urogenital anomaly found during sterilization. An exploratory laparotomy revealed a cyst adhering to the cervix and a unilateral renal agenesis. Histopathology and immunohistochemical analysis of the cyst was consistent with remnants of the Wolffian duct or a Gartner duct cyst. This is a rare case of a canine Gartner duct cyst with renal agenesis and uterine anomaly. We discuss the similarity of this case to that of humans and introduce a classification in the literature for these complex urogenital malformations for further clinical research into the precise diagnosis and appropriate surgical planning. PMID:27506089

  8.  Uterus Didelphys with Obstructed Right Hemivagina, Ipsilateral Renal Agenesis and Right Pyocolpos: A Case Report

    Directory of Open Access Journals (Sweden)

    Hansa Dhar

    2011-11-01

    Full Text Available  Uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis (OHVIRA Syndrome is a rare congenital anomaly of the female genital tract. Uterus didelphys occurs when the midline fusion of the mullerian ducts is arrested, either completely or incompletely. Women with didelphic uterus may be asymptomatic and unaware of having a double uterus. They may present with complaints of dysmenorrhoea and dyspareunia. A 25 year old woman attending the infertility clinic at Nizwa regional referral hospital presented with history of dysmenorrhoea and foul vaginal discharge with right cystic pelvic mass. She was diagnosed as a case of double uterus with obstructed right hemivagina and right pyocolpos with ipsilateral renal agenesis after routine ultrasonography in the clinic followed by MRI. Excision of the right vaginal septum with drainage of 200 ml of purulent discharge was performed. She was relieved of her symptoms and conceived promptly after the surgical excision of the partial vaginal septum.

  9.  Uterus Didelphys with Obstructed Right Hemivagina, Ipsilateral Renal Agenesis and Right Pyocolpos: A Case Report

    OpenAIRE

    Yasser A Razek; Ilham Hamdi; Hansa Dhar

    2011-01-01

     Uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis (OHVIRA Syndrome) is a rare congenital anomaly of the female genital tract. Uterus didelphys occurs when the midline fusion of the mullerian ducts is arrested, either completely or incompletely. Women with didelphic uterus may be asymptomatic and unaware of having a double uterus. They may present with complaints of dysmenorrhoea and dyspareunia. A 25 year old woman attending the infertility clinic at Nizwa regional r...

  10. Agenesis and Dysgenesis of the Corpus Callosum: Clinical, Genetic and Neuroimaging Findings in a Series of 41 Patients

    OpenAIRE

    Schell-Apacik, Chayim Can; Wagner, Kristina; Bihler, Moritz; Ertl-Wagner, Birgit; Heinrich, Uwe; Klopocki, Eva; Kalscheuer, Vera M.; Muenke, Maximilian; von Voss, Hubertus

    2008-01-01

    Agenesis of the corpus callosum (ACC) is among the most frequent human brain malformations with an incidence of 0.5–70 in 10,000. It is a heterogeneous condition, for which several different genetic causes are known, for example, ACC as part of monogenic syndromes or complex chromosomal rearrangements. We systematically evaluated the data of 172 patients with documented corpus callosum abnormalities in the records, and 23 patients with chromosomal rearrangements known to be associated with co...

  11. "Recurrent Bilateral Spontaneous Pneumothorax in Early Infancy: A Case of Langerhans Cell Histiocytosis"

    Directory of Open Access Journals (Sweden)

    S. Alavi

    2005-07-01

    Full Text Available Langerhans cell histiocytosis (LCH is a rare disorder characterized by infiltration of either single or multiple organs by S100 and CD1a positive cells. Patients with pulmonary LCH are predisposed to pneumothorax due to destructive changes in the lung parenchyma. Here, we report a case of multisystem LCH who presented at 2 months of age with simultaneous bilateral spontaneous pneumothorax.

  12. Unsuspected multiples myeloma presenting as bilateral pleural effusion – a cytological diagnosis

    OpenAIRE

    Dhingra Kajal; Singhal Niti; Nigam Sonu; Jain Shayama

    2007-01-01

    Abstract Background Multiple Myeloma presenting as a pleural effusion is extremely rare. It is usually a late complication and is associated with a poor prognosis. Case Presentation A 40-year-old male presented with dyspnea and fever of six months duration. Clinical diagnosis of pulmonary tuberculosis was considered. X-ray chest showed bilateral pleural effusion. Pleural cytology revealed numerous plasma cells, some of which were binucleated and atypical. Cytological differential diagnosis in...

  13. "Possible TRALI" developed during bilateral total knee arthroplasty replacement -A case report-

    OpenAIRE

    Kim, Youn Jin; Heo, Seok

    2012-01-01

    Transfusion-related acute lung injury (TRALI) is one of the leading causes of transfusion-related morbidity and mortality. However, it is frequently not diagnosed and under-reported, which could result in inappropriate treatment. Diagnostic definition for TRALI consists of hypoxia and bilateral pulmonary edema occurring during or within 6 hours of a transfusion in the absence of cardiac failure or intravascular volume overload. Here, we report a fatal case, which resulted from under-recogniti...

  14. Subxiphoid single-incision thoracoscopic surgery for bilateral primary spontaneous pneumothorax

    OpenAIRE

    Liu, Chao-Yu; Lin, Chen-Sung; Liu, Chia-Chuan

    2015-01-01

    It has been reported that single-incision thoracoscopic surgery can reduce postoperative pain without compromising the main surgical steps required for treating patients affected by primary spontaneous pneumothorax. However, all the reported thoracoscopic surgery cases with a single-incision procedure were via the intercostal route for unilateral pulmonary lesions. We present a novel single-incision thoracoscopic technique via a subxiphoid route to perform one-stage bilateral thoracoscopic su...

  15. Significance of diffuse pulmonary uptake in radiogallium scans: concise communication

    International Nuclear Information System (INIS)

    Diffuse pulmonary uptake of radiogallium was observed in 50 out of 510 scans (9.8%) performed in a general hospital over a period of 1 y. Of the 50 cases, 80% had bilateral, diffuse distribution, and 20% unilateral. A variety of clinical conditions produced a similar picture on the pulmonary images. The most common cause of the diffuse uptake was infectious disease (48%) followed by infiltrative disorders (30%) and neoplastic conditions (22%). On a repeat study there was significant reduction in the intensity of pulmonary radiogallium uptake following drug therapy in three patients - sarcoidosis on corticosteroid therapy, pneumocystis carinii treated with trimethoprim and sulfamethoxozole, and interstitial pulmonary fibrosis on corticosteroids. No close correlation was observed between the underlying clinical disorder and the pattern or intensity of pulmonary uptake of radiogallium

  16. Demonstration of chronic recurrent pulmonary emboli with spiral CT

    International Nuclear Information System (INIS)

    Chronic recurrent pulmonary emboli may lead, in rare cases, to progressive pulmonary arterial hypertension which cannot be treated medically. These patients may be helped by bilateral pulmonary thrombenarterectomy. The value of spiral CT in the diagnosis of this condition was demonstrated in 31 patients. In 12 patients, thrombi could be shown to be cause of the pulmonary arterial hypertension. Indirect criteria were vascular occlusions, changes in calibre and mural irregularities which were shown in varying degrees in all patients. In 22 of 31 patients there was non-homogeneity of perfusion with greater opacification of the perfused lung parenchyma. In 11 patients scars related to pleural surfaces were seen. Typical changes of right heart insufficiency (cardiomegaly, widened central pulmonary arteries) were shown in all patients by CT. (orig.)

  17. [Immunodepression and pulmonary infections].

    Science.gov (United States)

    Yao, N A; Ngoran, N; de Jaureguiberry, J P; Bérard, H; Jaubert, D

    2002-11-01

    The acquired immunosuppressed states are increasingly numerous. Pneumopathies are a frequent, serious complication and etiologic diagnosis is often difficult. The nature of the micro-organism in question is a function of the immunizing type of deficiency. In neutropenias, the infections are primarily bacterial, their potential gravity being correlated with the depth of the deficiency into polynuclear, or fungic, especially in prolonged neutropenias. The aspleened states are responsible for a deficit of the macrophage system and contribute to the infections with encapsulated germs (pneumococci, klebsiellas...). The organic grafts imply an attack of cell-mediated immunity, in the particular case of the auxiliary T lymphocytes (CD4)), with a special predisposition for viral and fungic infections. During VIH infection, the immunizing deficit of CD4 lymphocytes worsens with time. At the early stage, the infections are especially bacterial. At the more advanced stages, the pulmonary pneumocystosis and tuberculosis dominate. At the late stage, finally, deep immunosuppression allows emerging of the atypical mycobacteries. In the deficiencies of humoral immunity (congenital hypogammaglobulinemias, lymphoid hemopathies B), the germs to be mentioned are the pneumococcus, Haemophilus influenzae, the salmonellas and the legionellas. Immunosuppressed pneumopathies are characterized by radio-clinical pictures of very variable gravity, ranging from focused acute pneumopathy to bilateral diffuse pneumopathy with acute respiratory distress syndrome, with phases of atypical tables with respiratory symptomatology larval or absent. The highlighting of the micro-organisms in question requires urgent complementary investigations: hemocultures, bronchiolo-alveolar washing. In certain cases, it will be possible to resort to the transtracheal puncture or transthoracic puncture guided by tomodensitometry, and if necessary to pulmonary biopsy under videothoracoscopy. Emergency of the anti

  18. Laparoscopic Bilateral Adrenalectomy in a patient of Cushing syndrome: A Challenge for the Anaesthesiologist

    Directory of Open Access Journals (Sweden)

    Uma K Dahanukar

    2007-01-01

    Full Text Available We present a case of Cushing syndrome who underwent laparoscopic bilateral adrenalectomy and discuss her intraoperative management and postoperative course in ICU, especially pulmonary oedema, that occurred within 3 hours after resection (half life of cortisol is 80-110 minutes. [1] She was diagnosed to have bilateral adrenal hyperplasia with no pituitary involvement on CT scan. Preoperative workup revealed hypokalemia, anaemia, hypertension and hyperglycemia. She was posted for laparoscopic bilateral adrenalectomy. She received general anaesthesia; we did not give epidural analgesia as the patient had fracture of body of L1 vertebrae. Her intra-operative course was uneventful. Post-operative concerns included acute adrenal insufficiency, hypoglycaemia, hypotension and hyperkalemia, which were successfully managed in ICU. Patient was then given oral corticosteroids. One month later she was reassessed and was in better health.

  19. Bilateral ECT induces bilateral increases in regional cortical thickness.

    Science.gov (United States)

    van Eijndhoven, P; Mulders, P; Kwekkeboom, L; van Oostrom, I; van Beek, M; Janzing, J; Schene, A; Tendolkar, I

    2016-01-01

    Electroconvulsive therapy (ECT) is the most effective treatment for patients suffering from severe or treatment-resistant major depressive disorder (MDD). Unfortunately its underlying neurobiological mechanisms are still unclear. One line of evidence indicates that the seizures produced by ECT induce or stimulate neuroplasticity effects. Although these seizures also affect the cortex, the effect of ECT on cortical thickness is not investigated until now. We acquired structural magnetic resonance imaging data in 19 treatment-resistant MDD patients before and after a bilateral ECT course, and 16 healthy controls at 2 time points, and compared changes in cortical thickness between the groups. Our results reveal that ECT induces significant, bilateral increases in cortical thickness, including the temporal pole, inferior and middle temporal cortex and the insula. The pattern of increased cortical thickness was predominant in regions that are associated with seizure onset in ECT. Post hoc analyses showed that the increase in thickness of the insular cortex was larger in responders than in non-responders, which may point to a specific relationship of this region with treatment effects of ECT. PMID:27552587

  20. Pulmonary aspergillosis.

    Science.gov (United States)

    Chabi, M L; Goracci, A; Roche, N; Paugam, A; Lupo, A; Revel, M P

    2015-05-01

    Aspergillosis is a mycotic disease usually caused by Aspergillus fumigatus, a saprophytic and ubiquitous airborne fungus. Aspergillus-related lung diseases are traditionally classified into four different forms, whose occurrence depends on the immunologic status of the host and the existence of an underlying lung disease. Allergic broncho-pulmonary aspergillosis (ABPA) affects patients with asthma or cystic fibrosis. Saprophytic infection (aspergilloma) occurs in patients with abnormal airways (chronic obstructive pulmonary disease, bronchiectasis, cystic fibrosis) or chronic lung cavities. Chronic necrotizing aspergillosis (semi-invasive form) is described in patients with chronic lung pathology or mild immunodeficiency. Invasive aspergillosis (angio-invasive or broncho-invasive forms) occurs in severely immuno-compromised patients. Knowledge of the various radiological patterns for each form, as well as the corresponding associated immune disorders and/or underlying lung diseases, helps early recognition and accurate diagnosis. PMID:25753544

  1. Bilateral cleft lip nasal deformity

    Directory of Open Access Journals (Sweden)

    Singh Arun

    2009-01-01

    Full Text Available Bilateral cleft lip nose deformity is a multi-factorial and complex deformity which tends to aggravate with growth of the child, if not attended surgically. The goals of primary bilateral cleft lip nose surgery are, closure of the nasal floor and sill, lengthening of the columella, repositioning of the alar base, achieving nasal tip projection, repositioning of the lower lateral cartilages, and reorienting the nares from horizontal to oblique position. The multiplicity of procedures in the literature for correction of this deformity alludes to the fact that no single procedure is entirely effective. The timing for surgical intervention and its extent varies considerably. Early surgery on cartilage may adversely affect growth and development; at the same time, allowing the cartilage to grow in an abnormal position and contributing to aggravation of deformity. Some surgeons advocate correction of deformity at an early age. However, others like the cartilages to grow and mature before going in for surgery. With peer pressure also becoming an important consideration during the teens, the current trend is towards early intervention. There is no unanimity in the extent of nasal dissection to be done at the time of primary lip repair. While many perform limited nasal dissection for the fear of growth retardation, others opt for full cartilage correction at the time of primary surgery itself. The value of naso-alveolar moulding (NAM too is not universally accepted and has now more opponents than proponents. Also most centres in the developing world have neither the personnel nor the facilities for the same. The secondary cleft nasal deformity is variable and is affected by the extent of the original abnormality, any prior surgeries performed and alteration due to nasal growth. This article reviews the currently popular methods for correction of nasal deformity associated with bilateral cleft lip, it′s management both at the time of cleft lip repair

  2. Bart’s Syndrome Associated Corpus Callosum Agenesis and Choanal Atresia

    Directory of Open Access Journals (Sweden)

    Muhammad SAEED*

    2014-12-01

    Full Text Available How to Cite This Article: Saeed M, Haq A, Qadir Kh.Bart’s Syndrome Associated Corpus Callosum Agenesis and Choanal Atresia. Iran J Child Neurol. 2014 Autumn;8(4: 76-79.AbstractObjectiveBart’s syndrome is defined as congenital localized absence of skin, and associated with epidermolysis bullosa. A newborn with Bart’s syndrome is reported because it is a very rare condition, especially when associated with corpus callosum agenesis and concomitant choanal atresia. Clinically it is characterized by raw beefy areas of denuded skin mainly on hands and feet.We report a rare case of a term female newborn born to non-consanguineous parents who presented with congenital absence of skin in, face, trunk and extremities. To the best of our knowledge, this is the first report presenting a case of Bart’s syndrome associated with corpus callosum agenesis.ReferencesBart BJ, Garlin RJ, Anderson VE, Lynch FW. Congenital localized absence of skin and associated abnormalities resembling epidermolysis bullosa. A new syndrome. Arch Dermatol 1966; 93: 296-304.Bart BJ. Epidermolysis bullosa and congenital localized absence of skin. Arch Dermatol 1970; 101: 78-81.Skoven I, Drzewiecki KT. Congenital localized skin defect and epidermolysis bullosa hereditaria letalis. Acta Derm Venereol 1979; 59: 533-537.Wojnarowska FT, Eady RA, Wells RS. Dystrophic epidermolysis bullosa presenting with congenital localized absence of skin: report of four cases. Br J Dermatol 1983; 108: 477-483.Kanzler MH, Smoller B, Woodley DT. Congenital localized absence of the skin as a manifestation of epidermolysis bullosa. Arch Dermatol 1992; 128:1087-90.Maman E, Maor E, Kachko L, Carmi R. Epidermolysis bullosa, pyloric atresia, aplasia cutis congenita: histopathological delineation of an autosomal recessive disease. Am J Med Genet 1998; 78: 127-133.McCarthy MA, Clarke T, Powell FC. Epidermolysis bullosa and aplasia cutis. Int J Derm 1991; 30: 481-484.Puvabanditsin S, Garrow E, Daeun K

  3. Pulmonary Hypertension

    OpenAIRE

    Newman, John H.

    2005-01-01

    The modern era in cardiopulmonary medicine began in the 1940s, when Cournand and Richards pioneered right-heart catheterization. Until that time, no direct measurement of central vascular pressure had been performed in humans. Right-heart catheterization ignited an explosion of insights into function and dysfunction of the pulmonary circulation, cardiac performance, ventilation–perfusion relationships, lung–heart interactions, valvular function, and congenital heart disease. It marked the beg...

  4. BILATERAL OVARIAN MASS COMPLICATING PREGNANCY

    OpenAIRE

    Arjumand; Triveni

    2015-01-01

    A 22 years old patient with 9 months of Amenorrhoea with a abdominal swelling was admitted to our institution with an Ultrasonography report of Bilateral ovarian mass (14.3x5.9cm in left ovary) and (9.5x7cm in right ovary) with internal vascularity with septations along with single live intrauterine fetus. She delivered by elective caesarean section in V/O breech presenta tion. Left side ovarian cystectomy done Right side ovarian puncture done. Histopathology showed left sid...

  5. Immediate Sequential Bilateral Cataract Surgery

    DEFF Research Database (Denmark)

    Kessel, Line; Andresen, Jens; Erngaard, Ditte; Flesner, Per; Tendal, Britta; Hjortdal, Jesper

    2015-01-01

    performed using the Cochrane Review Manager software. The quality of the evidence was assessed using the GRADE method (Grading of Recommendation, Assessment, Development, and Evaluation). We did not find any difference in the risk of complications or visual outcome in patients randomized to ISBCS or surgery......-based national Danish guidelines for cataract surgery. A systematic literature review in PubMed, Embase, and Cochrane central databases identified three randomized controlled trials that compared outcome in patients randomized to ISBCS or bilateral cataract surgery on two different dates. Meta-analyses were...

  6. Bilateral molariform mandibular second premolars.

    Science.gov (United States)

    Acharya, Sonu; Kumar Mandal, Pradip; Ghosh, Chiranjit

    2015-01-01

    Macrodontia is a rare dental anomaly that refers to teeth that appear larger than normal. Generalised macrodontia can be associated with certain medical conditions and syndromes. This case report presents clinical and radiographic findings of isolated bilateral macrodontia in a 14-year-old child. The patient was referred to the clinic with local crowding of maxillary and mandibular teeth. Radiographic findings revealed the presence of impacted macrodont mandibular second premolar on one side and erupted macrodontic premolar on the other side and their distinct morphological appearance, characterized by large, multitubercular, and molariform crowns and tapering, single roots. PMID:25685564

  7. What Causes Pulmonary Hypertension?

    Science.gov (United States)

    ... from the NHLBI on Twitter. What Causes Pulmonary Hypertension? Pulmonary hypertension (PH) begins with inflammation and changes in the ... different types of PH. Group 1 pulmonary arterial hypertension (PAH) may have no known cause, or the ...

  8. Pulmonary Arterial Hypertension

    Science.gov (United States)

    Pulmonary Arterial Hypertension What Is Pulmonary Hypertension? To understand pulmonary hypertension (PH) it helps to understand how blood ows throughout your body. While the heart is one organ, it ...

  9. Pulmonary manifestations of malaria

    International Nuclear Information System (INIS)

    We report on the two different types of pulmonary manifestations in acute plasmodium falciparum malaria. The more severe variant shows long standing interstitial pulmonary infiltrates, whereas in the more benign courses only short-term pulmonary edemas are visible. (orig.)

  10. Pulmonary Fibrosis Foundation

    Science.gov (United States)

    ... most current news and updates from the Pulmonary Fibrosis Foundation. Life with PF Education & Support About PF ... LEARN MORE We Imagine a World Without Pulmonary Fibrosis The Pulmonary Fibrosis Foundation mobilizes people and resources ...

  11. Pulmonary manifestations of malaria

    Energy Technology Data Exchange (ETDEWEB)

    Rauber, K.; Enkerlin, H.L.; Riemann, H.; Schoeppe, W.

    1987-05-01

    We report on the two different types of pulmonary manifestations in acute plasmodium falciparum malaria. The more severe variant shows long standing interstitial pulmonary infiltrates, whereas in the more benign courses only short-term pulmonary edemas are visible.

  12. Idiopathic Pulmonary Fibrosis

    Science.gov (United States)

    ... Explore Idiopathic Pulmonary Fibrosis What Is... How the Lungs Work Other Names Causes Signs & Symptoms Diagnosis Treatments Living With Clinical Trials Links Related Topics How the Lungs Work Lung Transplant Pulmonary Hypertension Pulmonary Rehabilitation Respiratory Failure ...

  13. BILATERAL OVARIAN MASS COMPLICATING PREGNANCY

    Directory of Open Access Journals (Sweden)

    Arjumand

    2015-06-01

    Full Text Available A 22 years old patient with 9 months of Amenorrhoea with a abdominal swelling was admitted to our institution with an Ultrasonography report of Bilateral ovarian mass (14.3x5.9cm in left ovary and (9.5x7cm in right ovary with internal vascularity with septations along with single live intrauterine fetus. She delivered by elective caesarean section in V/O breech presenta tion. Left side ovarian cystectomy done Right side ovarian puncture done. Histopathology showed left side – Mucinous cystadenoma. Right Side – Follicular cyst. Mucinous cystadenomas are benign epithelial ovarian tumors which tend to be unilateral, bilateral a nd multilocular with smooth surface and contain mucinous fluid, 75% of all mucinous tumors are benign, while 10% are borderline and 15% are invasive carcinomas. Benign mucinous tumors are most common in the third to fifth decades of life and may be 20 - 30 c ms in size. The incidence of ovarian cysts during pregnancy is less than 5% and most of them are benign in nature. G i ant cysts found in less than 1% of the cases of ovarian cysts with pregnancy.

  14. [Simultaneous bilateral pneumothorax. Case report].

    Science.gov (United States)

    Paolini, A; Caminiti, F; Tosato, F; Ruggieri, M; Paolini, G; Carnevale, L; Corsini, F; Marano, S; Monsellato, I

    2001-04-01

    A case report of a 44 year-old white man admitted to the surgical unit for a bilateral simultaneous pneumothorax is presented. The pneumothorax occurred on day one after a surgical operation for discal hernia; in the past the patient already presented a right spontaneous pneumothorax at 32 years of age and a left pneumothorax at 37 years of age, both treated with a pleural drainage. A thoracic drain was bilaterally positioned with a good result only in the right side. The persistence of the left pneumothorax induced the authors to perform a postero-lateral thoracotomy bullae excision and pleurectomy with a good postoperative course. After a few months a new right pneumothorax occurred and the patient was treated with a right postero-lateral thoracotomy, bullae resection and pleurectomy. On the basis of the case reported, the authors consider the different opportunities in the treatment of spontaneous pneumothorax in relation to the present knowledges and technologies. Surgical procedure is to be preferred in case of persistence of pneumothorax despite a pleural drain and in case of pneumothorax in high risk subjects. Even if thoracoscopy seems to give better results regarding postoperative pain, it is not always possible with such a method to perform a careful pleurectomy neither to obtain it in all cases (above all in secondary pneumothorax). Every case must then be carefully studied to choose the best treatment at present available. PMID:11353349

  15. How Is Pulmonary Hypertension Treated?

    Science.gov (United States)

    ... from the NHLBI on Twitter. How Is Pulmonary Hypertension Treated? Pulmonary hypertension (PH) has no cure. However, ... Types of Pulmonary Hypertension." ) Group 1 Pulmonary Arterial Hypertension Group 1 pulmonary arterial hypertension (PAH) includes PH ...

  16. Multiresolution Bilateral Filtering for Image Denoising

    OpenAIRE

    Zhang, Ming; Gunturk, Bahadir K.

    2008-01-01

    The bilateral filter is a nonlinear filter that does spatial averaging without smoothing edges; it has shown to be an effective image denoising technique. An important issue with the application of the bilateral filter is the selection of the filter parameters, which affect the results significantly. There are two main contributions of this paper. The first contribution is an empirical study of the optimal bilateral filter parameter selection in image denoising applications. The second contri...

  17. Bilateral ovarian tumour in a young girl

    Directory of Open Access Journals (Sweden)

    Krishna Kumar Govindarajan

    2013-01-01

    Full Text Available Bilateral ovarian tumour in a girl presents the dilemma of conservative versus aggressive approach towards these tumours. When faced with suspicious tumour and complete replacement of the ovaries bilaterally, bilateral oophorectomy is a viable option, though the certain possibility of infertility and lifelong hormonal supplementation is unavoidable. We report a case of bilateral ovarian masses in a young girl, which on histopathological examination showed mature teratoma with aggregates of proliferating capillary and cavernous sized vessels in the tumour wall. Such associations are rare and must be differentiated from a vascular neoplasm.

  18. Bilateral Clavicle Fracture in two Newborn Infants

    Directory of Open Access Journals (Sweden)

    Ali Kanik

    2011-12-01

    Full Text Available Background: The fracture of clavicle is the most frequently observed bone fracture as birth trauma and it is usually unilateral. It is seen following shoulder dystocia deliveries or breech presentation of macrosomic newborns.Case Presentation: We report two macrosomic newborns with bilateral clavicle fracture and brachial plexus palsy due to birth trauma. Chest X-rays confirmed bilateral fracture of clavicles. Both patients were recovered without any sequel.Conclusion: Bilateral clavicular fracture should be considered in any neonate with bilateral absent Moro reflexes.

  19. Bilateral Clavicle Fracture in Two Newborn Infants

    Directory of Open Access Journals (Sweden)

    Esra Arun Ozer

    2011-12-01

    Full Text Available Background: The fracture of clavicle is the most frequently observed bone fracture as birth trauma and it is usually unilateral. It is seen following shoulder dystocia deliveries or breech presentation of macrosomic newborns.Case Presentation: We report two macrosomic newborns with bilateral clavicle fracture and brachial plexus palsy due to birth trauma. Chest X-rays confirmed bilateral fracture of clavicles. Both patients were recovered without any sequel.Conclusion: Bilateral clavicular fracture should be considered in any neonate with bilateral absent Moro reflexes.

  20. Drug-induced pulmonary injury: CT findings in hemopathic patients

    International Nuclear Information System (INIS)

    Objective: To investigate the spiral CT findings in hemopathic patients with drug-induced pulmonary injury. Methods: CT images obtained in 11 patients with drug-induced pulmonary injury were retrospectively analyzed. Six patients had antineoplastic agent-induced pulmonary injury and 5 patients had non-neoplastic agent-induced pulmonary injury (immunosuppressor in 2 patients, antifungal in 2 patients, antineoplastic immunomodulators in 1 patient). CT findings were reviewed by a chest radiologist. Results: All 11 patients had parenchymal abnormalities on MSCT scans, including ground-glass opacities (n=8), consolidation (n=5),interlobular septal thickening (n=3) and focal fibrosis (n=2). The abnormalities were bilateral and asymmetric in all patients. They were mainly in the peripheral lung regions in 6 patients, in the central lung regions in four, and irregularly located in one. The abnormalities involved mainly the lower lung zones in six patients, the upper lung zones in two,and all lung zones homogeneously in three. One patient had fluid in bilateral pleural cavities. Three patients were given the same agent once more after the imaging turned to normal, and they presented with same clinical symptoms and similar but more serious imaging findings. Conclusions: Drug-induced pulmonary injury usually manifests as areas of ground-glass opacity and consolidation, which most commonly involves the peripheral lungs and lower lung zones. Drug-induced pulmonary injury shows reproducible but more serious lesions when the patient is given the same agent once more. (authors)

  1. Agenesis of the Corpus Callosum Due to Defective Glial Wedge Formation in Lhx2 Mutant Mice.

    Science.gov (United States)

    Chinn, Gregory A; Hirokawa, Karla E; Chuang, Tony M; Urbina, Cecilia; Patel, Fenil; Fong, Jeanette; Funatsu, Nobuo; Monuki, Edwin S

    2015-09-01

    Establishment of the corpus callosum involves coordination between callosal projection neurons and multiple midline structures, including the glial wedge (GW) rostrally and hippocampal commissure caudally. GW defects have been associated with agenesis of the corpus callosum (ACC). Here we show that conditional Lhx2 inactivation in cortical radial glia using Emx1-Cre or Nestin-Cre drivers results in ACC. The ACC phenotype was characterized by aberrant ventrally projecting callosal axons rather than Probst bundles, and was 100% penetrant on 2 different mouse strain backgrounds. Lhx2 inactivation in postmitotic cortical neurons using Nex-Cre mice did not result in ACC, suggesting that the mutant phenotype was not autonomous to the callosal projection neurons. Instead, ACC was associated with an absent hippocampal commissure and a markedly reduced to absent GW. Expression studies demonstrated strong Lhx2 expression in the normal GW and in its radial glial progenitors, with absence of Lhx2 resulting in normal Emx1 and Sox2 expression, but premature exit from the cell cycle based on EdU-Ki67 double labeling. These studies define essential roles for Lhx2 in GW, hippocampal commissure, and corpus callosum formation, and suggest that defects in radial GW progenitors can give rise to ACC. PMID:24781987

  2. Computed tomographic assessment of the surgical risks associated with fibrocavernous pulmonary tuberculosis

    International Nuclear Information System (INIS)

    We evaluated the surgical risks associated with fibrocavernous pulmonary tuberculosis by retrospectively examining chest computed tomography (CT) scans. We reviewed the records of 40 patients who underwent pulmonary resection for fibrocavernous pulmonary tuberculosis, for whom preoperative CT scans were available. The disease was categorized as class I, defined as a cavity within one lobe without remarkable pleural thickness, in 21 patients; class II, defined as a cavity extending beyond one lobe or within one lobe with remarkable pleural thickness, in 10 patients; and class III, defined as bilateral cavities, in 9 patients. Four of the nine patients with bilateral cavities underwent bilateral pulmonary resection and five underwent unilateral pulmonary resection. The study parameters were intraoperative blood loss, operative time, hospital stay, major operative morbidity, and hospital death. Intraoperative blood loss and operative time were significantly greater and hospital stay was significantly longer in patients with advanced disease (P=0.046, P=0.000, and P=0.143, respectively). Major surgical morbidity mainly occurred in association with advanced disease (P=0.028) at the following incidences: class I, 5%; class II, 30%; class III, 44.4%. Two hospital deaths occurred, both following bilateral pulmonary resection for class III disease, accounting for an overall 5% mortality rate. The surgical risks associated with fibrocavernous pulmonary tuberculosis were well correlated with anatomic involvement, according to the extent of cavitation and the severity of pleural thickness, as depicted by CT. Staged pulmonary resection or the combination of one-sided resection with other modalities is recommended for the treatment of bilateral cavities. (author)

  3. BILATERAL VESICO-URETERAL REFLUX IN PATIENT WITH CROSSED RENAL ECTOPIA AND FUSION TYPE A

    Directory of Open Access Journals (Sweden)

    A.L Bulotta

    2012-05-01

    Full Text Available Introduction. Crossed renal ectopia with fusion is a very rare congenital anomaly and the reported incidence varies between 1:1000 and 1:7000. The kidney is located on the opposite site of the mid- line from where the ureter enters the bladder. Eighty-five percent of crossed renal ectopia kid- neys are fused from below to the normally located kidney. This anomaly is more frequent for left kidney and it’s associated with vesico-ureteral re- flux (VUR in 25-70% of cases. We report the management of a six-years-old patient with Pluri- malformative Syndrome, trisomy p16 and mono- somy q2, crossed renal ectopia with fusion type A and bilateral vesico-ureteral reflux (grade IV in the right kidney and grade III in the left.Materials and Methods. A 6-year-old boy was ammitted to our hospital for UTI in plurimalfor- mative syndrome characterized at birth by cleft palate, macrocephaly, congenital clubfeet, twisted right arm, congenital dysplasia of the hip, balanic hypospadias, bilateral inguinal hernia, right renal agenesis and epilepsy tonic-clonic. MRI revealed a fusion of the ectopic kidney with the left ortho- topic kidney (crossed renal ectopia with fusion type A. Voiding cystography showed a dilatated ureter of the crossed ectopic kidney passing across the midline and of the left ureter, and a bilateral vesico-ureteral reflux ( grade IV VUR in the right kidney and grade III VUR in the left. For this reason bilaterally endoscopic subureteral infiltra- tion was performed with Deflux ( 0.3 cc for side. Results. Patient was discharged in third day and he took antibiotic for one week. There weren’t complications like fever, obstruction or UTI. Fol- low-up after 1 month is normal and there weren’t UTI. Conclusion. Generally the outcome of patients with fused crossed renal ectopia is good. Presence of associated pathology likeVUR, could lead to a progressive deterioration of renal function. There- fore, in patient with uninhabited kidney area and

  4. Pulmonary Hypertension in Pulmonary Langerhans Cell Granulomatosis

    OpenAIRE

    Matthias Held; Philipp Schnabel; Arne Warth; Berthold Jany

    2012-01-01

    Introduction. Pulmonary Langerhans cell granulomatosis is a rare disease with a variable course. In pulmonary Langerhans cell granulomatosis pulmonary hypertension is frequent and has an independent prognostic impact. A vasculopathy which ist not related to ventilatory disturbance and fibrosis has been identified. An arteriopathy and even a venulopathy have been described. Due to this possible venulopathy vasodilators carry a significant risk for pulmonary congestion and edema. No drugs have ...

  5. A Case Report of Bilateral Mirror Clubfeet and Bilateral Hand Polydactyly

    OpenAIRE

    Nguyen, Mai P; Lawler, Ericka A.; Morcuende, Jose A.

    2014-01-01

    We report a rare case of a patient with bilateral mirror clubfeet and bilateral hand polydactyly. The patient presented to our orthopaedic clinic with bilateral mirror clubfeet, each with eight toes, and bilateral hands with six fingers and a hypoplastic thumb. The pattern does not fit any described syndrome such as Martin or Laurin-Sandrow syndrome. Treatments by an orthopaedic pediatric surgeon and an orthopaedic pediatric hand surgeon are described. The patient achieved excellent functiona...

  6. Bilateral microvascular second toe transfer for bilateral post-traumatic thumb amputation

    OpenAIRE

    Rajendra Nehete; Anita Nehete; Sandeep Singla; Harshad Adhav

    2012-01-01

    In bilateral thumb amputations, the functional impairment is serious and every attempt should be made to reconstruct the thumb. We report a case of bilateral post traumatic thumb amputation, reconstructed with bilateral second toe transfer. Only two such cases have been reported in literature so far. Though there are various modalities for the reconstruction of thumb, microvascular toe transfer has its own merits. The convalescent period is minimal with excellent function. It is bilaterally s...

  7. Bilateral metachronous breast cancer with bilateral recurrences: A case report and literature review

    Energy Technology Data Exchange (ETDEWEB)

    Park, So Hyun; Sohn, Yu Mee [Dept. of Radiology, Kyung Hee University Hospital, College of Medicine, Kyung Hee University, Seoul (Korea, Republic of); Kim, Eun Kyung [Dept. of Radiology, Research Institute of Radiological Science, Yonsei University College of Medicine, Seoul (Korea, Republic of)

    2014-05-15

    The incidence of bilateral breast cancer has been reported to range from 0.4% to 14%, and it increases gradually as a result of improved early detection capabilities and longer survival times. We report a rare case where the bilateral breast cancers occurred as a metachronous bilateral breast cancer with bilateral recurrences, detected by mammography, and the rapid growth of tumor that manifested as microcalcification and skin thickening within 3 months.

  8. [Pulmonary embolism].

    Science.gov (United States)

    Söffker, Gerold; Kluge, Stefan

    2015-01-01

    Acute pulmonary embolism is an important differential diagnosis of acute chest pain. The clinical signs are often non-specific. However, diagnosis and therapy must be done quickly in order to reduce morbidity and mortality. The new (2014) European guidelines for acute pulmonary embolism (PE) focus on risk-adapted diagnostic algorithms and prognosis adapted therapy concepts. According to the hemodynamic presentation the division in a high-risk group (unstable patient with persistent hypotension or shock) or in non-high-risk groups (hemodynamically stable) was proposed. In the high-risk group the immediate diagnosis is usually done by multidetector spiral computed tomography (MDCT) and primarily the medical therapy of right ventricular dysfunction and thrombolysis is recommended.In the non-high-risk group, this is subdivided into an intermediate-risk group and low-risk group, the diagnosis algorithm based on the PE-pretest probability--determined by validated scores. Moreover, the diagnosis is usually secured by MDCT--the new gold standard in the PE-diagnosis, scores, or it can be primarily ruled out due to the high negative predictive value of D-dimer determination. To improve the prognostic risk stratification in non-high-risk group patients the additional detection of right ventricular dysfunction (MDCT, echocardiography), cardiac biomarkers (troponin, NT proBNP) and validated scores (e.g. Pulmonary Embolism Severity Index) is recommended. Therefore, the intermediate-risk group can be further subdivided. For treatment of non-high-risk group patients, the initial anticoagulation (except those with severe renal insufficiency) using low molecular weight heparin/fondaparinux and conversion to vitamin-K antagonists or alternatively with direct oral anticoagulants (DOAK) is recommended. Hemodynamically stable patients with right ventricular dysfunction and myocardial ischemia (Intermediate-high-risk group patients) but with clinically progressive hemodynamic

  9. Medical image of the week: bilateral atrial appendange thrombi

    Directory of Open Access Journals (Sweden)

    Ateeli H

    2015-01-01

    Full Text Available No abstract available. Article truncated at 150 words. A 63-year-old man with a past history significant for hypertension, low back pain and polysubstance abuse (tobacco and marijuana presented with shortness of breath and hemoptysis for the last 8 days prior to admission. His initial exam showed elevated jugular venous pressure and bilateral basal crackles with reduced air entry on the right lower lung zone. The patient was found to be in atrial fibrillation with a rapid ventricular response. His initial chest X-ray showed a moderate right-sided pleural effusion. Immediate bedside echo was concerning for bilateral ventricular dysfunction with concerns of right-sided heart pressure and volume overload. A chest CT angiogram was obtained and showed acute lower lobe pulmonary embolism, with possible distal infarct, moderate right sided pleural effusion, and filling defects in both atrial appendages concerning for thrombi (Figure 1, Panels A & B. The patient was started on therapeutic anticoagulation and underwent therapeutic thoracentesis, gentle diuresis, and ...

  10. Compression of the Right Pulmonary Artery by a Massive Defects on Pulmonary Scintigraphy

    Energy Technology Data Exchange (ETDEWEB)

    Makis, William [Brandon Regional Health Centre, Brandon (Canada); Derbekyan, Vilma [McGill Univ. Health Centre, Montreal (Canada)

    2012-03-15

    A 67 year old woman, who presented with a 2 month history of dyspnea, had a vectilation and perfusion lung scan that showed absent perfusion of the entire right lung scan that showed absent perfusion of the entire right lung with normal ventilation, as well as a rounded matched defect in the left lower lung adjacent to mialine, suspicious for an aortic aneurysm or dissection. CT pulmonary angiography revealed a massive descending aortic aneurysm compressing the right pulmonary artery as well as the left lung parenchyma, accounting for the bilateral perfusion scan defects. We present the Xe 133 ventilation, Tc 99m MAA perfusion and CT pulmonary angiography imaging findings of this rare case.

  11. Bilateral Auricular Milia en Plaque

    Directory of Open Access Journals (Sweden)

    Savaş

    2011-12-01

    Full Text Available Milia en plaque (MEP is an extremely rare form of milia that is characterized by a number of milia-like cysts on an erythematous base and histologically exhibits features of keratin cysts. The etiology is not exactly known. MEP is generally seen among middle-aged women and especially on the head and neck region. Many of the diagnosed cases are localized in the retroauricular region, eyelids and the nose region. To date, no case of MEP in the auricula has been reported in the literature. In this article, we present the case of a 71-year-old female patient clinically and histologically diagnosed with bilateral auricular milia en plaque (in the external ear canal in the light of current literature. (Turk­derm 2011; 45: 216-8

  12. Sequential bilateral retinal artery occlusion

    Directory of Open Access Journals (Sweden)

    Padrón-Pérez N

    2014-04-01

    Full Text Available Noel Padrón-Pérez,1 Janny Rosario Aronés,2 Silvia Muñoz,1 Luis Arias-Barquet,1 Jorge Arruga1,31Department of Ophthalmology, Hospital Universitari de Bellvitge, 2Hospital de l'Esperança – Parc de Salut Mar, 3Institut Català de Retina, Barcelona, SpainAbstract: An 86 year old woman experienced a sequential bilateral loss of vision over a period of less than 24 hours. Clinical findings and complementary studies suggested a bilateral atherogenic embolic event. Initially, she presented a superior branch retinal artery occlusion in her right eye followed by a central retinal artery occlusion with cilioretinal artery sparing in her left eye. Some conservative maneuvers performed did not improve visual acuity in the left eye. Supra-aortic Doppler ultrasonography revealed mild right internal carotid artery stenosis and moderate left internal carotid artery stenosis with a small, smooth, and homogeneous plaque. The transthoracic echocardiography showed a severe calcification of the mitral valve with a mild-moderate rim of stenosis. Central retinal artery occlusion and branch retinal artery occlusion are characterized by painless monocular loss of vision. Clinical approach and management attempt to treat the acute event, find the source of the vascular occlusion, and prevent further vascular events from occurring. Giant cell arteritis is a potentially treatable cause of central retinal artery occlusion and should be excluded in every single patient over 50 years old.Keywords: loss of vision, branch retinal artery occlusion, central retinal artery occlusion, Hollenhorst plaque

  13. Pulmonary Lymphangioleiomyomatosis: A Rare Case

    Directory of Open Access Journals (Sweden)

    Yetkin AĞAÇKIRAN

    2014-09-01

    Full Text Available Lymphangioleiomyomatosis is an uncommon lung disease primarily affecting women of childbearing age. It is characterized by the progressive proliferation and infiltration of smooth muscle-like cells, which lead to cystic destruction of the lung parenchyma; obstruction of airways, blood vessels, and lymphatics; and loss of pulmonary function. We present the case of a 46-year-old female patient with chest pain, cough, sputum, and dyspnea on exertion for three weeks. Minimal pneumothorax was noted, and the patient was referred to our center for further investigation and treatment. High-resolution computed tomography revealed numerous bilateral thin-walled air cysts and interstitial thickening affecting the central and peripheral part of the upper zone of the lung. We performed an open-lung biopsy to confirm lymphangioleiomyomatosis. Our aim is to discuss the pathogenesis and other lesions noted in the differential diagnosis of this rare disease.

  14. Bilateral failure of adduction following orbital decompression.

    OpenAIRE

    Kinsella, F; Kyle, P.; Stansfield, A

    1990-01-01

    We report a case of bilateral complete failure of adduction following bilateral translid antralethmoidal orbital decompression. We believe the probable mechanism is neuropraxia (temporary dysfunction) of the third cranial nerves' supply to the medial recti, owing to these nerves' occupying an anatomically abnormal position. Partial recovery of adduction occurred over the ensuing six months.

  15. Bilateral breast involvement in acute myeloid leukemia

    Directory of Open Access Journals (Sweden)

    Hakeem A, Mandakini BT, Asif K, Firdaus, Shagufta RC

    2013-04-01

    Full Text Available Breast involvement by leukemic infiltration is usually bilateral, but may be unilateral. Clinically patients can present with either single or multiple masses, or with diffuse breast engorgement, with or without nodularity. The affected patients are predominantly young adults. We present a case of an adolescent girl with acute myeloid leukemia having bilateral breast infiltration by leukemic cells.

  16. BILATERAL OVARIAN TORSION: A CASE REPORT

    OpenAIRE

    Manohar; Gopalakrishna; Kavyashree; Kala; Karthik

    2014-01-01

    : Bilateral ovarian torsion is a rare event with only a few cases reported in literature in women using ovarian stimulation drugs and in pre-menarchal age group with synchronous and asynchronous ovarian tumors. We hereby present a case of bilateral ovarian torsion in a 16 year old girl who presented as an acute pain abdomen and was diagnosed intra operatively.

  17. Acral osteolysis in bilateral compartment syndrome

    Directory of Open Access Journals (Sweden)

    Iram Saeed

    2008-08-01

    Full Text Available Carpal tunnel syndrome is a common neurological condition with rare yet potentially serious cutaneous and skeletal complications. We present a case of mutilating/ulcerating bilateral carpal tunnel syndrome in a 63 year old female. Radiographs showed symmetrical acral osteolysis in the index and middle fingers distal phalanges bilaterally. Carpal tunnel decompressions provided symptomatic relief.

  18. Bilateral spontaneous adrenal haemorrhage complicating acute pancreatitis

    International Nuclear Information System (INIS)

    Bilateral adrenal haemorrhage is an event that mandates prompt diagnosis and treatment to prevent primary adrenocortical insufficiency and potential death. Presentation can be non-specific and incidentally diagnosed with imaging alone, primarily CT. We present a case of acute pancreatitis with spontaneous bilateral adrenal haemorrhage and briefly discuss imaging and treatment implications

  19. Simultaneous and spontaneous bilateral quadriceps tendons rupture.

    Science.gov (United States)

    Celik, Evrim Coşkun; Ozbaydar, Mehmet; Ofluoglu, Demet; Demircay, Emre

    2012-07-01

    Simultaneous and spontaneous bilateral quadriceps tendon rupture is an uncommon injury that is usually seen in association with multiple medical conditions and some medications. We report a case of simultaneous and spontaneous bilateral quadriceps tendon rupture that may be related to the long-term use of a statin. PMID:22561379

  20. Bilateral microperc in a severe kyphoscoliosis

    Science.gov (United States)

    Dağgülli, Mansur; Penbegül, Necmettin; Dede, Onur; Utanğaç, Mehmet Mazhar

    2016-01-01

    Percutaneous nephrolithotomy is the standard modality for large renal calculi in normal and abnormal renal anatomic situations. This case report describes a 57-year-old male patient who presented with bilateral kidney stones and severe kyphoscoliosis. He had successfully been treated with a bilateral microperc technique. PMID:27011881

  1. Secondary polycythaemia associated with bilateral renal lymphocoeles.

    OpenAIRE

    Burton, I E; Sambrook, P.; McWilliam, L J

    1994-01-01

    A patient with a 15 year history of secondary polycythaemia due to renal erythropoietin hypersecretion is presented. Subsequent spontaneous development of bilateral renal lymphocoeles, which contained high erythropoietin levels, was shown by computerized tomography. The lymphocoeles were successfully treated by bilateral peritoneal marsupialization. No cause for the persistent polycythaemia or lymphocoeles was found at laparotomy or on renal biopsy.

  2. The Guided Bilateral Filter: When the Joint/Cross Bilateral Filter Becomes Robust

    OpenAIRE

    Caraffa, Laurent; Tarel, Jean Philippe; Charbonnier, Pierre

    2015-01-01

    The bilateral filter and its variants such as the Joint/Cross bilateral filter are well known edge-preserving image smoothing tools used in many applications. The reason of this success is its simple definition and the possibility of many adaptations. The bilateral filter is known to be related to robust estimation. This link is lost by the ad hoc introduction of the guide image in the Joint/Cross bilateral filter. We here propose a new way to derive the Joint/Cross bilateral filter as a part...

  3. [Pulmonary circulation in embolic pulmonary edema].

    Science.gov (United States)

    Sanotskaia, N V; Polikarpov, V V; Matsievskiĭ, D D

    1989-02-01

    The ultrasonic method was used in acute experiments on cats with open chest under artificial lung ventilation to obtain blood flow in low-lobar pulmonary artery and vein, the blood pressure in pulmonary artery, as well as the left atrial pressure in fat (olive oil) and mechanical (Lycopodium spores) pulmonary embolism. It is shown that pulmonary embolism produces the decrease in the blood flow in pulmonary artery and vein, the increase of the pressure in pulmonary artery and left atria, the increase of lung vessels resistance. The decrease is observed of systemic arterial pressure, bradycardia, and extrasystole. After 5-10 min the restoration of arterial pressure and heart rhythm occur and partial restoration of blood flow in pulmonary artery and vein. In many experiments the blood flow in vein outdoes that in the artery--it allows to suppose the increase of the blood flow in bronchial artery. After 60-90 min there occur sudden decrease of systemic arterial pressure, the decrease of the blood flow in pulmonary artery and vein. The pressure in pulmonary artery and resistance of pulmonary vessels remain high. Pulmonary edema developed in all animals. The death occurs in 60-100 min after the beginning of embolism. PMID:2923969

  4. Novel PAX9 and COL1A2 missense mutations causing tooth agenesis and OI/DGI without skeletal abnormalities.

    Directory of Open Access Journals (Sweden)

    Shih-Kai Wang

    Full Text Available Inherited dentin defects are classified into three types of dentinogenesis imperfecta (DGI and two types of dentin dysplasia (DD. The genetic etiology of DD-I is unknown. Defects in dentin sialophosphoprotein (DSPP cause DD type II and DGI types II and III. DGI type I is the oral manifestation of osteogenesis imperfecta (OI, a systemic disease typically caused by defects in COL1A1 or COL1A2. Mutations in MSX1, PAX9, AXIN2, EDA and WNT10A can cause non-syndromic familial tooth agenesis. In this study a simplex pattern of clinical dentinogenesis imperfecta juxtaposed with a dominant pattern of hypodontia (mild tooth agenesis was evaluated, and available family members were recruited. Mutational analyses of the candidate genes for DGI and hypodontia were performed and the results validated. A spontaneous novel mutation in COL1A2 (c.1171G>A; p.Gly391Ser causing only dentin defects and a novel mutation in PAX9 (c.43T>A; p.Phe15Ile causing hypodontia were identified and correlated with the phenotypic presentations in the family. Bone radiographs of the proband's dominant leg and foot were within normal limits. We conclude that when no DSPP mutation is identified in clinically determined isolated DGI cases, COL1A1 and COL1A2 should be considered as candidate genes. PAX9 mutation p.Phe15Ile within the N-terminal β-hairpin structure of the PAX9 paired domain causes tooth agenesis.

  5. Pulmonary disease in patients with human immunodeficiency virus infection

    DEFF Research Database (Denmark)

    Lundgren, J D; Orholm, Marianne; Lundgren, B;

    1989-01-01

    Pulmonary disease is the most important cause of morbidity and mortality in patients infected with human immunodeficiency virus (HIV). All parts of the hospital system are expected to be involved in the diagnosis and treatment of HIV infected patients in the coming years. Many different processes...... cause pulmonary disease alone or in combination. Bilateral interstitial infiltrates are the most frequent chest x-ray abnormality and are most frequently caused by infection with Pneumocystis carinii. Cytomegalovirus, Mycobacterium tuberculosis, nonspecific interstitial pneumonitis and pulmonary Kaposi......'s sarcoma are the most important parts of the differential diagnosis. An aggressive approach to the diagnosis of pulmonary disease in this patient population is indicated in order to provide optimal care and assess new therapies....

  6. Pulmonary manifestation of leptospirosis: Clinical and experimental correlation

    International Nuclear Information System (INIS)

    Pulmonary radiographic findings were noted in 24 (56%) of 43 patients with leptospirosis, proved by positive serology. Three radiographic patterns were evident: (1) small nodular densities (n = 11), (2) patchy or confluent consolidation (n = 4), and (3) diffuse ill-defined groundglass density (n = 9). Abnormalities were bilateral, nonlobar, and peripheral. Pulmonary abnormalities all resolved within 10 days, except in three patients who died. Artificial infection was induced through intraperitoneal injection of Leptospira interohemorrhagiae into 20 guinea pigs. The lungs from the guinea pigs initially showed petechial hemorrhage which progressed to large confluent ares of hemorrhage. The typical pulmonary radiographic findings of leptopirosis are compatible with the multifocal pulmonary hemorrhage seen in the guinea pigs

  7. Apelin and pulmonary hypertension

    DEFF Research Database (Denmark)

    Andersen, Charlotte Uggerhøj; Hilberg, Ole; Mellemkjær, Søren;

    2011-01-01

    Pulmonary arterial hypertension (PAH) is a devastating disease characterized by pulmonary vasoconstriction, pulmonary arterial remodeling, abnormal angiogenesis and impaired right ventricular function. Despite progress in pharmacological therapy, there is still no cure for PAH. The peptide apelin...... vasoconstriction, and has positive inotropic and cardioprotective effects. Apelin attenuates vasoconstriction in isolated rat pulmonary arteries, and chronic treatment with apelin attenuates the development of pulmonary hypertension in animal models. The existing literature thus renders APLNR an interesting...

  8. An Unusual Case of Recurrent Pyocolpos Following Midtrimester Miscarriage Revealed as Obstructed Hemivagina with Ipsilateral Renal Agenesis (OHVIRA Syndrome

    Directory of Open Access Journals (Sweden)

    Haldar M

    2014-08-01

    Full Text Available Obstructed hemivagina with ipsilateral renal agenesis (OHVIRA Syndrome is a rare congenital anomaly. It mostly presents with severe dysmenorrhea and a palpable mass due to unilateral hematocolpos. Sometime it presents in unusual way with prolonged vaginal bleeding and profuse vaginal discharge. Here we report case of an 18 years old married female with OHVIRA syndrome presenting late with symptoms of recurrent pyocolpos following dilatation and evacuation for midtrimester miscarriage that was diagnosed on the basis of MRI and managed by vaginal septotomy.

  9. Perineal lipoma with accessory labioscrotal fold and penis-like phallus in a female infant with unilateral renal agenesis.

    Science.gov (United States)

    Mifsud, William; Sambandan, Nikola; Humphries, Paul; Sebire, Neil J; Mushtaq, Imran

    2014-07-01

    We present a case of a female 46,XX infant with a perineal lipoma within an accessory labioscrotal fold containing a penis-like phallus, associated with contralateral renal agenesis and complete absence of Müllerian/paramesonephric structures. To our knowledge, this is the first report of perineal lipoma and accessory labioscrotal fold associated with urogenital abnormalities in a female. The case also has an exceptional penis-like phallus in the absence of Y chromosome material or evidence of virilization. PMID:24857274

  10. Pulmonary veno-occlusive disease

    Science.gov (United States)

    Pulmonary vaso-occlusive disease ... common among children and young adults. As the disease gets worse, it causes narrowed pulmonary veins, pulmonary artery hypertension , and congestion and swelling ...

  11. Learning and memory in individuals with agenesis of the corpus callosum.

    Science.gov (United States)

    Paul, Lynn K; Erickson, Roger L; Hartman, Jo Ann; Brown, Warren S

    2016-06-01

    Damage to long white matter pathways in the cerebral cortex is known to affect memory capacity. However, the specific contribution of interhemispheric connectivity in memory functioning is only beginning to become understood. The present study examined verbal and visual memory processing in individuals with agenesis of the corpus callosum (AgCC) using the Wechsler Memory Scale-Third Edition (WMS-III; Wechsler, 1997b). Thirty participants with AgCC (FSIQ >78) were compared against 30 healthy age and IQ matched controls on auditory/verbal (Logical Memory, Verbal Paired Associates) and visual (Visual Reproduction, Faces) memory subtests. Performance was worse in AgCC than controls on immediate and delayed verbal recall for rote word pairs and on delayed recall of faces, as well as on percent recall for these tasks. Immediate recall for thematic information from stories was also worse in AgCC, but groups did not differ on memory for details from narratives or on recall for thematic information following a time delay. Groups also did not differ on memory for abstract figures or immediate recall of faces. On all subtests, individuals with AgCC had greater frequency of clinically significant impairments than predicted by the normal distribution. Results suggest less efficient overall verbal and visual learning and memory with relative weaknesses processing verbal pairs and delayed recall for faces. These findings suggest that the corpus callosum facilitates more efficient learning and recall for both verbal and visual information, that individuals with AgCC may benefit from receiving verbal information within semantic context, and that known deficits in facial processing in individuals with AgCC may contribute to their impairments in recall for faces. PMID:27091586

  12. [Differential diagnosis of pulmonary tuberculosis and community-acquired pneumonia].

    Science.gov (United States)

    Deĭkina, O N; Mishin, V Iu; Demikhova, O V

    2007-01-01

    The purpose of this investigation was to enhance the efficiency of differential diagnosis of pneumonia and pulmonary tuberculosis. A hundred and fifty-nine adult patients were examined. These included 78 patients with pulmonary tuberculosis and 81 with community-acquired p neumonia. The clinical features of infiltrative pulmonary tuberculosis (n = 48) and mild community-acquired pneumonia (n = 51) were compared. The course of caseous pneumonia (n = 30) was compared with that of moderate and severe community-acquired pneumonia (n = 30). Significant differences in the manifestations of the intoxication and bronchopulmonary syndrome were not found in patients with community-acquired pneumonia and infiltrative pulmonary tuberculosis. Physical studies showed that in patients with community-acquired pneumonia, moist rale (54.9%) and crepitation (11.8%) were prevalent, but in those with infiltrative tuberculosis rale was absent in 60.4% of cases and the pattern of respiration was unchanged in 79.2%. Chest X-ray studies indicated that in patients with community-acquired pneumonia, lower lobar inflammatory changes were predominant in 62.8% of cases whereas in those with infiltrative pulmonary tuberculosis the process was mainly bilateral (43.8%) with the presence of destructive changes (83.3%) and bronchogenic dissemination (66.7%). In patients with caseous pneumonia, the intoxication syndrome was more significant than in those with severe community-acquired pneumonia. Chest X-ray studies demonstrated that in patients with caseous pneumonia, specific changes were bilateral with the involvement of 2 lobes or more, with destruction and bronchogenic dissemination while in those with community-acquired pneumonia, the pulmonary processes were predominantly bilateral (76.6%) at the lower lobar site (36.7%). PMID:17338353

  13. Idiopathic pulmonary hemosiderosis complicated by Down syndrome.

    Science.gov (United States)

    Watanabe, Hirofumi; Ayusawa, Mamoru; Kato, Masataka; Chou, Ami; Komori, Akiko; Abe, Yuriko; Matsumura, Masaharu; Kamiyama, Hiroshi; Izumi, Hiroyuki; Takahashi, Shori

    2015-10-01

    We report the case of a 9-year-old girl with Down syndrome (DS) diagnosed with idiopathic pulmonary hemosiderosis (IPH). Although acute pneumonia complicated by hemolytic anemia was suspected, IPH was finally diagnosed on bronchoscopy. Treatment with prednisolone achieved good clinical response. An association between IPH and DS was not able to be identified, but immunological issues in DS may contribute to the onset of IPH. Recurrent and intractable respiratory symptoms with marked infiltrative shadows in the bilateral lungs and complicated by severe anemia in patients with DS should suggest IPH. PMID:26508184

  14. Pulmonary metastatic calcification: a case report

    Energy Technology Data Exchange (ETDEWEB)

    Bozi, Lilian Christine Franchiotti [Radiology, Hospital Universitario Antonio Pedro (HUAP), Niteroi, RJ (Brazil); Melo, Alessandro Severo Alves de; Marchiori, Edson, E-mail: edmarchiori@gmail.com [Department of Radiology, School of Medicine, Universidade Federal Fluminense (UFF), Niteroi, RJ (Brazil)

    2012-09-15

    The present report describes the case of a 48-year-old female patient suffering from chronic renal failure on dialysis for 13 years. She presented with hemoptysis, fever, productive cough and dyspnoea. Chest radiography showed predominance of ill-defined opacities in the middle and lower lung fields, bilaterally. Chest computed tomography showed ground glass opacities associated with poorly defined centrilobular nodules with ground-glass attenuation. The patient was submitted to bronchoalveolar lavage that was negative for mycobacteria and fungi. On the basis of such findings, open lung biopsy was performed, which revealed metastatic pulmonary calcification. (author)

  15. Bilateral panophthalmitis in dengue fever

    Directory of Open Access Journals (Sweden)

    Sangeetha Sriram

    2015-01-01

    Full Text Available We report the case of a 25-year-old male patient who presented with bilateral panophthalmitis as the initial ocular manifestation of dengue fever. The diagnosis was a little confusing as he initially presented with features suggestive of retrobulbar hemorrhage secondary to his very low platelet count, which is a common feature of dengue fever. Ophthalmic complications are usually seen in young adults who often present at the nadir of thrombocytopenia. Ocular findings may include anterior uveitis, vitritis, retinal hemorrhages, retinal vascular sheathing, yellow subretinal dots, retinal pigment epithelium (RPE mottling, foveolitis that is clinically seen as a round subretinal yellowish lesion at the fovea, retinochoroiditis, choroidal effusion, optic disc swelling, optic neuritis, neuroretinitis, and oculomotor nerve palsy. [1] There is only one reported case of unilateral endogenous panophthalmitis due to dengue fever. Hence, clinicians and ophthalmologists have to be aware of this vision-threatening complication of dengue for early recognition and prompt treatment to save the vision of these young patients and prevent morbidity.

  16. Bilateral symmetry across Aphrodite Terra

    International Nuclear Information System (INIS)

    There are three main highland areas on Venus: Beta Regio, Ishtar Terra and Aphrodite Terra. The latter is least known and the least mapped, yet existing analyses of Aphrodite Terra based on available Pioneer-Venus orbiter data suggest that it may be the site of extensive rifting. Some of the highest resolution (30 km) PV data (SAR) included most of the western half of Aphrodite Terra. Recent analysis of the SAR data together with Arecibo range-doppler topographic profiling (10 X 100 km horizontal and 10 m vertical resolution) across parts of Aphrodite, further characterized the nature of possible tectonic processes in the equatorial highlands. The existence of distinct topographic and radar morphologic linear discontinuities across the nearly east-west strike of Aphrodite Terra is indicated. Another prominent set of linear features is distinctly parallel to and orthogonal to the ground tracks of the PV spacecraft and are not included because of the possibility that they are artifacts. Study of the northwest trending cross-strike discontinuities (CSD's) and the nature of topographic and morphologic features along their strike suggest the presence of bilateral topographic and morphologic symmetry about the long axis of Aphrodite Terra

  17. [An autopsy case of pulmonary tumor thrombotic microangiopathy complicated with interstitial pneumonia and lipoid pneumonia].

    Science.gov (United States)

    Ota, Kyoko; Matsuyama, Masashi; Kokuho, Nariaki; Masuko, Hironori; Hayashi, Hiroki; Iizuka, Takashi; Hayashibara, Kenji; Saito, Takefumi; Kawabata, Yoshinori

    2009-06-01

    A 67-year-old woman was admitted to our hospital because of progressive dyspnea, cough, bloody sputum, and backache. Chest radiography and CT scans showed bilateral diffuse interstitial shadows, bilateral pleural effusion and dilatation of the pulmonary artery. Echocardiography indicated pulmonary hypertension, and the serum tumor marker levels were elevated. We performed right cardiac catheterization, and withdrew some blood from a pulmonary artery catheter in the wedge position. We confirmed moderate pulmonary hypertension, and adenocarcinoma-like malignant cells were seen in the aspirated blood. The patient died of progressive respiratory failure despite supportive care. In addition to PTTM and lymphangiosis carcinomatosa, autopsy of the right lung revealed interstitial pneumonia and lipoid pneumonia, both of which were not reported before to be associated with PTTM. PMID:19601530

  18. Radiologic findings of diffuse Pulmonary hemorrhage

    Energy Technology Data Exchange (ETDEWEB)

    Seo, Mi Ra; Song, Koun Sik; Lee, Jin Seong; Lim, Tae Hwan [Ulsan Univ. College of Medicine, Seoul (Korea, Republic of)

    1998-12-01

    To describe the chest radiographic and CT findings of diffuse pulmonary hemorrhage. Two radiologists retrospectively analysed the chest radiographic and CT findings of six patients with diffuse pulmonary hemorrhage. Using open lung biopsy(n=3D2) and transbronchial lung biopsy or bronchoalveolar lavage(n=3D4), diagnosis was based on the presence of hemosiderin-laden macrophage or intra-alveolar hemorrhage. Underlying diseases were Wegener's granulomatosis(n=3D2), antiphospholipid antibody syndrome(n=3D2), Henoch-Schonlein purpura(n=3D1), and idopathic pulmonary hemosiderosis(n=3D1). In all patients, sequential chest radiographs, obtained during a one to six-month period, were available. HRCT scans were obtained in five patinets, and conventional CT scans in one. Follow-up HRCT scans were obtained in two. We also analyzed the patterns of involvement, distribution and sequential changes in the pulmonary abnormalities seen on chest radiographs and CT scans. Chest radiographs showed multifocal patchy consolidation(n=3D6), ground-glass opacity(n=3D3), and multiple granular or nodular opacity(n=3D3). These lesions were intermingled in five patients, while in one there was consolidation only. Sequential chest radiographs demonstrated the improvement of initial pulmonary abnormalities and appearance of new lesions elsewhere within 5-6 days, though within 7-25 (average, 13) days, these had almost normalized. HRCT scans showed patchy consolidation(n=3D5), multiple patchy ground-glass opacity(n=3D5), or ill-defined air space nodules(n=3D4). These lesions were intermingled in five patients, and in one, ground-glass opacity only was noted. In two patients there were interlobular septal thickening and intalobular reticular opacity. The distribution of these abnormalities was almost always bilateral, diffuse with no zonal predominancy, and spared the apex of the lung and subpleural region were less affected. Although chest radiographic and CT findings of diffuse pulmonary

  19. Radiologic findings of diffuse Pulmonary hemorrhage

    International Nuclear Information System (INIS)

    To describe the chest radiographic and CT findings of diffuse pulmonary hemorrhage. Two radiologists retrospectively analysed the chest radiographic and CT findings of six patients with diffuse pulmonary hemorrhage. Using open lung biopsy(n=3D2) and transbronchial lung biopsy or bronchoalveolar lavage(n=3D4), diagnosis was based on the presence of hemosiderin-laden macrophage or intra-alveolar hemorrhage. Underlying diseases were Wegener's granulomatosis(n=3D2), antiphospholipid antibody syndrome(n=3D2), Henoch-Schonlein purpura(n=3D1), and idopathic pulmonary hemosiderosis(n=3D1). In all patients, sequential chest radiographs, obtained during a one to six-month period, were available. HRCT scans were obtained in five patinets, and conventional CT scans in one. Follow-up HRCT scans were obtained in two. We also analyzed the patterns of involvement, distribution and sequential changes in the pulmonary abnormalities seen on chest radiographs and CT scans. Chest radiographs showed multifocal patchy consolidation(n=3D6), ground-glass opacity(n=3D3), and multiple granular or nodular opacity(n=3D3). These lesions were intermingled in five patients, while in one there was consolidation only. Sequential chest radiographs demonstrated the improvement of initial pulmonary abnormalities and appearance of new lesions elsewhere within 5-6 days, though within 7-25 (average, 13) days, these had almost normalized. HRCT scans showed patchy consolidation(n=3D5), multiple patchy ground-glass opacity(n=3D5), or ill-defined air space nodules(n=3D4). These lesions were intermingled in five patients, and in one, ground-glass opacity only was noted. In two patients there were interlobular septal thickening and intalobular reticular opacity. The distribution of these abnormalities was almost always bilateral, diffuse with no zonal predominancy, and spared the apex of the lung and subpleural region were less affected. Although chest radiographic and CT findings of diffuse pulmonary

  20. Bilateral Petit’s Triangle Hernia

    Directory of Open Access Journals (Sweden)

    Sanjay Kumar Bhasin, Arshad Bashir Khan, Sanjay Sharma

    2006-07-01

    Full Text Available Lumbar traingle hernia that occurs through lumbar triangles is very rare type of hernia. Only about 300 cases havebeen reported till date. Bilateral Petit’s triangle hernia find further rarity and the case under reference is probably thefirst ever reported case of Primary bilateral Petit’s triangle hernia. The present case is of a 46 years old married,multigravida female who presented with 1 year duration of LBA and subsequently notice of swelling both sides oflow back. FNAC revealed lipoma and on exploration it turned out to be rarest extra peritoneal bilateral Petit’s trianglehernia, fat as contents.

  1. Bilateral facial palsy associated with leptospirosis

    Directory of Open Access Journals (Sweden)

    Andressa Alves da Silva

    2009-08-01

    Full Text Available Leptospirosis is a zoonosis of worldwide occurrence caused by the spirochete Leptospira interrogans. It is an acute feverish disease with a broad clinical spectrum and follows a characteristic biphasic course. Bilateral facial palsy is a rare clinical condition and the differential diagnosis of its causes is extensive. The objective of this exploratory study, presented as a case report, is to describe the occurrence of bilateral facial palsy as an unusual manifestation of leptospirosis. This suggestion should not be overlooked when analyzing the causes for bilateral facial palsy, and should be considered with other possible differential diagnoses, some of which are potentially fatal.

  2. Pulmonary complications after bone marrow transplantation in chest radiography

    International Nuclear Information System (INIS)

    In a retrospective study chest radiographs of 87 bone marrow transplant recipients were analysed. 36 patients had pulmonary complications with lung opacifications. Interstitial changes were more frequent than air-space pneumonias. The latter were caused by bacteria and fungi only. The most common cause of pulmonary complications was cytomegalovirus pneumonia. It was characterised uniformly by a bilateral diffuse interstitial pattern. Idiopathic interstitial pneumonias were indistinguishable from CMV infection. Pneumonias caused by Epstein-Barr virus and protozoa, diffuse radiation pneumonitis and leukaemic infiltrates were rare and also associated with interstitial changes. (orig.)

  3. Pulmonary perfusion scintigraphy in children with chronic pulmonary damage: Preliminary results from application of the SPECT technique

    International Nuclear Information System (INIS)

    The aim was to describe lung perfusion SPECT (LPS) findings in children with chronic pulmonary damage. Material and Method: We revised 106 LPS of children (age:3.9 ± 3.3 yr) with chronic pulmonary damage, performed with a two headed gamma camera. The most common clinical diagnosis were adenovirus bronchopneumonia sequelae (ADV)(29%), unknown origin chronic pulmonary damage (UOPD) (21%), bronchopulmonary dysplasia (BPD)(15%), and chronic obstructed bronchitis (COB)(14%). Severity of pulmonary alterations were evaluated using a Score, which considered extent and type of lung involvement and differential pulmonary perfusion. Pattern of involvement and Score were correlated with diagnosis and gender. Results: Ninety one percent (96/106) of LPS were abnormal (62% boys). Fifty two (54%) showed bilateral alterations, being this finding (p:0.019) more frequent in boys. ADV group showed mainly a mixed bilateral pattern (35%), UOPD a focal unilateral pattern (32%), COB a diffuse unilateral pattern (33%), and BPD a normal pattern (25%). Miscellaneous and UOPD showed the highest score values, and BPD and COB the lowest ones. Conclusion: In this population, LPS is abnormal in high proportion, with some differential characteristics according gender and original diagnosis (au)

  4. Diffuse Pulmonary Hemorrhage After Fibrinolytic Therapy for Acute Myocardial Infarction in a Cocaine Abuser Patient

    OpenAIRE

    Mohammad Parsa Mahjoob; Isa Khaheshi; Koosha Paydary

    2014-01-01

    We report a 45-year-old man with antroseptal myocardial infarction who developed bilateral basal alveolar infiltrates after initiating the fibrinolytic therapy. Although thrombolytic therapy with streptokinase is generally used in the course of acute myocardial infarction and has diminished morbidity and mortality, pulmonary hemorrhage is an uncommon, but a potentially life-threatening complication that should be regarded as one of the differential diagnoses of pulmonary infiltrates or droppi...

  5. Invasive Pulmonary Aspergillosis in the Intensive Care Unit: A Case Report

    OpenAIRE

    Mehmet Akif Yazar; Aytuna Kuzucuoğlu,; Mehmet Barış Açıkgöz

    2016-01-01

    SUMMARY Invasive pulmonary aspergillosis (IPA) is an infection rarely seen in intensive care units (ICU). We aimed to discuss the case of IPA found in a patient followed up in our ICU due to chronic obstructive pulmonary disease (COPD). A 58-year old patient diagnosed with COPD has been admitted to our ICU due to respiratory failure. There were not any other diseases except from COPD. In his radiological imaging there were diffuse bilateral infiltrates in the ...

  6. BODY SIZE REDUCTION AND TOOTH AGENESIS IN LATE PLEISTOCENE MELES MELES (CARNIVORA, MAMMALIA FROM INGARANO (SOUTHERN ITALY

    Directory of Open Access Journals (Sweden)

    DAWID ADAM IURINO

    2014-03-01

    Full Text Available In mammals combined factors such as body size reduction and loss of peripheral teeth are often associated with endemism phenomena. This condition is particularly evident in insular contexts where is a complete geographic isolation. During the Pleistocene there have been several glacial stages, which changed the physiognomy of the Italian peninsula strongly influencing the distribution and morphology of mammalian faunas. Several genetic studies have shown that some Southern Italian areas have particular endemic species of small and medium size mammals. During Pleistocene these areas have been characterized by particular climatic/environmental conditions, and are generally called "glacial refugia". They represent geographically isolated areas over time, where the origin of faunas with peculiar features is favoured. In this study, the occurrence of Meles meles from the Late Pleistocene site of Ingarano (Apulia, Southern Italy is documented for the first time. This taxon is represented only by a partial skull (splancnocranum that, despite the relative completeness, includes peculiar and well-preserved dental features that could be related to a partial endemic condition. The fossil shows a reduced body size and the agenesis of peripheral teeth, both conditions that are typical of the extant badgers from Crete, Rhodes and Japan. To test this hypothesis, tomographic analysis have been provided to establish the dental agenesis, and, in order to understand the magnitude of the body size reduction, biometric analyses have been carried on. The obtained data have been compared to measures of the extant Eurasian badgers.SHORT NOTE

  7. Giant infantile pulmonary hemangioma

    International Nuclear Information System (INIS)

    We present a very unusual case of giant infantile pulmonary hemangioma presenting as a large solitary pulmonary mass. This was successfully managed with surgical resection. Histological examination revealed that the mass was positive for GLUT-1 receptor, a marker for infantile hemangioma. To our knowledge only a few cases of pulmonary hemangioma have been described previously in the literature. Pulmonary hemangiomas are very rare lesions, most of them presenting as a pulmonary mass. This case emphasizes the fact that this rare lesion should be considered in the differential of an enhancing pulmonary mass in an infant. (orig.)

  8. Pulmonary function in advanced pulmonary hypertension.

    OpenAIRE

    Burke, C. M.; Glanville, A R; Morris, A J; Rubin, D.; Harvey, J. A.; Theodore, J; Robin, E. D.

    1987-01-01

    Pulmonary mechanical function and gas exchange were studied in 33 patients with advanced pulmonary vascular disease, resulting from primary pulmonary hypertension in 18 cases and from Eisenmenger physiology in 15 cases. Evidence of airway obstruction was found in most patients. In addition, mean total lung capacity (TLC) was only 81.5% of predicted and 27% of our subjects had values of TLC less than one standard deviation below the mean predicted value. The mean value for transfer factor (TLC...

  9. Pulmonary malaria: high-resolution computed tomography findings - a case report; Malaria pulmonar: aspectos na tomografia computadorizada de alta resolucao - relato de caso

    Energy Technology Data Exchange (ETDEWEB)

    Rodrigues, Rosana; Souza, Daniel Andrade Tinoco de [Universidade Federal, Rio de Janeiro, RJ (Brazil). Hospital Universitario Clementino Fraga Filho. Servico de Radiodiagnostico; Hospital Copa D' Or, Rio de Janeiro, RJ (Brazil); Marchiori, Edson [Universidade Federal Fluminense, Niteroi, RJ (Brazil). Dept. de Radiologia]. E-mail: edmarchiori@bol.com.br

    2004-04-01

    We report the case of a 38-year-old man with pulmonary malaria. High-resolution computed tomography showed thickening of the peribronchovascular interstitium and interlobular septa, areas of consolidation and ground glass attenuation and bilateral pleural effusion suggesting pulmonary edema. The patient recovered well after receiving specific treatment and was discharged after 11 days of hospitalization. (author)

  10. FLOWING BILATERAL FILTER: DEFINITION AND IMPLEMENTATIONS

    Directory of Open Access Journals (Sweden)

    Maxime Moreaud

    2015-06-01

    Full Text Available The bilateral filter plays a key role in image processing applications due to its intuitive parameterization and its high quality filter result, smoothing homogeneous regions while preserving the edges of the objects. Considering the image as a topological relief, seeing pixel intensities as peaks and valleys, we introduce a way to control the tonal weighting coefficients, the flowing bilateral filter, reducing "halo" artifacts typically produced by the regular bilateral filter around a large peak surrounded by two valleys of lower values. In this paper we propose to investigate exact and approximated versions of CPU and parallel GPU (Graphical Processing Unit based implementations of the regular and flowing bilateral filter using the NVidia CUDA API. Fast implementations of these filters are important for the processing of large 3D volumes up to several GB acquired by x-ray or electron tomography.

  11. THE EUROPEAN UNION’S BILATERAL APPROACH

    Directory of Open Access Journals (Sweden)

    Ludmila BORTA

    2014-12-01

    Full Text Available The EU is a world economic power and a major trading partner for most countries. All the time, this region has been interested and has acted towards a free and fair trade. The decrease and even the elimination of tariff and non-tariff barriers in the world trade are among the main objectives of the EU strategy for international trade. At the moment, the elusive outcome of the WTO Doha Round has led to the proliferation of bilateral trade agreements worldwide. Although the EU remains committed to further development of the multilateral trading system, however, the EU still has appealed also to the development of bilateral trade relations. The aim of this paper is to illustrate the current bilateral dimension of the common commercial policy of the EU. In conclusion, to describe this bilateral approach of the EU we are using one word, namely “diversity”.

  12. An unusual cause of bilateral diaphragmatic paralysis.

    OpenAIRE

    Pandit, A; Kalra, S.; Woodcock, A

    1992-01-01

    In a patient who had a sudden onset of bilateral diaphragmatic paralysis after forceful neck manipulation complete, though gradual, recovery in lung function and transdiaphragmatic pressures was seen over three years. This is a previously unrecognised risk of neck osteopathy.

  13. Exercise-Induced Pulmonary Edema in a Triathlon

    Directory of Open Access Journals (Sweden)

    Hirotomo Yamanashi

    2015-01-01

    Full Text Available Introduction. Family physicians have more opportunities to attend athletic competitions as medical staff at first-aid centers because of the increasing popularity of endurance sports. Case. A 38-year-old man who participated in a triathlon race experienced difficulty in breathing after swimming and was moved to a first-aid center. His initial oxygen saturation was 82% and a thoracic computed tomography scan showed bilateral ground glass opacity in the peripheral lungs. His diagnosis was noncardiogenic pulmonary edema associated with exercise or swimming: exercise-induced pulmonary edema (EIPE or swimming-induced pulmonary edema (SIPE. Treatment with furosemide and corticosteroid relieved his symptoms of pulmonary edema. Discussion. Noncardiogenic pulmonary edema associated with endurance sports is not common, but knowledge about EIPE/SIPE or neurogenic pulmonary edema associated with hyponatremia, which is called Ayus-Arieff syndrome, is crucial. Knowledge and caution for possible risk factors, such as exposure to cold water or overhydration, are essential for both medical staff and endurance athletes. Conclusion. To determine the presence of pulmonary edema associated with strenuous exercise, oxygen saturation should be used as a screening tool at a first-aid center. To avoid risks for EIPE/SIPE, knowledge about these diseases is essential for medical staff and for athletes who perform extreme exercise.

  14. Novel missense mutation in the L1 gene in a child with corpus callosum agenesis, retardation, adducted thumbs, spastic paraparesis, and hydrocephalus

    NARCIS (Netherlands)

    Sztriha, L; Frossard, P; Hofstra, RMW; Verlind, E; Nork, M

    2000-01-01

    Corpus callosum agenesis, retardation, adducted thumbs, spastic paraparesis, and hydrocephalus (CRASH syndrome) is an X-linked recessive disorder caused by mutations in the neuronal cell adhesion molecule L1 (L1CAM) gene. L1 plays a key role in axon outgrowth and pathfinding during the development o

  15. Lumbar discal cyst causing bilateral radiculopathy

    OpenAIRE

    Hyung-Jun, Kwak; Dae-Yong, Kim; Tae-Ho, Kim; Ho-Sang, Park; Jae-Sung, Kim; Jae-Won, Jang; Jung-Kil, Lee; Pawl, Ron; Nancy E Epstein; Bydon, Ali

    2011-01-01

    Background: Discal cyst is a rare lesion that can result in clinical symptoms typical of disc herniation manifesting as a unilateral single nerve root lesion. To the best of the authors’ knowledge, this is the first reported case of discal cyst resulting in bilateral radiculopathy. Case Description: A 48-year-old female presented with bilateral sciatica and neurogenic claudication for 3 months. Magnetic resonance imaging revealed an extradural cystic lesion compressing the ventral aspect of t...

  16. Diagnosis and management of bilateral nasolabial cysts

    OpenAIRE

    Rajkumar Parwani; Simran Parwani; Sangeeta Wanjari

    2013-01-01

    Nasolabial cysts are painless, submucosal, non-odontogenic jaw cysts presenting as soft tissue swellings in the maxillary anterior mucolabial fold lateral to midline, leading to elevation of nasal ala. Present case documents bilateral nasolabial cysts in a 69-year-old Asian female patient. In the present case, extraoral swelling of maxillary lip and elevation of nasal ala was observed on right side of the face. Intraorally, soft and fluctuant bilateral cysts were observed. Straw-colored fluid...

  17. Accounting Relations in Bilateral Value Added Trade

    OpenAIRE

    Robert Stehrer

    2013-01-01

    Abstract The increasing international fragmentation of production has triggered the development of a number of widely used indicators accounting for value added flows in the world economy. This paper generalises these measures by simultaneously considering the import side and focusing on bilateral gross and value added trade flows. It discusses how these indicators relate to each other, the role of double counting in bilateral value added trade, and aggregation issues in global value added flows...

  18. Bilateral Hypertrophic Olivary Degeneration in Wilson Disease

    OpenAIRE

    Otto, Josephin; Guenther, Peter; Hoffmann, Karl-Titus

    2013-01-01

    Hypertrophic olivary degeneration resulting from lesions of the dento-rubro-olivary pathway, also called Guillain-Mollaret-triangle, has been described previously in a number of cases. Reports about bilateral hypertrophic olivary degeneration of the inferior olivary nuclei are very limited, and the magnetic resonance imaging findings of hypertrophic olivary degeneration in Wilson disease have not yet been described to the best of our knowledge. Herein, we present the first report of bilateral...

  19. Bilateral synchronous plasmacytoma of the testis.

    Science.gov (United States)

    Narayanan, Geetha; Joseph, Rona; Soman, Lali V

    2016-04-01

    Extramedullary plasmacytoma (EMP) is usually seen in the head and neck regions and in the upper respiratory, gastrointestinal, and central nervous systems. Testis is a rare site for EMP, and bilateral synchronous testicular plasmacytoma occurring as an isolated event at initial presentation has been reported only once previously. We present herein the second such report in a 70-year-old man who underwent bilateral orchidectomy. PMID:27034568

  20. Superpixel Convolutional Networks using Bilateral Inceptions

    OpenAIRE

    Gadde, Raghudeep; Jampani, Varun; Kiefel, Martin; Gehler, Peter V.

    2015-01-01

    In this paper we propose a CNN architecture for image segmentation. We introduce a new "bilateral inception" layer that is used on top of a convolutional architecture. The bilateral inception performs a filtering between superpixels in an image. This addresses two problems that arise with CNN segmentation architectures. First, this layer propagates information between (super) pixels while respecting image edges, thus using the structured information of the problem for improved results. Second...

  1. Bilateral lipoma arborescens associated with early osteoarthritis

    Energy Technology Data Exchange (ETDEWEB)

    Al-Ismail, Khalid; Torreggiani, William C.; Keogh, Ciaran; Munk, Peter L. [Department of Radiology, Vancouver General Hospital, 855 West 12th Avenue, Vancouver, BC, Canada V5Z 1M9 (Canada); Al-Sheikh, Fahad [Department of Radiology, Riyadh Military Hospital (Saudi Arabia)

    2002-11-01

    Lipoma arborescens is a rare disease that has generally been considered to be unilateral. We report a case of bilateral disease in a patient with associated marked premature osteoarthritis of the knees. We postulate a possible association between these two conditions and explain a possible underlying mechanism. We suggest that lipoma arborescens be considered as a rare underlying cause of young patients with bilateral premature osteoarthritis, and recommend MRI as the definitive imaging modality. (orig.)

  2. Complementarity between Bilateral Trade and Financial Integration

    OpenAIRE

    Shin, Kwanho; Yang, Doo Yong

    2006-01-01

    This paper explores the complementarities between bilateral trade in goods and financial assets. By utilizing a gravity model specification with an extended dataset in terms of time span and asset classification as well as alternative instrumental variables, we confirm the existence of positive evidence for complementarities. We find that common factors such as bilateral distance and other economic size variables that determine both cross-border trade and financial flows contribute to complem...

  3. MODERN VIEWS ON BILATERAL BREAST CANCER

    Directory of Open Access Journals (Sweden)

    Ye. A. Fesik

    2014-01-01

    Full Text Available Presented modern literature data on the features of the pathogenesis, course, clinical and morphological expression and tumor characteristics, parameters and nodal metastasis of hematogenous bilateral breast cancer. Highlight the results of domestic and foreign studies in recent years to determine the prognostic factors and recurrence of synchronous and metachronous bilateral breast cancer. It was revealed that the frequency of bilateral breast tumor lesions varies widely, ranging from 0.1 to 20%, with metachronous tumors recorded significantly higher (69.6% than the synchronous (22.7%. The probability of occurrence of metachronous breast cancer is higher in women with a family history, as well as if they have a gene mutation BRCA-1. Found that the most common histological type of breast tumor with bilateral lesions is invasive ductal. However, the incidence of invasive lobular cancer and non-invasive lobular cancer is slightly higher among synchronous bilateral cancer compared with unilateral disease. Studies have shown that in a double-sided synchronous breast cancer tumor, as a rule, has a lower degree of differentiation, and the higher the expression level of estrogen receptors and progesterone receptors. Relevance of the issue because the identification of patterns in the study of lymphatic and hematogenous features bilateral metastasis of mammary tumors provides a basis for speculation about the differences in the progression of neoplastic disease in these groups and is a cause for further detailed research in this area to identify and evaluate the prognosis and also the choice of tactics of such patients.

  4. Bilateral Keratectasia 34 Years after Corneal Transplant

    Directory of Open Access Journals (Sweden)

    Xavier Valldeperas

    2010-07-01

    Full Text Available We report the clinical findings of a patient with severe bilateral keratectasia 34 years after a penetrating keratoplasty (PK in both eyes. An otherwise healthy 67-year-old man complained of deterioration of the eyesight in both eyes over the last 6 months. The patient was diagnosed with bilateral keratoconus at the age of 32 years, and he underwent a bilateral PK. At presentation, visual acuity was 20/200 in the right eye and light perception in the left eye. A Pentacam pachymetric map revealed a central pachymetry of 720 µm in the right eye and of 710 µm in the left eye, as well as an average paracentral pachymetry of 436 and 270 µm in the 9-mm zone in the right and the left eye, respectively. Corneal topography revealed bilateral irregular and asymmetric bowing with generalized steepening and high corneal power. We describe a case of bilateral keratectasia 34 years after PK in a patient who was originally diagnosed with bilateral keratoconus.

  5. A Case Report of Bilateral Mirror Clubfeet and Bilateral Hand Polydactyly

    Science.gov (United States)

    Nguyen, Mai P; Lawler, Ericka A; Morcuende, Jose A

    2014-01-01

    We report a rare case of a patient with bilateral mirror clubfeet and bilateral hand polydactyly. The patient presented to our orthopaedic clinic with bilateral mirror clubfeet, each with eight toes, and bilateral hands with six fingers and a hypoplastic thumb. The pattern does not fit any described syndrome such as Martin or Laurin-Sandrow syndrome. Treatments by an orthopaedic pediatric surgeon and an orthopaedic pediatric hand surgeon are described. The patient achieved excellent functional and cosmetic outcomes at four year follow-up. PMID:25328478

  6. Bilateral microvascular second toe transfer for bilateral post-traumatic thumb amputation

    Directory of Open Access Journals (Sweden)

    Rajendra Nehete

    2012-01-01

    Full Text Available In bilateral thumb amputations, the functional impairment is serious and every attempt should be made to reconstruct the thumb. We report a case of bilateral post traumatic thumb amputation, reconstructed with bilateral second toe transfer. Only two such cases have been reported in literature so far. Though there are various modalities for the reconstruction of thumb, microvascular toe transfer has its own merits. The convalescent period is minimal with excellent function. It is bilaterally symmetric and aesthetically superior to the osteoplastic reconstruction. The technical details are discussed, and the long term functional and aesthetic results are presented.

  7. Diffuse pulmonary hemosiderosis after exposure to pesticides - A case report

    OpenAIRE

    Kayser, Klaus; Plodziszewska, Monika; Waitr, Elzbieta; Slodkowska, Janina; Altiner, Mithat; Gabius, Hans-Joachim

    1998-01-01

    This report describes the clinical, radiological, microscopical and ligandohistochemical findings in a 17-year-old woman who suffered from an acute onset of pulmonary hemosiderosis after inhalation of pesticides used for the cultivation of strawberries. She complained of headache, dyspnea, rhinitis, weakness and recurrent severe hemoptysis. Chest radiographs revealed bilateral patchy infiltrates, predominantly in the lower parts of both lungs. The consecutive severe anemia was treated by mult...

  8. Beware of the devastating pulmonary aspergillosis syndromes In certain environments

    LENUS (Irish Health Repository)

    Kooblall, M

    2016-02-01

    Nowadays with more stem cell transplants and immunosuppressive therapies there has been a rise in pulmonary aspergillosis syndromes. The following illustrates such a case. A 49 year old man had a past history of ankylosing spondylitis with a bilateral hip replacement. He was also on surveillance for a superficial bladder tumour since 2007. His chest x-ray in 2008 was normal. In 2010 his CXR showed patchy opacification in the right apex. CT thorax confirm fibrotic changes.

  9. Pulmonary Idiopathic Alveolar Ossification in a Raccoon (Procyon lotor)

    OpenAIRE

    Hamir, Amir N; Rupprecht, Charles E.

    2010-01-01

    Here we describe gross and histopathologic findings in a laboratory-confined adult male raccoon (Procyon lotor) with microscopic ossified areas in pulmonary alveoli. At the time of necropsy, gross lesions were present in the kidneys and in one thyroid gland. Noteworthy microscopic findings included multifocal foci of osseous tissue within the alveoli of the lungs, bilateral thyroid adenomas, pancreatic islet cell amyloidosis, cortical kidney infarcts, cystic adenomatous hyperplasia of urinary...

  10. Spontaneous idiopathic bilateral adrenal haemorrhage: a rare cause of abdominal pain.

    Science.gov (United States)

    Nazir, Salik; Sivarajah, Surendra; Fiscus, Valena; York, Eugene

    2016-01-01

    We describe a case of a 62-year-old woman with a history of chronic obstructive pulmonary disease and gastro-oesophageal reflux disease who presented to the emergency department with left lower quadrant abdominal pain, flank pain with nausea and no history of preceding trauma. The patient had finished a course of azithromycin and oral methylprednisolone 1 day prior to presentation. Abdominal and pelvic CT scan identified changes suggestive of bilateral adrenal haemorrhage. The patient did not show signs of acute adrenal insufficiency but was started on steroid replacement therapy because of concerns about possible disease progression. All recognised causes of adrenal haemorrhage were excluded suggesting this was a case of spontaneous idiopathic bilateral adrenal haemorrhage, a rarely reported phenomenon in the literature. The patient was discharged after clinical improvement following 6 days in hospital, taking oral steroid replacement. PMID:27166002

  11. Pulmonary arteriovenous fistula

    Science.gov (United States)

    Pulmonary arteriovenous fistula is an abnormal connection between an artery and vein in the lungs. As a result, blood passes ... Pulmonary arteriovenous fistulas are usually the result of abnormal development of the blood vessels of the lung. Most occur in ...

  12. Pulmonary alveolar proteinosis

    Science.gov (United States)

    Alveolar proteinosis; Pulmonary alveolar phospholipoproteinosis ... In some cases, the cause of pulmonary alveolar proteinosis is unknown. In others, it occurs with lung infection or an immune problem. It also can occur with cancers of the blood system, ...

  13. Hantavirus Pulmonary Syndrome (HPS)

    Science.gov (United States)

    ... this page: About CDC.gov . Hantavirus Share Compartir Hantavirus Pulmonary Syndrome (HPS) Severe HPS. Image courtesy D. ... the workers showed evidence of infection or illness. Hantavirus Pulmonary Syndrome (HPS) Topics Transmission Where HPS is ...

  14. Candidate gene analysis of tooth agenesis identifies novel mutations in six genes and suggests significant role for WNT and EDA signaling and allele combinations.

    Science.gov (United States)

    Arte, Sirpa; Parmanen, Satu; Pirinen, Sinikka; Alaluusua, Satu; Nieminen, Pekka

    2013-01-01

    Failure to develop complete dentition, tooth agenesis, is a common developmental anomaly manifested most often as isolated but also as associated with many developmental syndromes. It typically affects third molars or one or few other permanent teeth but severe agenesis is also relatively prevalent. Here we report mutational analyses of seven candidate genes in a cohort of 127 probands with non-syndromic tooth agenesis. 82 lacked more than five permanent teeth excluding third molars, called as oligodontia. We identified 28 mutations, 17 of which were novel. Together with our previous reports, we have identified two mutations in MSX1, AXIN2 and EDARADD, five in PAX9, four in EDA and EDAR, and nine in WNT10A. They were observed in 58 probands (44%), with a mean number of missing teeth of 11.7 (range 4 to 34). Almost all of these probands had severe agenesis. Only few of the probands but several relatives with heterozygous genotypes of WNT10A or EDAR conformed to the common type of non-syndromic tooth agenesis, incisor-premolar hypodontia. Mutations in MSX1 and PAX9 affected predominantly posterior teeth, whereas both deciduous and permanent incisors were especially sensitive to mutations in EDA and EDAR. Many mutations in EDAR, EDARADD and WNT10A were present in several families. Biallelic or heterozygous genotypes of WNT10A were observed in 32 and hemizygous or heterozygous genotypes of EDA, EDAR or EDARADD in 22 probands. An EDARADD variant were in seven probands present together with variants in EDAR or WNT10A, suggesting combined phenotypic effects of alleles in distinct genes. PMID:23991204

  15. Candidate gene analysis of tooth agenesis identifies novel mutations in six genes and suggests significant role for WNT and EDA signaling and allele combinations.

    Directory of Open Access Journals (Sweden)

    Sirpa Arte

    Full Text Available Failure to develop complete dentition, tooth agenesis, is a common developmental anomaly manifested most often as isolated but also as associated with many developmental syndromes. It typically affects third molars or one or few other permanent teeth but severe agenesis is also relatively prevalent. Here we report mutational analyses of seven candidate genes in a cohort of 127 probands with non-syndromic tooth agenesis. 82 lacked more than five permanent teeth excluding third molars, called as oligodontia. We identified 28 mutations, 17 of which were novel. Together with our previous reports, we have identified two mutations in MSX1, AXIN2 and EDARADD, five in PAX9, four in EDA and EDAR, and nine in WNT10A. They were observed in 58 probands (44%, with a mean number of missing teeth of 11.7 (range 4 to 34. Almost all of these probands had severe agenesis. Only few of the probands but several relatives with heterozygous genotypes of WNT10A or EDAR conformed to the common type of non-syndromic tooth agenesis, incisor-premolar hypodontia. Mutations in MSX1 and PAX9 affected predominantly posterior teeth, whereas both deciduous and permanent incisors were especially sensitive to mutations in EDA and EDAR. Many mutations in EDAR, EDARADD and WNT10A were present in several families. Biallelic or heterozygous genotypes of WNT10A were observed in 32 and hemizygous or heterozygous genotypes of EDA, EDAR or EDARADD in 22 probands. An EDARADD variant were in seven probands present together with variants in EDAR or WNT10A, suggesting combined phenotypic effects of alleles in distinct genes.

  16. Extensive pulmonary alveolar microlithiasis

    OpenAIRE

    Rittayamai, Nuttapol; Muangman, Nisa; Ruangchira-Urai, Ruchira

    2013-01-01

    Pulmonary alveolar microlithiasis is a rare pulmonary disorder that is caused by abnormal sodium-dependent phosphate co-transporter from the mutation of SLC34A2 gene, leading to accumulation of microliths in the alveoli. We report the extensive pulmonary alveolar microlithiasis in an elderly woman who presented with progressive dyspnea for 2 months. Chest radiograph revealed diffuse pulmonary calcification. Tissue histopathology from open lung biopsy demonstrated widespread intra-alveolar lam...

  17. The pulmonary extracellular lining.

    OpenAIRE

    George, G; Hook, G E

    1984-01-01

    The extracellular lining of the lungs is reviewed. The pulmonary extracellular lining is a complex mixture of phospholipids, proteins and carbohydrates which is absolutely essential for the maintenance of normal pulmonary functions such as gas exchange. Without the lining the lungs would collapse. Alterations in the pulmonary extracellular lining may underlie some disease conditions induced by toxic agents, especially those which interfere with the formation of pulmonary surfactant. The extra...

  18. Multi-vesicular pulmonary hydatid cyst, the potent underestimated factor in the formation of daughter cysts of pulmonary hydatid disease

    OpenAIRE

    Mohsen Sokouti; Babak Sokouti; Behrooz Shokouhi; Mohammad Hossein Rahimi-Rad

    2015-01-01

    Pulmonary multi-vesicular hydatid disease (HD) with Echinococcus granulosus is rare. A 28-year-old woman presented to our center with cough and respiratory distress. Chest x-ray and computerized tomography scan revealed bilateral giant cysts with water-lily sign (ruptured hydatid cysts). The left cyst was in vicinity of heart. With thoracotomy cysts of both lungs were removed. Thousands of translucent, homogenized small daughter cysts were discovered from the left side cyst. Pathologic examin...

  19. Symptoms, Diagnosis and Treatment for Pulmonary Fibrosis

    Science.gov (United States)

    ... www.lung.org > Lung Health and Diseases > Lung Disease Lookup > Pulmonary Fibrosis Pulmonary Fibrosis Symptoms, Causes & Risk Factors Pulmonary ... while processing XML file."); } }); } } --> Blank Section Header Lung Disease Lookup Pulmonary Fibrosis Learn About Pulmonary Fibrosis Pulmonary Fibrosis Symptoms, ...

  20. Idiopathic pulmonary hemosiderosis

    OpenAIRE

    Sunilkumar, B M; Sathishkumar, K M; Somashekhar, A R; P.P. Maiya

    2010-01-01

    Recurrent or chronic pulmonary hemorrhage is rare in children. Idiopathic pulmonary hemosiderosis (IPH) manifests as hemoptysis, diffuse parenchymal infiltrates on chest radiographs and microcytic hypochromic anemia. The hemoptysis present may be mistaken for more common diseases, delaying the diagnosis and further management. Idiopathic pulmonary hemosiderosis is a disorder of unknown etiology. Treatment of IPH includes immunosuppressive drugs along with supportive measures.

  1. Efficacy of total lymphoid irradiation for chronic allograft rejection following bilateral lung transplantation

    International Nuclear Information System (INIS)

    Purpose: To assess the safety and efficacy of total lymphoid irradiation (TLI) in patients experiencing chronic rejection following bilateral lung transplantation (BLT). Patients and Materials: Eleven patients received TLI for chronic allograft rejection (bronchiolitis obliterans syndrome) refractory to conventional treatment modalities. Radiation therapy (RT) was prescribed as 8 Gy delivered in 10 0.8-Gy fractions, 2 fractions/week, via mantle, paraaortic, and inverted-Y fields. Serial pre- and post-RT pulmonary function values, complete blood counts, and immunosuppressive augmentation requirements [use of methylprednisolone, murine anti-human mature T-cell monoclonal antibody (OKT3), polyclonal antithymocyte globulin (ATG), and tacrolimus] were monitored. Results: In the 3 months preceding TLI, the average decrease in forced expiratory volume in 1 s (FEV1) was 34% (range 0-75%) and the median number of immunosuppression augmentations was 3 (range 0-5). Only 4 of 11 patients completed all 10 TLI treatment fractions. Reasons for discontinuation included progressive pulmonary decline (four patients), worsening pulmonary infection (two patients), and persistent thrombocytopenia (one patient). Seven of the 11 patients failed within 8 weeks of treatment cessation. One patient had unabated rejection and received bilateral living related-donor transplants; he is alive and well. Six patients died. Two of these deaths were due to pulmonary infection from organisms isolated prior to the start of RT; the other four deaths were from progressive pulmonary decline. The four remaining patients had durable positive responses to TLI (mean follow-up of 47 weeks; range 24-72). Comparing the 3 months preceding RT to the 3 months following treatment, these four patients had improvements in average FEV1 (40% decline vs. 1% improvement) and fewer median number of immunosuppressive augmentations (3.5 vs. 0). None of these patients has developed lymphoproliferative disease or has died

  2. Bilateral ovarian cystic teratomata mimicking bilateral pure ovarian hemangiomata: case report.

    Science.gov (United States)

    Feuerstein, I M; Aronson, B L; McCarthy, E F

    1984-01-01

    This report describes a case of bilateral, benign cystic ovarian teratomata which were composed predominantly of cavernous hemangiomatous elements. The right-sided lesion, in particular, mimicked a pure ovarian hemangioma. The clinical presentation, bilaterality of the lesions, the sizes of the hemangiomata, and the subsequent hemangioma of the leg are all of interest in this very rare lesion. PMID:6511164

  3. Simultaneus bilateral spontaneus pneumothoraces: A Retrospective Analysis of 11 Cases

    Directory of Open Access Journals (Sweden)

    Ufuk Cobanoglu

    2011-09-01

    Full Text Available Spontaneous pneumothorax (SP is relatively common in clinical practice and occurs more frequently in young, tall thin men, and in smokers. However, simultaneous bilateral spontaneous pneumothorax (SBSP is a rare clinical condition that often presents with significant respiratory distress. It is often dangerous; therefore, the chest drain should be inserted immediately. In this study, simultaneus bilateral spontaneus pneumothoraces cases were divided into two groups and retrospectively evaluated according to age, sex, diagnostic methods, treatments, and results. Material and Methods :Between January 2006 and May 2009, 11 patients with SBSP were enrolled into our study. Age, gender, underlying lung disease, smoking history, symptoms, diagnosis, treatment type, surgical indication, morbidity, recurrence, mortality, duration of chest tube and postoperative hospital stay of the patients were reviewed. Arterial blood gas values (before and after intervention in patiens with primary and secondary spontaneous pneumothorax were evaluated. Results; 7 patients (63.63 % were male and 4 patients (36.37 % were female and their mean age was 34,5±6.81 years. There were 4 (36.37% primary SP and 4 (36.37% secondary SP patients. Chronic obstructive pulmonary disease (COPD was the most common cause in secondary SP patients. In two (18.18% patients recurrency were observed. Eight (72.72% patients had smoking history. The most common symptom in both groups was dyspnea. All patients had immediate bilateral chest tubes on admission. We treated these patients with chest drain insertion, VATS (Video-Assisted Thoracoscopic Surgery, axillary thoracotomy, and chemical pleurodesis. Postoperative morbidity was detected in 3 (27.27% patients (prolonged air leak in 1 case, empyema in 1 case and pneumonia in 1 case. No mortality was observed in alll cases. Recurrence developed in 3 (27.27% patients in this series. Conclusions: An urgent and effective treatment requires in the

  4. Bilateral electric energy contracts: return and risk

    Energy Technology Data Exchange (ETDEWEB)

    Gunn, Laura K.; Silva, Elisa B.; Correia, Paulo B. [State University of Campinas (UNICAMP), SP (Brazil). College of Mechanical Engineering

    2009-07-01

    In Brazil electricity is traded through three segments: the spot market that balances offer and demand, with prices calculated by a cost-based computational model; the regulated market , where prices are settled in public auctions, and the free market for bilateral contracts. As spot and regulated market prices are public information, a seller is able to calculate his opportunity price to trade a bilateral contract in the free market by using the non-arbitrage principle. Thus, the seller searches the price of a bilateral contract in the free market that balances his/her revenues with the value expected in case it were negotiated in the regulated and the spot market. Besides the expected revenue, the seller may also consider the CVaR to measure the risk of her/his bilateral contract in the free market. So this paper develops a binomial lattice approach to price bilateral contracts in the free market, considering the seller's opportunity of negotiations in both regulated and spot markets, and measuring the contract risk directly. (author)

  5. Pulmonary Alveolar Proteinosis in Association with Congenital Dyserythropoietic Anemia: A Case Report

    OpenAIRE

    Carden, Marcus A.; Ashish Barman; Gita Massey

    2012-01-01

    A two-year-old girl with congenital dyserythropoietic anemia (CDA) acutely developed fever, tachypnea, and increased oxygen requirement. Chest X-ray revealed bilateral interstitial infiltrates and mild cardiomegaly. Blood cultures grew no infectious agents, while pulmonary specimens grew cytomegalovirus (CMV). Treatment with intravenous ganciclovir was initiated but without response. Final cytologic preparations of bronchoalveolar lavage (BAL) fluid revealed eosinophilic amorphous material co...

  6. Pulmonary vascular diseases.

    Science.gov (United States)

    Mélot, C; Naeije, R

    2011-04-01

    Diseases of the pulmonary vasculature are a cause of increased pulmonary vascular resistance (PVR) in pulmonary embolism, chronic thromboembolic pulmonary hypertension (CTEPH), and pulmonary arterial hypertension or decreased PVR in pulmonary arteriovenous malformations on hereditary hemorrhagic telangiectasia, portal hypertension, or cavopulmonary anastomosis. All these conditions are associated with a decrease in both arterial PO2 and PCO2. Gas exchange in pulmonary vascular diseases with increased PVR is characterized by a shift of ventilation and perfusion to high ventilation-perfusion ratios, a mild to moderate increase in perfusion to low ventilation-perfusion ratios, and an increased physiologic dead space. Hypoxemia in these patients is essentially explained by altered ventilation-perfusion matching amplified by a decreased mixed venous PO2 caused by a low cardiac output. Hypocapnia is accounted for by hyperventilation, which is essentially related to an increased chemosensitivity. A cardiac shunt on a patent foramen ovale may be a cause of severe hypoxemia in a proportion of patients with pulmonary hypertension and an increase in right atrial pressure. Gas exchange in pulmonary arteriovenous malformations is characterized by variable degree of pulmonary shunting and/or diffusion-perfusion imbalance. Hypocapnia is caused by an increased ventilation in relation to an increased pulmonary blood flow with direct peripheral chemoreceptor stimulation by shunted mixed venous blood flow. PMID:23737196

  7. Chronic Bilateral Tibial Stress Fractures with Valgus Treated with Bilateral Intramedullary Nailing: A Case Report

    Directory of Open Access Journals (Sweden)

    Steven K Dailey

    2014-01-01

    Full Text Available Introduction: Stress fractures are overuse injuries most commonly seen in athletes, military recruits, and individuals with endocrine abnormalities. It has been demonstrated that chronic cases of anterior tibial stress fractures refractory to conservative management respond well to intramedullary nailing. To our knowledge, only one report has been published concerning patients with bilateral tibial stress fractures treated with bilateral intramedullary nailing. All patients in the series were high-level athletes. We present the case of a non-athletic patient with chronic bilateral tibial stress fractures and associated deformity successfully treated with bilateral intramedullary nails. Case Report: A 23-year-old Caucasian female full-time student presented with chronic bilateral shin pain for approximately five years. She had failed an extensive regimen of conservative management. She was diagnosed with chronic bilateral tibial stress fractures based on history, physical examination, and radiologic findings. She subsequently underwent sequential intramedullary nailing of her tibiae. Both tibiae were in valgus alignment; however, this did not preclude nail placement. The nails deformed upon insertion into the sclerotic canals to conform to the deformation. Post operatively the tibiae united and patient was relieved of her symptoms. Conclusion: Bilateral intramedullary nailing of chronic bilateral tibial stress fractures should be considered as a treatment option for all patients, not just high-level athletes, who fail a trial of conservative management. Additionally, mild to moderate tibial malalignment does not necessarily preclude tibial nailing as the smaller nails placed in sclerotic canals will likely deform on insertion and conform to the canal. Keywords: Bilateral intramedullary nailing, bilateral tibial stress fractures, valgus

  8. Multidetector computed tomography pulmonary angiography in childhood acute pulmonary embolism

    International Nuclear Information System (INIS)

    Pulmonary embolism is a life-threatening condition affecting people of all ages. Multidetector row CT pulmonary angiography has improved the imaging of pulmonary embolism in both adults and children and is now regarded as the routine modality for detection of pulmonary embolism. Advanced CT pulmonary angiography techniques developed in recent years, such as dual-energy CT, have been applied as a one-stop modality for pulmonary embolism diagnosis in children, as they can simultaneously provide anatomical and functional information. We discuss CT pulmonary angiography techniques, common and uncommon findings of pulmonary embolism in both conventional and dual-energy CT pulmonary angiography, and radiation dose considerations. (orig.)

  9. Multidetector computed tomography pulmonary angiography in childhood acute pulmonary embolism

    Energy Technology Data Exchange (ETDEWEB)

    Tang, Chun Xiang; Zhang, Long Jiang; Lu, Guang Ming [Medical School of Nanjing University, Department of Medical Imaging, Jinling Hospital, Nanjing, Jiangsu (China); Schoepf, U.J. [Medical School of Nanjing University, Department of Medical Imaging, Jinling Hospital, Nanjing, Jiangsu (China); Medical University of South Carolina, Department of Radiology and Radiological Science, Charleston, SC (United States); Medical University of South Carolina, Department of Pediatrics, Charleston, SC (United States); Chowdhury, Shahryar M. [Medical University of South Carolina, Department of Pediatrics, Charleston, SC (United States); Fox, Mary A. [Medical University of South Carolina, Department of Radiology and Radiological Science, Charleston, SC (United States)

    2015-09-15

    Pulmonary embolism is a life-threatening condition affecting people of all ages. Multidetector row CT pulmonary angiography has improved the imaging of pulmonary embolism in both adults and children and is now regarded as the routine modality for detection of pulmonary embolism. Advanced CT pulmonary angiography techniques developed in recent years, such as dual-energy CT, have been applied as a one-stop modality for pulmonary embolism diagnosis in children, as they can simultaneously provide anatomical and functional information. We discuss CT pulmonary angiography techniques, common and uncommon findings of pulmonary embolism in both conventional and dual-energy CT pulmonary angiography, and radiation dose considerations. (orig.)

  10. Unsuspected pulmonary alveolar proteinosis in a patient with acquired immunodeficiency syndrome: a case report

    Directory of Open Access Journals (Sweden)

    Niazi Masooma

    2011-02-01

    Full Text Available Abstract Introduction Diffuse lung infiltrates are a common finding in patients with acquired immunodeficiency syndrome and causes range from infectious processes to malignancies or interstitial lung diseases. Pulmonary alveolar proteinosis is a rare pulmonary disorder rarely reported in patients infected with human immunodeficiency virus. Secondary pulmonary alveolar proteinosis is associated with conditions involving functional impairment or reduced numbers of alveolar macrophages. It can be caused by hematologic malignancies, inhalation of toxic dust, fumes or gases, infectious or pharmacologic immunosuppression, or lysinuric protein intolerance. Case presentation A 42-year-old African American man infected with human immunodeficiency virus was admitted with chronic respiratory symptoms and diffuse pulmonary infiltrates. Chest computed tomography revealed bilateral spontaneous pneumothoraces, for which he required bilateral chest tubes. Initial laboratory investigations did not reveal any contributory conditions. Histological examination of a lung biopsy taken during video-assisted thoracoscopy showed pulmonary alveolar proteinosis concurrent with cytomegalovirus pneumonitis. After ganciclovir treatment, our patient showed radiologic and clinical improvement. Conclusion The differential diagnosis for patients with immunosuppression and lung infiltrates requires extensive investigations. As pulmonary alveolar proteinosis is rare, the diagnosis can be easily missed. Our case highlights the importance of invasive investigations and histology in the management of patients infected with human immunodeficiency virus and pulmonary disease who do not respond to empiric therapy.

  11. Prognosis of synchronous bilateral breast cancer

    DEFF Research Database (Denmark)

    Holm, Marianne; Tjønneland, Anne; Balslev, Eva;

    2014-01-01

    Currently, no consistent evidence-based guidelines for the management of synchronous bilateral breast cancer (SBBC) exist and it is uncertain how presenting with SBBC affects patients' prognosis. We conducted a review of studies analyzing the association between SBBC and prognosis. The studies that...... reported adjusted effect measures were included in meta-analyses of effect of bilaterality on breast cancer mortality. From 57 initially identified records 17 studies from 11 different countries including 8,050 SBBC patients were included. The quality of the studies varied but was generally low with small...... sample sizes, and lack of consistent, detailed histo-pathological information. When doing meta-analysis on the subgroup of studies that provided adjusted effect estimates on breast cancer mortality (nine studies including 3,631 SBBC cases), we found that bilaterality in itself had a negative impact on...

  12. Langerhans Cell Histiocytosis in Bilateral Mastoid Cavity

    Directory of Open Access Journals (Sweden)

    Kazım Bozdemir

    2013-01-01

    Full Text Available A 39-year-old male was admitted to our clinic with symptoms of headache, dizziness, nausea, otalgia, otorrhea, tinnitus, and hearing loss in both ears for 3 weeks. Physical examination revealed edema in the tympanic membrane and external ear canal, and pain by palpation in the mastoid area bilaterally. There was no nystagmus, and the rest of the physical examination was otherwise normal. Temporal bone high resolution computed tomography (CT showed a lesion causing erosion in the mastoid cortex, tegmen tympani, ossicles, and in the bone covering the sigmoid sinus bilaterally. There was also erosion in the superior semicircular canal and petrous bone on the left side. Cortical mastoidectomy was performed under general anesthesia. Histopathologic examination of the tissue revealed Langerhans cell histiocytosis (LCH. In this paper a case with LCH, presenting with bilateral mastoid involvement which has been rarely reported in the literature, is discussed with the existing literature.

  13. Bilateral Supernumerary Kidney: A Very Rare Presentation

    International Nuclear Information System (INIS)

    To our knowledge, bilateral supernumerary kidney is a very rare renal abnormality and there are five cases presented in the literature. It is difficult to diagnose supernumerary kidney and clinicians have not detected most cases preoperatively. Laboratory and imaging studies were acquired and carefully examined. The normal laboratory tests were found. Emergency ultrasonography was performed and they revealed no signs of parenchymal abnormality in both kidneys. Serial imaging study including enhanced computed tomography (CT) was performed. An imaging study identified bilateral supernumerary kidney with expanded collecting systems. On each side, significant rotation anomaly was found. In addition, there were two different renal arteries originating from the aorta. This report presents radiological determinations of supernumerary kidney bilaterally in a young man. We think that CT commonly appears to be enough for the diagnosis of supernumerary kidneys

  14. Danish Exports and Danish Bilateral Aid

    DEFF Research Database (Denmark)

    Hansen, Henrik; Rand, John

    was much higher in the 1980s compared to in particular the most recent decade. This may be related to factors such as untying of aid in the same period. The econometric analysis has two important limitations. First of all, the model can only give information about marginal changes in aid. As a...... trade policies. The main result of the study is that Danish bilateral aid has a positive and statistically significant impact on Danish exports to the recipient countries. Bilateral development assistance may affect exports through several channels. Three of the main channels are direct aid tying......; increasing recipient income where higher income leads to higher imports, and decreased trade costs, say due to improved information about cultural and administrative customs and practices. Thus, as for preferential trade arrangements, bilateral aid has two potential economic effects; trade creation working...

  15. Fistulectomy as a surgical option for pulmonary arteriovenous malformation

    Institute of Scientific and Technical Information of China (English)

    ZHANG Shao-yan; ZHANG Zhi-tai; OU Song-lei; HU Yan-sheng; SONG Fei-qiang; LI Xin; MA Xu-chen; MA Xin-xin; LIANG Lin; LI Dong; GUO Lin; SUN Zhen

    2009-01-01

    Background Surgical resection remains the treatment of choice for pulmonary arteriovenous malformation but removes some normal lung parenchyma. This study aimed to evaluate the effect and safety of the lung-saving procedure of fistulectomy as an alternative to lung resection.Methods From July 2003 to July 2008, 6 selected patients with pulmonary arteriovenous malformations underwent fistulectomies. Among them, 1 patient underwent emergency operation and 2 underwent bilateral operations. One patient received postoperative embolotherapy.Results No hospital deaths or postoperative morbidity occurred. PaO_2 increased significantly after operation. All patients were free of symptoms and hypoxia during a follow-up for 9 months to 5 years.Conclusions Fistulectomy is a safe and effective procedure for patients with pulmonary arteriovenous malformation and may be an alternative to lung resection.

  16. Agenesis and dysgenesis of the corpus callosum: clinical, genetic and neuroimaging findings in a series of 41 patients.

    Science.gov (United States)

    Schell-Apacik, Chayim Can; Wagner, Kristina; Bihler, Moritz; Ertl-Wagner, Birgit; Heinrich, Uwe; Klopocki, Eva; Kalscheuer, Vera M; Muenke, Maximilian; von Voss, Hubertus

    2008-10-01

    Agenesis of the corpus callosum (ACC) is among the most frequent human brain malformations with an incidence of 0.5-70 in 10,000. It is a heterogeneous condition, for which several different genetic causes are known, for example, ACC as part of monogenic syndromes or complex chromosomal rearrangements. We systematically evaluated the data of 172 patients with documented corpus callosum abnormalities in the records, and 23 patients with chromosomal rearrangements known to be associated with corpus callosum changes. All available neuroimaging data, including CT and MRI, were re-evaluated following a standardized protocol. Whenever feasible chromosome and subtelomere analyses as well as molecular genetic testing were performed in patients with disorders of the corpus callosum in order to identify a genetic diagnosis. Our results showed that 41 patients with complete absence (agenesis of the corpus callosum-ACC) or partial absence (dysgenesis of the corpus callosum-DCC) were identified. Out of these 28 had ACC, 13 had DCC. In 11 of the 28 patients with ACC, the following diagnoses could be established: Mowat-Wilson syndrome (n = 2), Walker-Warburg syndrome (n = 1), oro-facial-digital syndrome type 1 (n = 1), and chromosomal rearrangements (n = 7), including a patient with an apparently balanced reciprocal translocation, which led to the disruption and a predicted loss of function in the FOXG1B gene. The cause of the ACC in 17 patients remained unclear. In 2 of the 13 patients with DCC, unbalanced chromosomal rearrangements could be detected (n = 2), while the cause of DCC in 11 patients remained unclear. In our series of cases a variety of genetic causes of disorders of the corpus callosum were identified with cytogenetic anomalies representing the most common underlying etiology. PMID:18792984

  17. Awake operative videothoracoscopic pulmonary resections.

    Science.gov (United States)

    Pompeo, Eugenio; Mineo, Tommaso C

    2008-08-01

    moderately increasing the depth of sedation while maintaining spontaneous breathing. Finally, as long as the physiologic impact of awake metastasectomy is definitively elucidated, the authors believe this modality should be used for unilateral procedures, while deserving a staged bilateral approach for bilateral lung metastasectomy. Avoidance of general anesthesia results in a faster recovery with immediate return to many daily life activities, including drinking, eating, and walking, and a reduction in hospital stay and procedure-related costs. If confirmed with future studies, these results could advocate earlier resection of peripheral solitary pulmonary nodules, reducing the risk for delaying a diagnosis of unexpected pulmonary malignancy. Furthermore, potential new frontiers of awake thoracoscopic surgery might include assessment of feasibility and safety of anatomic resections in properly selected instances. Ethical and economical concerns push remorselessly for less frequent and less-invasive surgery. Administrators advocate minimal hospitalization and cost-saving treatments, whereas patients ultimately ask for appropriate health care. Thoracic surgeons of the third millennium must accept the challenge of this dynamic and rapidly evolving scenario without loosing the right root, which probably lays just between well-established conventional surgery techniques and newly available advanced technology tools. Awake thoracic surgery will benefit from evidence-based data that are progressively accumulating. Findings will stimulate experts to continue an active clinical investigation in this unpredictably evolving surgical field, which might ultimately lead to a better understanding of cardiorespiratory physiology and effects of the surgical pneumothorax and thoracic epidural anesthesia on perioperative, respiratory function in awake patients. As the Italian architect Renzo Piano recently stated, "Recovering in the past can be reassuring but the future is the only place

  18. Bilateral subepidermal calcified nodules of the eyelid

    OpenAIRE

    Malki, Salem Al; Al-Faky, Yasser H.; Al-Rikabi, Ammar C

    2010-01-01

    A subepidermal calcified nodule (SCN) is an uncommon benign lesion, which usually develops in early childhood and is typically solitary. A rare form of this lesion has been reported in the eyelid. We report here a case of a 12-year-old girl with bilateral nodules of the right upper eyelid and a left lower eyelid simulating epidermal cyst. The histopathologic examination of the excised nodule confirmed the diagnosis. To our knowledge, this may be the first bilateral case of eyelid SCN reported...

  19. Congenital bilateral perisylvian syndrome with hydrocephalus

    Directory of Open Access Journals (Sweden)

    Salih Hattapoğlu

    2012-12-01

    Full Text Available Congenital bilateral perisylvian syndrome (CBPS, whichis seen by indications of mental retardasyon, epilepsi,speech disorder and pseudobulbar palsy, is a diseasewhich comes up with genetic and non-genetic reasons.Revealing characteristic indications (like polymicrogyriawith MR imaging and clinic indications contributes makingdiagnosis. In present paper, we aimed to present 18month girl case report who diagnosed as CBPS with hydrocephaliindication. J Clin Exp Invest 2012; 3(4: 552-554Key words: Epilepsy, congenital bilateral perisylviansyndrome, polymicrogyria, hydrocephalus, pseudobulbarpalsy

  20. Bilateral hydronephrosis caused by vaginal prolapse

    Directory of Open Access Journals (Sweden)

    Begliomini Helio

    2003-01-01

    Full Text Available INTRODUCTION: Even though it is uncommon, uterine prolapse can cause compression of ureters and bilateral hydronephrosis, predisposing to arterial hypertension and renal failure. Hydronephrosis consequent to cystocele and to vaginal prolapse is even rarer. CASE REPORT: This paper reports on a 59 year-old patient, Caucasian, obese and hysterectomized who presented complete vaginal prolapse with bilateral hydronephrosis and slight alteration in serum urea and creatinine. Patient underwent correction of vaginal prolapse by endoscopic suspension technique with improvement of hydronephrosis and normalization of renal function. This work emphasizes the rarity of such case and the requirement of surgical approach.