Cases reported "Rare Diseases"

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1/49. The abdominal cocoon: a case report.

    The abdominal cocoon is a rare disease that is characterized by a total or partial encasement of the small bowel by a thick and fibrotic membrane. Thirty-five cases were reported since it was first described. It occurs primarily in females. Preoperative diagnosis is a matter of challenge and usually made at laparotomy. We report a patient with partial intestinal obstruction and abdominal cocoon which was diagnosed peri-operatively. We review the literature and discuss the etiology of this disease.
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2/49. Double omental hernia--case report on a very rare cause of intestinal obstruction.

    INTRODUCTION: We report a case of transomental herniation of the ileum through a defect in the gastrocolic ligament with re-emergence through a defect in the gastrohepatic ligament. This type of herniation is extremely rare and a review of the literature is presented. CLINICAL PRESENTATION: A 41-year-old Chinese male presented with signs and symptoms of intestinal obstruction. TREATMENT: An urgent laparotomy was undertaken and he was found to have a rare form of intra-abdominal hernia. The hernia was reduced and the defects were closed. OUTCOME: Postoperative recovery was unremarkable. CONCLUSION: Urgent operation should not be delayed because of the high mortality associated with strangulation.
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3/49. wandering spleen presenting as a right hypochondrial mass and intestinal obstruction.

    This is a case report of a 23 year old multiparous woman who presented with intestinal obstruction and a right hypochondrial mass. Laparatomy revealed an infarcted 1.4 Kg spleen in the right lumbar region compressing the ascending colon. There was also ileal volvulus around the splenic pedicle. This is probably the first documented case of wandering spleen in the right hypochondrium, presenting as right large bowel obstruction, to be reported in our region. wandering spleen is a rare condition, often asymptomatic, but may present as an acute abdomen. Pre-operative diagnosis is difficult and rarely made. Laboratory tests are seldom useful, but imaging studies do assist. Up to 1971 only 350 cases had been reported in the western literature. review of English literature from 1900 to 1991 reported only 51 cases in children. In our region 11 cases were reported in uganda between 1968 and 1971. No other literature is available from our region. Clinical presentation, aetiology, investigation, and management of wandering spleen is discussed.
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4/49. A rare case of metastatic ovarian carcinoma originating from primary intrahepatic cholangiocarcinoma. Case report.

    BACKGROUND: A rare case of metastatic ovarian carcinoma arising from intrahepatic cholangiocarcinoma is reported and the literature reviewed. CASE: A 49-year-old woman presented with ascites and a left pelvic mass. Optimal debulking surgery was carried out including a segmental resection of segment 5/6 of the liver. Histopathology confirmed an intrahepatic cholangiocarcinoma metastatic to the ovaries and omentum. CONCLUSION: Distinguishing a metastatic tumor from a primary ovarian tumoris critical for appropriate management. A high index of suspicion intraoperatively and subsequent expert pathological review are essential in making the correct diagnosis.
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5/49. Very severe aplastic anemia appearing after thymectomy.

    Aplastic anemia is a rare complication of thymoma and is extremely infrequent after thymectomy. We present a case of a 60-year-old woman with very severe aplastic anemia appearing sixteen months after thymectomy for a thymoma. She underwent thymectomy for a thymoma in April 2000. Preoperative examination revealed no hematologic abnormality. About sixteen months after the operation, she was readmitted because of pancytopenia with cough and fever. bone marrow aspiration revealed a very severe hypoplasia in all the three cell lines with over 80% fatty tissue, and chest CT revealed no recurrence of thymoma. Her aplastic anemia had responded to cyclosporine A and granulocyte-colony stimulating factor (G-CSF).
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6/49. Extension of adrenocortical carcinoma into the right atrium--echocardiographic diagnosis: a case report.

    BACKGROUND: adrenocortical carcinoma is a rare, highly malignant tumor. Cardiac involvement of the tumor is very rare. echocardiography facilitates the evaluation of the cardiac involvement of the tumor. CASE PRESENTATION: We describe a patient with an adrenal tumor. Transthoracic echo showed its extension into the right atrium. Accordingly, a combined abdominal and cardiac operation was performed, monitored by transesophageal echocardiography. CONCLUSION: This case highlights the importance of echocardiography in revealing the cardiac involvement by this tumor and in planning the operative procedure.
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7/49. Retrorectal cyst: a rare tumor frequently misdiagnosed.

    BACKGROUND: The rarity of retrorectal cysts and their nonspecific clinical presentations often lead to misdiagnoses and inappropriate operations. In recent years, several such patients have been referred to our institutions for evaluation and treatment of misdiagnosed retrorectal cysts. A review of these patients is presented. STUDY DESIGN: medical records of the colorectal surgery divisions at two institutions were reviewed. patients found to have previously misdiagnosed retrorectal cysts were identified. Preliminary diagnoses, radiologic examinations, operative procedures, and final diagnoses were obtained. RESULTS: Seven patients with retrorectal cysts who had been misdiagnosed before referral were identified. These patients had been treated for fistulae in ano, pilonidal cysts, perianal abscesses; psychogenic, lower back, posttraumatic, or postpartum pain, and proctalgia fugax before the correct diagnosis was made. patients underwent an average of 4.1 operative procedures. physical examination in combination with CT scanning made the correct diagnosis in all patients. All patients underwent successful resection through a parasacrococcygeal approach, and six of seven did not require coccygectomy. The resected tumors included four hamartomas, two epidermoid cysts, and one enteric duplication cyst. CONCLUSIONS: Retrorectal cysts are a rare entity that can be difficult to diagnose without a high index of clinical suspicion. A history of multiple unsuccessful procedures should alert the clinician to the diagnosis of retrorectal cyst. Once suspected, the correct diagnosis can be made with physical examination and a CT scan before a definitive surgical procedure.
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8/49. Calcified patent ductus arteriosus diagnosed following aortic valve replacement.

    A 71-year-old woman underwent aortic valve replacement for severe, symptomatic aortic stenosis. The left ventricle filled rapidly when the left ventricular vent was switched off and postoperatively she was slow to recover with bilateral pleural effusions. These findings prompted early reinvestigation, initially with echocardiography and subsequently with multi-detector row computed tomography. Using a retrospectively electrocardiographic-gated acquisition, adapted from a noninvasive coronary angiography protocol, a calcified, persistently patent ductus arteriosus was identified as the cause for her perioperative and postoperative condition. The defect has since been closed successfully using a transcatheter technique.
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9/49. Follicular dendritic cell sarcoma of the tonsil: report of a rare case.

    Follicular dendritic cell tumors are extremely rare. Only 17 cases have been previously described in the literature, and only three of them involved primary tumours of the oral cavity. We describe a new case of the latter, which occurred in a 51-year-old man who sought evaluation for a painless enlargement of his left palatine tonsil. The tonsil was excised, and histologic examination revealed that the tumor was a primary sarcoma that had arisen from the dendritic reticulum cells of the palatine tonsil. Postoperatively, the tumor site was treated with percutaneous irradiation (total dose: 70 Gy). After more than 5 years of follow-up, the patient showed no evidence of recurrence. We also discuss the salient features of the immunohistochemical examination.
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10/49. Huge lymphangioma of the tongue: a case report.

    lymphangioma of the tongue is relatively rare and may cause facial structural deformity. Using a combination of a V-shaped and central resection, we successfully treated a 6-year-old girl who had massive lymphangioma of the tongue. Postoperatively, her tongue was located completely within her mouth with good cosmetic results. Sensory and motor nerves to the tongue appeared to be intact. Her speech was also improved.
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