Cases reported "Chylothorax"

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1/13. chylothorax complicating Gorham's disease.

    Gorham's disease is a rare disorder characterized by a proliferation of thin-walled lymphatic vessels (lymphangiectasia) resulting in an osteolysis. A chylothorax is present in about one-fifth of the patients and carries a poor prognosis. In this circumstance, surgery including thoracic duct ligation, pleurodesis, and excision of involved tissue is probably the treatment of choice. It is facilitated by a precise radiological assessment using a computed tomographic scanning coupled with a lymphography. We report such a case.
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keywords = osteolysis
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2/13. chylothorax associated with massive osteolysis (Gorham's syndrome).

    We report a 15-year-old boy with bilateral chylothorax complicating Gorham's syndrome. thoracic duct ligation failed to prevent fluid reaccumulation. The patient died of lymphocytopenia. autopsy revealed vascular proliferation in the parietal and visceral pleura as well as in the ribs, which seemed to cause persistent chylothorax responsible for the poor prognosis of this patient. We reviewed treatments provided to 22 patients reported in the literature with Gorham's syndrome and chylothorax.
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keywords = osteolysis
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3/13. Mediastinal lymphangioma and chylothorax: thoracic involvement of Gorham's disease.

    We report a case of mediastinal lymphangioma associated with Gorham's disease in a 38-year-old man who had suffered recurrent clavicular fractures during a seven-year period. Mediastinal widening associated with osteolysis of the clavicles and the sternal manubrium was revealed by chest radiography, while computed tomography demonstrated a cystic anterior mediastinal mass infiltrating mediastinal fat and associated with osseous destruction of the clavicles and manubrium. chylothorax recurred during the course of the disease.
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ranking = 1
keywords = osteolysis
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4/13. chylothorax in Gorham's disease.

    A 25-yr-old woman presented with a right pleural effusion. Destruction of 9th through 12th ribs, adjacent vertebral bodies, and transverse processes was noted on plain radiograph and a large low-attenuated, irregular shaped mass lesion with peripheral rim enhancement, destroying vertebral body and transverse process, was revealed on the computed tomographic scan. magnetic resonance imaging showed high signal on T1- weighted image and iso- and low signal on T2-weighted image for the mass lesion replacing the vertebral bony cortex and marrow space. An open rib biopsy revealed the histopathological changes of Gorham's disease (essential osteolysis), even though only bloody fluid filling the empty space and rib and vertebral transverse process destruction were grossly observed on operation. Even though there was no definite response to radiotherapy and pleurodesis, the patient showed stable condition up to 20 months after diagnosis.
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ranking = 1
keywords = osteolysis
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5/13. Gorham Stout syndrome (disappearing bone disease): two additional case reports and a review of the literature.

    Gorham-Stout syndrome, or disappearing bone disease, is characterized by the proliferation of thin-walled vascular channels associated with regional osteolysis. There have been fewer than 150 cases reported in the literature. In this clinical report, we describe 2 additional cases of Gorham-Stout syndrome affecting the maxillofacial skeleton. We provide a review of the clinical diagnosis of this syndrome and describe treatment options.
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ranking = 1
keywords = osteolysis
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6/13. Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: a severe case of Gorham-Stout syndrome.

    The treatment of massive osteolysis with lymphangioma and/or hemangioma (Gorham-Stout syndrome) has been controversial. The authors report on a patient with multiple massive osteolyses and extensive lymph-hemangiomatosis whose lesions were reduced by interferon alfa therapy. A 2-year-old girl had complained of left chylothorax. thoracoscopy showed an increase in small lymphatic vessels in the chest wall. The chylothorax was improved by coagulation of the lymphatic vessels. Later, multiple massive osteolyses appeared in the left 11th and 12th ribs, the TH10-L3 vertebrae, and the right femur. There were also hemangiomas in the liver and spleen, a tumor lesion in the left lower chest wall, and hemangiomatous change on the skin surface of the left back. The left lung had only a minimal air content. After OK-432 was injected into the femur and chest wall lesions, the femur lesion disappeared. Then, as right chylothorax appeared, OK-432 was injected into the right pulmonary cavity. The chylothorax disappeared, but pericardial effusion appeared. After steroid pulse therapy, pericardial effusion disappeared. During these treatments, the 7th to 10th ribs disappeared from the x-ray and scoliosis developed. One month later, a cloudy fluid collection in the right lung was found on computed tomography. Interferon alfa and steroid pulse therapy were started. Interferon alfa (1,500,000 units) was subcutaneously administered daily for 2 months and was gradually reduced and maintained at 1,500,000 unit/wk. steroids were also reduced and maintained at 5 mg/d of predonine. Later, the progress of osteolysis and the extension of lymph-hemangiomatosis stopped. Ten months later, hemangioma in the back disappeared, and the 7th to 10th ribs, which had disappeared, reappeared. The interferon alfa therapy was stopped 14 months after it was administered. The patient's condition has been stable for 10 months since then. At this time, computed tomography shows regression of the hemangiomatous lesion in the back. The authors clinically diagnosed the patient as having Gorham-Stout syndrome with extension of lymph-hemangiomatosis. Interferon alfa with or without steroid therapy should be a choice for patients with extension lesions.
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ranking = 6
keywords = osteolysis
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7/13. A case of Gorham's disease with chylothorax treated curatively with radiation therapy.

    Gorham's disease, also known as vanishing bone disease or massive osteolysis, is a rare disorder of uncertain etiology. It is characterized by uncontrolled proliferation of vascular or lymphatic capillaries within bone, leading to resorption and replacement with angiomatous tissue. It can be complicated by chylous pericardial and pleural effusions, which can be life threatening. patients are also at risk of mortality or serious morbidity due to bone destruction, especially when the disease involves the spine. We report the case of a 31-year-old female with Gorham's disease involving several bones along with chylous pericardial and pleural effusions. She was effectively treated with definitive radiation therapy.
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ranking = 1
keywords = osteolysis
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8/13. Gorham disease: an intraoperative case study.

    Gorham disease is a rare chronic disorder that is characterized by the abnormal proliferation of thin-walled capillaries and small lymphatic vessels that results in the massive osteolysis of adjacent bone. Clinical manifestations are determined by the area of involvement, which may include the chest and ribs. The case presented involves a 47-year-old man with Gorham disease complicated by unilateral chylothorax who was treated with thoracic duct ligation. The anesthetic implications associated with Gorham disease are discussed, and nonsurgical primary and adjunctive treatments for chylothorax are summarized.
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keywords = osteolysis
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9/13. Disappearing bone disease: a case report and review of the literature.

    Disappearing bone disease is a rare condition manifested by massive osteolysis. Its etiology is unknown. Histologically, involved bones show a nonmalignant proliferation of thin-walled vessels. The mechanism of bone absorption remains unclear. No treatment has proven effective in arresting the disease. Cases in which there is extraosseous involvement have a much poorer prognosis.
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ranking = 1
keywords = osteolysis
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10/13. Massive Gorham osteolysis of the right hemipelvis complicated by chylothorax: report of a case in a 9-year-old boy successfully treated by pleurodesis.

    In a 9-year-old boy with progressive massive osteolysis (Gorham disease) of the right hemipelvis, the course was complicated by life-threatening chylothorax. A total of 42.6 L of chylous fluid was tapped over a 3-month period. A simple thoracoscopic intervention with tetracycline instillation was successful, and there has been no recurrence of the pleural effusion.
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ranking = 5
keywords = osteolysis
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