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KMID : 0816120140170010052
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Korean Journal of Pediatric Gastroenterolology and Nutrition
2014 Volume.17 No. 1 p.52 ~ p.56
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Gastrointestinal Tract Involvement of Gorham’s Disease with Expression of D2-40 in Duodenum
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Choi Bong-Seok
Hong Suk-Jin
Chu Mi-Ae
Lee Seok-Jong
Lee Jong-Min
Bae Han-Ik
Choe Byung-Ho
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Abstract
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We present a case of a 13-year-old boy with Gorham’s disease involving the thoracic and lumbar spine, femur, and gastrointestinal (GI) tract, which was complicated by recurrent chylothorax and GI bleeding. The presenting symp-toms were intermittent abdominal pain, back pain, and melena. Esophagogastroduodenoscopy and colonoscopy showed no abnormal lesions, but duodenal biopsy showed marked dilation of the lymphatics in the mucosa and submucosa, which revealed positive staining with a D2-40 immunohistochemical marker. In cases of GI bleeding with osteolysis, the expression of a D2-40 marker in the lymphatic endothelium of the GI tract may help to diagnose GI involvement in Gorham’s disease. To the best of our knowledge, this is the first case report to pathologically demon-strate intestinal lymphatic malformation as a cause of GI bleeding in Gorham’s disease.
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KEYWORD
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Gorham disease, Osteolysis, Gastrointestinal tract, Melena, Occult blood, Child
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