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당뇨병성 케톤산증을 동반한 십이지장소마토스타틴종 1 예
남문석(Moon Suk Nam),이은직(Eun Jig Lee),조재화(Jae Hwa Cho),이홍우(Hong Woo Lee),정준근(Jun Keun Chung),김경래(Kyung Rae Kim),이현철(Hyun Chul Lee),이희대(Hy De Lee),김기황(Ki Hwang Kim),정현주(Hyeon Joo Jeong),허갑범(Kap Bum Huh) 대한내과학회 1995 대한내과학회지 Vol.48 No.6
The syndrome induced by excessive somatostatin secretion in somatostatinoma is characterized by steatorrhea, hypochlorhydria, cholelithiasis, and mild non-ketotic hyperglycemia. Since the first case of pancreatic somatostatinoma reported by Larsson et al in 1977, approximately 25 cases of pancreatic somatostatinama have been reported. Extrapancreatic somatostatinoma(mainly gastrointestinal somatostatinoma) was relatively rare, and first described by Kaneko et al in 1979. Gastrointestinal somatostatinomas are somewhat different from pancreatic somatostatinomas. Somatostatinoma syndrome is rarely seen in gastrointestinal somatostatinoma because of its low level of somatostatin. Due to the fact that somatostatin inhibits the secretion of insulin and glucagon simultaneously, the absence of diabetic ketoacidosis has been regarded. Jackson et al first described the diabetic ketoacidosis in the patient with malignant extrapancreatic somatostatinoma originated from the lung. As far as we can determine, intestinal somatostatinoma presenting with diabetic ketoacidosis has not been reported previously. In our case, the increased circulating level of somatostatin was detected and fasting insulin and C-peptide levels were suppressed, but plasma glucagon was not suppressed below normal level. Diabetic ketoacidosis was promptly corrected with insulin replacement and supportive fluid therapies, and after operation, the hyperglycemia was disappeared without insulin replacement. The mass was located at the second portion of duodenum and oval shaped, solid mass was seperated from the head of pancreas by a thick fibrous capsule. Light microscopic examination revealed that each tumor cells were polygonal shaped and had plenty of finely granular cytoplasm. The immunohistochemical stainings of tumor cells showed positive immunoreactivity for chromogranin and somatostatin but insulin, glucagon, calcitonin, and serotonin were negatively stained. The electronicroscopy of tumor cells showed electron-dense secretory granules and tonofilament bundles in cytoplasm. We presented a case of duodenal somatostatinoma in whom the diagnosis was recognized after presentation with diabetic ketoacidosis.