고인슐린혈성 저혈당을 보인 성인에서의 Nesidiodysplasia 1예
A case of nesidiodysplasia in adult with hyperinsulinemic hypoglycemia
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초록
고인슐린혈성 저혈당의 원인 중 nesidioblastosis와 nesidiodysplasia는 신생아 및 유아기에는 가장 많은 원 인 질환이나 어른에서는 매우 드물고 여전히 진단 및 치 료에 있어서 논란이 많다. 저자 등은 의식저하를 주소로 내원한 성인에서 고인슐린혈성 저혈당으로 진단하여 처 음에는 인슐린종을 의심하여 종양 확인을 위한 검사를 시행하여 경피적 경감문맥 정맥혈 채취술과 수술 중 시 행한 초음파에서 종양이 의심되는 결절이 있어서 적출 하였으나, 수술 후 병리조직 소견으로 nesidiodysplasia 가 진단된 1예를 경험하였기에 이에 문헌고찰과 함께 보 고하는 바이다.
Abstract
Nesidiodysplasia encompasses increased, maldistributed, and/or, improperly regulated or programmed endocrine cells associated with a clinical endocrine dysfunction. It is a frequent cause of hyperinsulinemic hypoglycemia in neonates and infants, but rare in adults. A 63 year-old woman was admitted due to the loss of consciousness and intense sweating. Hyperinsulinemic hypogiycemia was diagnosed. Under the presumptive diagnosis of insulinoma, abdominal ultrasonography and CT scan were done, but no definite tumor was found. Only a suspicious gradient in insulin concentration was found around the body of the pancreas by percutaneous portal venous sampling. While an exploratory laparotomy was performed, an intraoperative sonography was done. As a result, cystic mass like lesion was found. Then mass enucleation was done. Pathologic examination of the enucleated pancreas revealed no mass and the characteristic nesidiodysplasia. However, after the operation, hypoglycemic symptoms were repeated. So we are instructing her to eat meals frequently.(Korean J Med 64:695-699, 2003)