Endocrinology and Metabolism

Case Reports

Diffuse Nesidioblastosis of the Pancreasin Adult with Persistent Hyperinsulinemic Hypoglycemia.: Seoung Ha Lee, Kean Young Hyoung, Geom Seog Seo, Bong Joo Shin, Chung Gu Cho, Kwang Soo Yang, Kwon Mook Chae, Ki Jung Yun; J Korean Endocr Soc. 1996;11(2):247-253. Published online November 7, 2019

Abstract PDF: Nesidioblastosis is a term that describes multifocal hyperplasia of all panereatic cell components and is characterized primarily by their disorganization and proliferation throughout the entire panaeas. Adult onset nesidioblastosis is an extremely rare entity associated with hypersecretion of insulin. The authors have recently experieneed a case of nesidioblastosis in an adult. A 41-year old man was admitted due to interrnittenr hypoglycemic symptoms, that had been relieved by carbohydrate ingestion. Hyperinsulinemic hypoglycemia was documented during prolonged fast. Under the presumptive diagnosis of insulinoma, abdominal CT, celiac angiogram and percutaneous transhepatic portal venous sampling were done but we could not find any definitive mass. Eight-five percent of the panacas was removed. Pathologic examination of the resected pancreas revealed irregularly sized islets and scattering of small endocrine cell clusters throughout the acinar tissue and ductuloinsular complex.

A Case of Nesidioblastosis in Adult with Hyperinsulinemic Hypoglycemia.: Young Kee Shong, Hong Kyu Kim, Young Joo Min, Joong Yeol Park, Sung Kwan Hong, Ki Up Lee, Duck Jong Han, Ho Jeong Lee, Ghi Su Kim, Gyung Yub Gong; J Korean Endocr Soc. 1994;10(3):273-277. Published online November 6, 2019

Abstract PDF: Nesidioblastosis is a primarily childhood disease which is often associated with hyperinsulinemic hypoglycemia. It is very rarely found in adults. Only a few well documented cases are found in the world interature. The authors have recently experienced a case of nesidioblastosis in an adult. A 55-year-old man was admitted due to repeated episodes of reversible loss of consciousness. Hyperinsulinemic hypoglycemia was documented. Under the presumptive diagnosis of insulinoma, localization procedures were done but no definite tumor was found. Only suspicious gradient in insulin concentration was found around the head of pancreas by percutaneous transhepatic portal venous sampling. Exploratory laparotomy was performed and Whipple's operation was done. Seventy percent of proximal pancreas was removed. Histomorphometric study of the resected specimen revealed uneven graded hyperplasia of the islet cells with the most profuse hyperplasia in the head region and progressive decrease in the degree of hyperplasia to the body and tail. The patient remains euglycemia until 6 months after operation since immediate postoperative period and can tolerate 24 hour fasting without any medication.

A Case of Focal Type Nesidioblastosis in Adult Treated with Distal Pancreatectomy.: Dae Gyu Seo, Wang Sik Shin, Gil Sung Han, Jae Gun Jung, Ho Sang Shon, Ki Sung Ahn, Hoon Kyu Oh, Jae bok Park, Young Jin Kim; J Korean Endocr Soc. 1994;10(2):136-141. Published online November 6, 2019

Abstract PDF: Nesidioblastosis, the process of differentiation of pancreatic islets from ductular epithelium, is a frequent cause of hyperinsulinemic hypoglycemia in neonates and infants, but rare in adults. A 38-year-old man had suffered from intermittent hypoglycemic symtoms for 10 years. He had inappropriately elevated plasma immunoreactive insulin level and I/G ratio during a prolonged fast. There was no evidence of tumor on the abdominal CT scan and operation although celiac angiography showed a round mass like lesion in the distal pancreatic area.His hypoglycemic symptoms were improved by distal pancreatectomy.Pathologic finding in the resected pancreas was that of focal type nesidioblastosis I.e, increased size and number of islets with scattered numerous small islets around ductules in focal lesion. Distal pancreatectomy as an initial operation may be effective in the treatment of focal type nesidioblastosis.

Original Article

Nesidioblastosis in an Elderly Patient with Hyperinsulinemic Hypoglycemia.: Ye Kyung Seo, Jik Hwa Nam, Byung Ho Sin, Jung Guk Kim, Sung Woo Ha, Bo Whn Kim, Sang Won Jung, Young Guk Yun, In Su Seo, Chang Ho Cho; J Korean Endocr Soc. 1997;12(3):485-492. Published online January 1, 2001

Abstract PDF: Nesidioblastosis is characterized by a diffuse proliferation of islet cells arising from pancreatic ducts and is the most common cause of hyperinsulinemic hypoglycemia in newborns and infantile. It is exceedingly rare in adults and no concensus regarding its diagnosis and management is available. We herein describe an elderly man with fasting hypoglycemia, inappropriate insulin hypersecretion. And pathologic examination of his pancreas revealed the characteristic finding of nesidioblastosis confirmed by immunohistochemical stain.

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