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다발성 글루카곤 생성 내분비세포종을 동반한 췌장 알파세포의 Nesidioblastosis 및 과다증식 1예

Other Titles
 A case of alpha-cell nesidioblastosis and hyperplasia with multiple glucagon-producing endocrine cell tumor of the pancreas 
Authors
 강화평  ;  김세화  ;  임태섭  ;  이혜원  ;  최흔  ;  강창무  ;  김호근  ;  방승민 
Citation
 Korean Journal of Gastroenterology, Vol.63(4) : 253-257, 2014 
Journal Title
Korean Journal of Gastroenterology
ISSN
 1598-9992 
Issue Date
2014
MeSH
Adult ; Chromogranin A/blood ; Female ; Glucagon/metabolism* ; Glucagon-Secreting Cells/metabolism ; Humans ; Hyperplasia/complications ; Hyperplasia/diagnosis* ; Islets of Langerhans/diagnostic imaging ; Islets of Langerhans/metabolism ; Nesidioblastosis/complications ; Nesidioblastosis/diagnosis* ; Neuroendocrine Tumors/complications ; Neuroendocrine Tumors/diagnosis* ; Neuroendocrine Tumors/pathology ; Pancreas/pathology* ; Tomography, X-Ray Computed ; Ultrasonography
Abstract
Nesidioblastosis is a term used to describe pathologic overgrowth of pancreatic islet cells. It also means maldistribution of islet cells within the ductules of exocrine pancreas. Generally, nesidioblastosis occurs in beta-cell and causes neonatal hyperinsulinemic hypoglycemia or adult noninsulinoma pancreatogenous hypoglycemia syndrome. Alpha-cell nesidioblastosis and hyperplasia is an extremely rare disorder. It often accompanies glucagon-producing marco- and mircoadenoma without typical glucagonoma syndrome. A 35-year-old female was referred to our hospital with recurrent acute pancreatitis. On radiologic studies, 1.5 cm sized mass was noted in pancreas tail. Cytological evaluation with EUS-fine-needle aspiration suggested serous cystadenoma. She received distal pancreatectomy. The histologic examination revealed a 1.7 cm sized neuroendocrine tumor positive for immunohistochemical staining with glucagon antibody. Multiple glucagon-producing micro endocrine cell tumors were scattered next to the main tumor. Additionally, diffuse hyperplasia of pancreatic islets and ectopic proliferation of islet cells in centroacinar area, findings compatible to nesidioblastosis, were seen. These hyperplasia and almost all nesidioblastic cells were positive for glucagon immunochemistry. Even though serum glucagon level still remained higher than the reference value, she has been followed-up without any evidence of recurrence or hormone related symptoms. Herein, we report a case of alpha-cell nesidioblastosis and hyperplasia combined with glucagon-producing neuroendocrine tumor with literature review.
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Appears in Collections:
1. College of Medicine (의과대학) > Research Institute (부설연구소) > 1. Journal Papers
1. College of Medicine (의과대학) > Dept. of Internal Medicine (내과학교실) > 1. Journal Papers
1. College of Medicine (의과대학) > Dept. of Pathology (병리학교실) > 1. Journal Papers
1. College of Medicine (의과대학) > Dept. of Surgery (외과학교실) > 1. Journal Papers
Yonsei Authors
Kang, Chang Moo(강창무) ORCID logo https://orcid.org/0000-0002-5382-4658
Kang, Huapyong(강화평) ORCID logo https://orcid.org/0000-0003-1790-0809
Kim, Se Hwa(김세화)
Kim, Hogeun(김호근)
Bang, Seungmin(방승민) ORCID logo https://orcid.org/0000-0001-5209-8351
Lee, Hye Won(이혜원) ORCID logo https://orcid.org/0000-0002-3552-3560
Lim, Tae Seop(임태섭) ORCID logo https://orcid.org/0000-0002-4578-8685
Choi, Heun(최흔) ORCID logo https://orcid.org/0000-0002-9622-9381
URI
https://ir.ymlib.yonsei.ac.kr/handle/22282913/165966
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