Insulinoma with suspected mutant somatostatin receptor expression according to histological examination

Key Clinical Message This case highlights the possibility of an insulinoma expressing an aberrant form of SSTRs resulting in a discrepancy between the preoperative octreotide assessment and postoperative SSTR expression. Abstract Insulinoma is a pancreatic disease that causes hyperinsulinemic hypogl...

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Main Authors: Shunichiro Onish, Aki Takada‐Watanabe, Robert Y. Osamura, Takayuki Shiomi, Hiroyuki Kusano, Yoshiro Maezawa, Hiroyuki Murai, Makoto Miyabayashi, Sakutaro Koike, Tomohiko Yoshida, Minoru Takemoto
Format: Article
Language:English
Published: Wiley 2024-11-01
Series:Clinical Case Reports
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Online Access:https://doi.org/10.1002/ccr3.9390
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author Shunichiro Onish
Aki Takada‐Watanabe
Robert Y. Osamura
Takayuki Shiomi
Hiroyuki Kusano
Yoshiro Maezawa
Hiroyuki Murai
Makoto Miyabayashi
Sakutaro Koike
Tomohiko Yoshida
Minoru Takemoto
author_facet Shunichiro Onish
Aki Takada‐Watanabe
Robert Y. Osamura
Takayuki Shiomi
Hiroyuki Kusano
Yoshiro Maezawa
Hiroyuki Murai
Makoto Miyabayashi
Sakutaro Koike
Tomohiko Yoshida
Minoru Takemoto
author_sort Shunichiro Onish
collection DOAJ
description Key Clinical Message This case highlights the possibility of an insulinoma expressing an aberrant form of SSTRs resulting in a discrepancy between the preoperative octreotide assessment and postoperative SSTR expression. Abstract Insulinoma is a pancreatic disease that causes hyperinsulinemic hypoglycemia. The first‐line treatment is surgery; however, somatostatin derivatives are administered in cases where surgery is not a viable option and to prevent preoperative hypoglycemia. Here, we report a case in which preoperative examination indicated a potential tumor with low somatostatin receptor 2 (SSTR2) and SSTR5 expression, whereas postoperative pathological examination indicated strong SSTR expression. We report the case of a 69‐year‐old Japanese female who experienced hypoglycemia‐like symptoms for a decade such as sweating, fatigue, hunger, and confusion with an increase in episode frequency per year. Dynamic computed tomography revealed a 13‐mm diameter nodule and aberrant blood flow in the pancreatic tail. Subsequently, the patient was diagnosed with pancreatic insulinoma. A preoperative octreotide test did not relieve hypoglycemia, and no uptake of 111indium‐pentetreotide was observed, suggesting an insulinoma with low SSTR expression. However, postoperative histological studies suggested that the intracellular domain of SSTRs were highly expressed, while the extracellular domain may be mutated. We present a rare case of insulinoma expressing an aberrant form of SSTRs resulting in a discrepancy between the preoperative octreotide assessment and postoperative SSTR expression.
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spelling doaj-art-3aec445c0b56421487ef9d4fd25e753b2025-08-20T02:23:34ZengWileyClinical Case Reports2050-09042024-11-011211n/an/a10.1002/ccr3.9390Insulinoma with suspected mutant somatostatin receptor expression according to histological examinationShunichiro Onish0Aki Takada‐Watanabe1Robert Y. Osamura2Takayuki Shiomi3Hiroyuki Kusano4Yoshiro Maezawa5Hiroyuki Murai6Makoto Miyabayashi7Sakutaro Koike8Tomohiko Yoshida9Minoru Takemoto10Department of Diabetes, Metabolism, and Endocrinology International University of Health and Welfare, Narita Hospital Chiba JapanDepartment of Endocrinology, Hematology and Gerontology Chiba University Graduate School of Medicine Chiba JapanDepartment of Diagnostic Pathology Nippon Koukan Hospital Kawasaki City JapanDepartment of Anatomic Pathology International University of Health and Welfare, Narita Hospital Chiba JapanDepartment of Anatomic Pathology International University of Health and Welfare, Narita Hospital Chiba JapanDepartment of Endocrinology, Hematology and Gerontology Chiba University Graduate School of Medicine Chiba JapanDepartment of Neurology International University of Health and Welfare, Narita Hospital Chiba JapanDepartment of Endocrinology, Hematology and Gerontology Chiba University Graduate School of Medicine Chiba JapanSchool of Medicine International University of Health and Welfare Chiba JapanDepartment of Diabetes, Metabolism, and Endocrinology International University of Health and Welfare, Narita Hospital Chiba JapanDepartment of Diabetes, Metabolism, and Endocrinology International University of Health and Welfare, Narita Hospital Chiba JapanKey Clinical Message This case highlights the possibility of an insulinoma expressing an aberrant form of SSTRs resulting in a discrepancy between the preoperative octreotide assessment and postoperative SSTR expression. Abstract Insulinoma is a pancreatic disease that causes hyperinsulinemic hypoglycemia. The first‐line treatment is surgery; however, somatostatin derivatives are administered in cases where surgery is not a viable option and to prevent preoperative hypoglycemia. Here, we report a case in which preoperative examination indicated a potential tumor with low somatostatin receptor 2 (SSTR2) and SSTR5 expression, whereas postoperative pathological examination indicated strong SSTR expression. We report the case of a 69‐year‐old Japanese female who experienced hypoglycemia‐like symptoms for a decade such as sweating, fatigue, hunger, and confusion with an increase in episode frequency per year. Dynamic computed tomography revealed a 13‐mm diameter nodule and aberrant blood flow in the pancreatic tail. Subsequently, the patient was diagnosed with pancreatic insulinoma. A preoperative octreotide test did not relieve hypoglycemia, and no uptake of 111indium‐pentetreotide was observed, suggesting an insulinoma with low SSTR expression. However, postoperative histological studies suggested that the intracellular domain of SSTRs were highly expressed, while the extracellular domain may be mutated. We present a rare case of insulinoma expressing an aberrant form of SSTRs resulting in a discrepancy between the preoperative octreotide assessment and postoperative SSTR expression.https://doi.org/10.1002/ccr3.9390hypoglycemiainsulinomaoctreotidesomatostatin receptor
spellingShingle Shunichiro Onish
Aki Takada‐Watanabe
Robert Y. Osamura
Takayuki Shiomi
Hiroyuki Kusano
Yoshiro Maezawa
Hiroyuki Murai
Makoto Miyabayashi
Sakutaro Koike
Tomohiko Yoshida
Minoru Takemoto
Insulinoma with suspected mutant somatostatin receptor expression according to histological examination
Clinical Case Reports
hypoglycemia
insulinoma
octreotide
somatostatin receptor
title Insulinoma with suspected mutant somatostatin receptor expression according to histological examination
title_full Insulinoma with suspected mutant somatostatin receptor expression according to histological examination
title_fullStr Insulinoma with suspected mutant somatostatin receptor expression according to histological examination
title_full_unstemmed Insulinoma with suspected mutant somatostatin receptor expression according to histological examination
title_short Insulinoma with suspected mutant somatostatin receptor expression according to histological examination
title_sort insulinoma with suspected mutant somatostatin receptor expression according to histological examination
topic hypoglycemia
insulinoma
octreotide
somatostatin receptor
url https://doi.org/10.1002/ccr3.9390
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