Non-islet cell tumor hypoglycemia concurrent with acromegalic features: A case report and literature review

BackgroundNon-islet cell tumor hypoglycemia (NICTH) is a rare cause of hypoglycemia due to the overproduction of high molecular weight insulin-like growth factor (big-IGF2), which activates the insulin receptor and subsequently caused hypoglycemia. But NICTH with acromegaly had rarely been reported....

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Main Authors: Xiaojing Wang, Naishi Li, Yi Xie, Liang Zhu, Ji Li, Feng Gu, Xinhua Xiao
Format: Article
Language:English
Published: Frontiers Media S.A. 2022-09-01
Series:Frontiers in Surgery
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fsurg.2022.968077/full
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author Xiaojing Wang
Xiaojing Wang
Naishi Li
Yi Xie
Liang Zhu
Ji Li
Feng Gu
Xinhua Xiao
author_facet Xiaojing Wang
Xiaojing Wang
Naishi Li
Yi Xie
Liang Zhu
Ji Li
Feng Gu
Xinhua Xiao
author_sort Xiaojing Wang
collection DOAJ
description BackgroundNon-islet cell tumor hypoglycemia (NICTH) is a rare cause of hypoglycemia due to the overproduction of high molecular weight insulin-like growth factor (big-IGF2), which activates the insulin receptor and subsequently caused hypoglycemia. But NICTH with acromegaly had rarely been reported. We firstly reported a rare case of NICTH concurrent with acromegalic facial features induced by a retroperitoneal hemangiopericytoma and reviewed similar cases in the literature.Case presentationA 30-year old man was admitted to hospital because of recurrent unconscious, which usually occurred in the late afternoon or early morning before supper or breakfast. On one unconscious occasion, his blood glucose was 2.4 mmol/L. His consciousness recovered rapidly with intravenous 50% glucose administration. Physical examination showed that he had coarse oily facial features with acne, prominent forehead and brow, broad nose, prominent nasolabial folds. At the time of hypoglycemia, suppressed serum insulin, GH and IGF-1 levels was found. Computed Tomography further revealed a large left retroperitoneal mass measuring 7.0 cm × 12.3 cm × 13.0 cm. He underwent complete surgical resection of the mass. Surgical pathology demonstrated a hemangiopericytoma and strong positive for IGF-2. He did not experience further episodes of hypoglycemia after the operation during the 2.5 years follow-up.ConclusionsFibrous origin is the most common tumor type for NICTH with acromegaly features. NICTH should be considered in non-diabetic patients who have recurrent hypoglycemia along with suppressed serum insulin and IGF-1 levels.
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spelling doaj.art-a9087c15ecb64385a30754a8b6d9992b2022-12-22T03:47:55ZengFrontiers Media S.A.Frontiers in Surgery2296-875X2022-09-01910.3389/fsurg.2022.968077968077Non-islet cell tumor hypoglycemia concurrent with acromegalic features: A case report and literature reviewXiaojing Wang0Xiaojing Wang1Naishi Li2Yi Xie3Liang Zhu4Ji Li5Feng Gu6Xinhua Xiao7Department of Endocrinology, Key Laboratory of Endocrinology of National Health Commission, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, ChinaDepartment of Endocrinology, Beijing Tsinghua Changgung Hospital, Tsinghua University, Beijing, ChinaDepartment of Endocrinology, Key Laboratory of Endocrinology of National Health Commission, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, ChinaDepartment of Urology Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, ChinaDepartment of Radiology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, ChinaDepartment of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, ChinaDepartment of Endocrinology, Key Laboratory of Endocrinology of National Health Commission, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, ChinaDepartment of Endocrinology, Key Laboratory of Endocrinology of National Health Commission, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, ChinaBackgroundNon-islet cell tumor hypoglycemia (NICTH) is a rare cause of hypoglycemia due to the overproduction of high molecular weight insulin-like growth factor (big-IGF2), which activates the insulin receptor and subsequently caused hypoglycemia. But NICTH with acromegaly had rarely been reported. We firstly reported a rare case of NICTH concurrent with acromegalic facial features induced by a retroperitoneal hemangiopericytoma and reviewed similar cases in the literature.Case presentationA 30-year old man was admitted to hospital because of recurrent unconscious, which usually occurred in the late afternoon or early morning before supper or breakfast. On one unconscious occasion, his blood glucose was 2.4 mmol/L. His consciousness recovered rapidly with intravenous 50% glucose administration. Physical examination showed that he had coarse oily facial features with acne, prominent forehead and brow, broad nose, prominent nasolabial folds. At the time of hypoglycemia, suppressed serum insulin, GH and IGF-1 levels was found. Computed Tomography further revealed a large left retroperitoneal mass measuring 7.0 cm × 12.3 cm × 13.0 cm. He underwent complete surgical resection of the mass. Surgical pathology demonstrated a hemangiopericytoma and strong positive for IGF-2. He did not experience further episodes of hypoglycemia after the operation during the 2.5 years follow-up.ConclusionsFibrous origin is the most common tumor type for NICTH with acromegaly features. NICTH should be considered in non-diabetic patients who have recurrent hypoglycemia along with suppressed serum insulin and IGF-1 levels.https://www.frontiersin.org/articles/10.3389/fsurg.2022.968077/fullnon-islet cell tumor hypoglycemiaacromegalyIGF-2solitary fibrous tumorhemangiopericytoma
spellingShingle Xiaojing Wang
Xiaojing Wang
Naishi Li
Yi Xie
Liang Zhu
Ji Li
Feng Gu
Xinhua Xiao
Non-islet cell tumor hypoglycemia concurrent with acromegalic features: A case report and literature review
Frontiers in Surgery
non-islet cell tumor hypoglycemia
acromegaly
IGF-2
solitary fibrous tumor
hemangiopericytoma
title Non-islet cell tumor hypoglycemia concurrent with acromegalic features: A case report and literature review
title_full Non-islet cell tumor hypoglycemia concurrent with acromegalic features: A case report and literature review
title_fullStr Non-islet cell tumor hypoglycemia concurrent with acromegalic features: A case report and literature review
title_full_unstemmed Non-islet cell tumor hypoglycemia concurrent with acromegalic features: A case report and literature review
title_short Non-islet cell tumor hypoglycemia concurrent with acromegalic features: A case report and literature review
title_sort non islet cell tumor hypoglycemia concurrent with acromegalic features a case report and literature review
topic non-islet cell tumor hypoglycemia
acromegaly
IGF-2
solitary fibrous tumor
hemangiopericytoma
url https://www.frontiersin.org/articles/10.3389/fsurg.2022.968077/full
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