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Unmasked insulinoma occasioned by severe hypoglycemic coma immediately postpartum: a case report

BACKGROUND: Insulinoma in women during pregnancy and postpartum is very rare; approximately 65% of cases are diagnosed early in pregnancy and ~ 35% immediately after delivery, few being found in middle or late pregnancy, likely due to increased insulin resistance seen after early-stage pregnancy. We...

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Autores principales: Matsumoto, Kiyoshi, Watanabe, Miyu, Takao, Ken, Takahashi, Hirokazu, Daido, Hisashi, Shibata, Toshiro, Hirose, Tokuyuki, Kato, Takehiro, Mizuno, Masami, Hirota, Takuo, Suwa, Tetsuya, Horikawa, Yukio, Murakami, Takaaki, Yabe, Daisuke
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10413590/
https://www.ncbi.nlm.nih.gov/pubmed/37563593
http://dx.doi.org/10.1186/s12902-023-01415-1
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author Matsumoto, Kiyoshi
Watanabe, Miyu
Takao, Ken
Takahashi, Hirokazu
Daido, Hisashi
Shibata, Toshiro
Hirose, Tokuyuki
Kato, Takehiro
Mizuno, Masami
Hirota, Takuo
Suwa, Tetsuya
Horikawa, Yukio
Murakami, Takaaki
Yabe, Daisuke
author_facet Matsumoto, Kiyoshi
Watanabe, Miyu
Takao, Ken
Takahashi, Hirokazu
Daido, Hisashi
Shibata, Toshiro
Hirose, Tokuyuki
Kato, Takehiro
Mizuno, Masami
Hirota, Takuo
Suwa, Tetsuya
Horikawa, Yukio
Murakami, Takaaki
Yabe, Daisuke
author_sort Matsumoto, Kiyoshi
collection PubMed
description BACKGROUND: Insulinoma in women during pregnancy and postpartum is very rare; approximately 65% of cases are diagnosed early in pregnancy and ~ 35% immediately after delivery, few being found in middle or late pregnancy, likely due to increased insulin resistance seen after early-stage pregnancy. We successfully treated a case of insulinoma in which severe hypoglycemic coma immediately after delivery occasioned detailed investigation and diagnosis. CASE PRESENTATION: Our patient experienced hypoglycemic coma in the 3(rd) month of pregnancy (initially considered due to her hyperemesis gravidarum) that improved spontaneously during the gestational period. No abnormalities of plasma glucose or body weight were found in regular checkups during her pregnancy; however, recurrence of hypoglycemic coma after delivery led us to suspect insulinoma. While contrast enhanced computer tomography and endoscopic ultrasonography (EUS) initially failed to detect a tumor in the pancreas, selective arterial calcium stimulation test revealed an insulin-secreting tumor localized in the pancreatic body. She then underwent spleen-preserving distal pancreatectomy; a 10-mm tumor positive for chromogranin A, synaptophysin and insulin was identified. CONCLUSIONS: Although pregnancy can mask insulinoma-associated symptoms and make diagnosis challenging, hypoglycemic episodes during early pregnancy, which were observed in this case, are suggestive of insulinoma. Importantly, in this case, accurate preoperative localization of the tumor enabled prompt curative surgery after delivery. Thus, clinical vigilance for the occurrence of insulinoma and its localization is appropriate for pregnant women suffering severe hypoglycemia.
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spelling pubmed-104135902023-08-11 Unmasked insulinoma occasioned by severe hypoglycemic coma immediately postpartum: a case report Matsumoto, Kiyoshi Watanabe, Miyu Takao, Ken Takahashi, Hirokazu Daido, Hisashi Shibata, Toshiro Hirose, Tokuyuki Kato, Takehiro Mizuno, Masami Hirota, Takuo Suwa, Tetsuya Horikawa, Yukio Murakami, Takaaki Yabe, Daisuke BMC Endocr Disord Case Report BACKGROUND: Insulinoma in women during pregnancy and postpartum is very rare; approximately 65% of cases are diagnosed early in pregnancy and ~ 35% immediately after delivery, few being found in middle or late pregnancy, likely due to increased insulin resistance seen after early-stage pregnancy. We successfully treated a case of insulinoma in which severe hypoglycemic coma immediately after delivery occasioned detailed investigation and diagnosis. CASE PRESENTATION: Our patient experienced hypoglycemic coma in the 3(rd) month of pregnancy (initially considered due to her hyperemesis gravidarum) that improved spontaneously during the gestational period. No abnormalities of plasma glucose or body weight were found in regular checkups during her pregnancy; however, recurrence of hypoglycemic coma after delivery led us to suspect insulinoma. While contrast enhanced computer tomography and endoscopic ultrasonography (EUS) initially failed to detect a tumor in the pancreas, selective arterial calcium stimulation test revealed an insulin-secreting tumor localized in the pancreatic body. She then underwent spleen-preserving distal pancreatectomy; a 10-mm tumor positive for chromogranin A, synaptophysin and insulin was identified. CONCLUSIONS: Although pregnancy can mask insulinoma-associated symptoms and make diagnosis challenging, hypoglycemic episodes during early pregnancy, which were observed in this case, are suggestive of insulinoma. Importantly, in this case, accurate preoperative localization of the tumor enabled prompt curative surgery after delivery. Thus, clinical vigilance for the occurrence of insulinoma and its localization is appropriate for pregnant women suffering severe hypoglycemia. BioMed Central 2023-08-10 /pmc/articles/PMC10413590/ /pubmed/37563593 http://dx.doi.org/10.1186/s12902-023-01415-1 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Matsumoto, Kiyoshi
Watanabe, Miyu
Takao, Ken
Takahashi, Hirokazu
Daido, Hisashi
Shibata, Toshiro
Hirose, Tokuyuki
Kato, Takehiro
Mizuno, Masami
Hirota, Takuo
Suwa, Tetsuya
Horikawa, Yukio
Murakami, Takaaki
Yabe, Daisuke
Unmasked insulinoma occasioned by severe hypoglycemic coma immediately postpartum: a case report
title Unmasked insulinoma occasioned by severe hypoglycemic coma immediately postpartum: a case report
title_full Unmasked insulinoma occasioned by severe hypoglycemic coma immediately postpartum: a case report
title_fullStr Unmasked insulinoma occasioned by severe hypoglycemic coma immediately postpartum: a case report
title_full_unstemmed Unmasked insulinoma occasioned by severe hypoglycemic coma immediately postpartum: a case report
title_short Unmasked insulinoma occasioned by severe hypoglycemic coma immediately postpartum: a case report
title_sort unmasked insulinoma occasioned by severe hypoglycemic coma immediately postpartum: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10413590/
https://www.ncbi.nlm.nih.gov/pubmed/37563593
http://dx.doi.org/10.1186/s12902-023-01415-1
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