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MON-141 Non-Islet Cell Tumor Hypoglycemia Due to Relapsed Uterine Carcinosarcoma
Background: Non-islet cell tumor hypoglycemia is a rare cause of spontaneous hypoglycemia caused by abnormal high molecular weight IGF-2 production. Clinical Case: 78yo woman with history of uterine carcinosarcoma and DM type two was admitted to the medicine service for altered mental status, acute...
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Endocrine Society
2019
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6550857/ http://dx.doi.org/10.1210/js.2019-MON-141 |
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author | Caraccio, Donald |
author_facet | Caraccio, Donald |
author_sort | Caraccio, Donald |
collection | PubMed |
description | Background: Non-islet cell tumor hypoglycemia is a rare cause of spontaneous hypoglycemia caused by abnormal high molecular weight IGF-2 production. Clinical Case: 78yo woman with history of uterine carcinosarcoma and DM type two was admitted to the medicine service for altered mental status, acute kidney injury, and hypoglycemia. Her lowest glucose in the emergency room was 36mg/dl (capillary) and 48mg/dl (plasma). She denied any diaphoresis, palpitations or tremulousness while hypoglycemic. She did endorse feeling “sleepy”, intermittently, which would improve after eating. She reported poor appetite, early satiety, and lower abdominal pain for the past several weeks. She denied recent weight loss. For diabetes treatment, she was on metformin 850mg daily at home. Her hemoglobin A1C was 4.8%, down from 5.6% one year prior. Random cortisol was 17.3ug/dl (1.7-14.1). Urine culture grew strep anginosus and blood cultures were negative. After initial stabilization of glucose with dextrose infusion, she was taken off dextrose containing fluids to better evaluate the hypoglycemia. Approximately 24 hours later, she was noted to have a plasma glucose of 52mg/dl. She reported feeling her typical somnolence that improved after she consumed glucose, fulfilling Whipple’s Triad. While hypoglycemic, her insulin level was <0.5mU/L (3.0-25.0), proinsulin 4.7pmol/L (3.6-22), and c-peptide 0.3ng/L (1.1-4.4); consistent with non insulin mediated hypoglycemia. Sulfonylurea screen was negative. Betahydroxybutyrate was 0.12mmol/L (0.02-0.27); which was not consistent with starvation and glycogen depletion. IGF 1 was 14ng/ml (34-182) and IGF2 was 708ng/ml (333-967). The calculated IGF2: IGF1 ratio was 50.6. In reviews of patients with non islet cell tumor hypoglycemia, a ratio above 10 is highly suggestive of this diagnosis and should lead to imaging evaluation for malignancy (1). Her CT abdomen and pelvis that showed recurrent metastatic disease consistent with her history of uterine carcinosarcoma. She was started on hydrocortisone 30mg IV every 12 hours before transitioning to prednisone 15mg daily the following day. She had no recurrences of hypoglycemia after starting on glucocorticoids. Unfortunately, her hospital course was complicated by DVT and intraperitoneal hemorrhage resulting in hypovolemic shock and renal failure. Patient died on hospital day 16 after transitioning to comfort care only. Conclusion: This case illustrates that non-islet cell tumor hypoglycemia will often present with neuroglycopenic symptoms, but without autonomic symptoms due to recurrent hypoglycemic events. Hypoglycemia can be improved with glucocorticoids, although tumor directed treatment is needed for cure. References:Fukuda I, Hizuka N, Ishikawa Y, et al. Clinical features of insulin-like growth factor-II producing non-islet-cell tumor hypoglycemia. Growth Horm IGF Res. 2006;16:211-216. |
format | Online Article Text |
id | pubmed-6550857 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2019 |
publisher | Endocrine Society |
record_format | MEDLINE/PubMed |
spelling | pubmed-65508572019-06-13 MON-141 Non-Islet Cell Tumor Hypoglycemia Due to Relapsed Uterine Carcinosarcoma Caraccio, Donald J Endocr Soc Diabetes Mellitus and Glucose Metabolism Background: Non-islet cell tumor hypoglycemia is a rare cause of spontaneous hypoglycemia caused by abnormal high molecular weight IGF-2 production. Clinical Case: 78yo woman with history of uterine carcinosarcoma and DM type two was admitted to the medicine service for altered mental status, acute kidney injury, and hypoglycemia. Her lowest glucose in the emergency room was 36mg/dl (capillary) and 48mg/dl (plasma). She denied any diaphoresis, palpitations or tremulousness while hypoglycemic. She did endorse feeling “sleepy”, intermittently, which would improve after eating. She reported poor appetite, early satiety, and lower abdominal pain for the past several weeks. She denied recent weight loss. For diabetes treatment, she was on metformin 850mg daily at home. Her hemoglobin A1C was 4.8%, down from 5.6% one year prior. Random cortisol was 17.3ug/dl (1.7-14.1). Urine culture grew strep anginosus and blood cultures were negative. After initial stabilization of glucose with dextrose infusion, she was taken off dextrose containing fluids to better evaluate the hypoglycemia. Approximately 24 hours later, she was noted to have a plasma glucose of 52mg/dl. She reported feeling her typical somnolence that improved after she consumed glucose, fulfilling Whipple’s Triad. While hypoglycemic, her insulin level was <0.5mU/L (3.0-25.0), proinsulin 4.7pmol/L (3.6-22), and c-peptide 0.3ng/L (1.1-4.4); consistent with non insulin mediated hypoglycemia. Sulfonylurea screen was negative. Betahydroxybutyrate was 0.12mmol/L (0.02-0.27); which was not consistent with starvation and glycogen depletion. IGF 1 was 14ng/ml (34-182) and IGF2 was 708ng/ml (333-967). The calculated IGF2: IGF1 ratio was 50.6. In reviews of patients with non islet cell tumor hypoglycemia, a ratio above 10 is highly suggestive of this diagnosis and should lead to imaging evaluation for malignancy (1). Her CT abdomen and pelvis that showed recurrent metastatic disease consistent with her history of uterine carcinosarcoma. She was started on hydrocortisone 30mg IV every 12 hours before transitioning to prednisone 15mg daily the following day. She had no recurrences of hypoglycemia after starting on glucocorticoids. Unfortunately, her hospital course was complicated by DVT and intraperitoneal hemorrhage resulting in hypovolemic shock and renal failure. Patient died on hospital day 16 after transitioning to comfort care only. Conclusion: This case illustrates that non-islet cell tumor hypoglycemia will often present with neuroglycopenic symptoms, but without autonomic symptoms due to recurrent hypoglycemic events. Hypoglycemia can be improved with glucocorticoids, although tumor directed treatment is needed for cure. References:Fukuda I, Hizuka N, Ishikawa Y, et al. Clinical features of insulin-like growth factor-II producing non-islet-cell tumor hypoglycemia. Growth Horm IGF Res. 2006;16:211-216. Endocrine Society 2019-04-30 /pmc/articles/PMC6550857/ http://dx.doi.org/10.1210/js.2019-MON-141 Text en Copyright © 2019 Endocrine Society https://creativecommons.org/licenses/by-nc-nd/4.0/ This article has been published under the terms of the Creative Commons Attribution Non-Commercial, No-Derivatives License (CC BY-NC-ND; https://creativecommons.org/licenses/by-nc-nd/4.0/). |
spellingShingle | Diabetes Mellitus and Glucose Metabolism Caraccio, Donald MON-141 Non-Islet Cell Tumor Hypoglycemia Due to Relapsed Uterine Carcinosarcoma |
title | MON-141 Non-Islet Cell Tumor Hypoglycemia Due to Relapsed Uterine Carcinosarcoma |
title_full | MON-141 Non-Islet Cell Tumor Hypoglycemia Due to Relapsed Uterine Carcinosarcoma |
title_fullStr | MON-141 Non-Islet Cell Tumor Hypoglycemia Due to Relapsed Uterine Carcinosarcoma |
title_full_unstemmed | MON-141 Non-Islet Cell Tumor Hypoglycemia Due to Relapsed Uterine Carcinosarcoma |
title_short | MON-141 Non-Islet Cell Tumor Hypoglycemia Due to Relapsed Uterine Carcinosarcoma |
title_sort | mon-141 non-islet cell tumor hypoglycemia due to relapsed uterine carcinosarcoma |
topic | Diabetes Mellitus and Glucose Metabolism |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6550857/ http://dx.doi.org/10.1210/js.2019-MON-141 |
work_keys_str_mv | AT caracciodonald mon141nonisletcelltumorhypoglycemiaduetorelapseduterinecarcinosarcoma |