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ODP213 Insulin Autoimmune Syndrome a rare case report

BACKGROUND: Insulin autoimmune syndrome IAS is cause of endogenous hyperinsulinemic hypoglycemia. Insulin autoimmune syndrome has been described with various drugs. Here we describe a case of IAS with use of Proton pump inhibitors, PPIs. CASE REPORT: 49year old man came with complaints of recurrent...

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Autor principal: Paladugu, Sridevi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9629212/
http://dx.doi.org/10.1210/jendso/bvac150.665
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author Paladugu, Sridevi
author_facet Paladugu, Sridevi
author_sort Paladugu, Sridevi
collection PubMed
description BACKGROUND: Insulin autoimmune syndrome IAS is cause of endogenous hyperinsulinemic hypoglycemia. Insulin autoimmune syndrome has been described with various drugs. Here we describe a case of IAS with use of Proton pump inhibitors, PPIs. CASE REPORT: 49year old man came with complaints of recurrent documented hypoglycemia for the past 3months. He was a diagnosed as having Gastroesophageal reflux GERD 6 months back and has been on treatment with Pantoprazole with which his symptoms have subsided. Since past 3 months he started having episodes of excess sweating, tremulousness and palpitations. He was diagnosed as having hypoglycemia during one such episode using a glucometer value <60 mg/dl. After this he started monitoring his blood glucose values on glucometer and had recorded values as low as 50mg/dl, 55mg/dl, 47mg/dl. There was no specific association with meal intake. Every time his symptoms subsided with intake of diet. He was not a diabetic nor did he have any access to anti diabetic agents. With these complaints he was admitted and put on prolonged fasting, after 6 hours of fasting his blood glucose levels dropped to 42mg/dl. His critical sample revealed RBS- 42mg/dl, C peptide -15.7ng/ml (reference range 0.9-7.1ng/ml), serum insulin >300 µIU/ml (1.9-23µIU/ml fasting state) and serum cortisol 20.65 ug/dl(5-25ug/dl). His Sulphonyl urea screen was negative. Biochemically Endogenous hyperinsulinemia was thus proved but on imaging ultrasound examination was normal, CT scan abdomen did not reveal any pancreatic lesions. His endoscopic ultrasound also did not reveal any lesion in the pancreas. Insulin antibodies test showed positive antibodies >300 U/ml (reference range - <12U/ml). He is a known case of hypothyroidism on regular treatment. His thyroid profile was normal. There was no history of usage of methimazole, carbimazole, ALA, captopril, diltiazem, procainamide, Isoniazid, Imipenem, penicillin, Interferon, clopidogrel, gliclazide etc. He was treated with Diazoxide, Prednisolone and Acarbose. His blood glucose levels stabilized and he did not have a recurrence of hypoglycemia. Gradually he was tapered off diazoxide and prednisolone over a period of 6 months. His repeat insulin antibodies after 6 months showed 43.23U/ml (reference range<12). He is asymptomatic and is on follow up. IAS with PPI's is described 1 . Detailed history and physical examination did not reveal any other cause for IAS. CONCLUSION: PPIs are commonly prescribed agents used ubiquitously. Here we wanted to highlight the importance of IAS with PPIs. There is no specific regimen used to treat IAS. Our patient showed good response to a combination of prednisolone, diazoxide and acarbose. This triple combination addresses the multiple mechanisms of hypoglycemia in IAS. References: Simone Censi et al Insulin autoimmune syndrome: diagnosis to clinical management. Annals of Translational medicine 2018 sep;6(17): 335. Presentation: No date and time listed
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spelling pubmed-96292122022-11-04 ODP213 Insulin Autoimmune Syndrome a rare case report Paladugu, Sridevi J Endocr Soc Diabetes & Glucose Metabolism BACKGROUND: Insulin autoimmune syndrome IAS is cause of endogenous hyperinsulinemic hypoglycemia. Insulin autoimmune syndrome has been described with various drugs. Here we describe a case of IAS with use of Proton pump inhibitors, PPIs. CASE REPORT: 49year old man came with complaints of recurrent documented hypoglycemia for the past 3months. He was a diagnosed as having Gastroesophageal reflux GERD 6 months back and has been on treatment with Pantoprazole with which his symptoms have subsided. Since past 3 months he started having episodes of excess sweating, tremulousness and palpitations. He was diagnosed as having hypoglycemia during one such episode using a glucometer value <60 mg/dl. After this he started monitoring his blood glucose values on glucometer and had recorded values as low as 50mg/dl, 55mg/dl, 47mg/dl. There was no specific association with meal intake. Every time his symptoms subsided with intake of diet. He was not a diabetic nor did he have any access to anti diabetic agents. With these complaints he was admitted and put on prolonged fasting, after 6 hours of fasting his blood glucose levels dropped to 42mg/dl. His critical sample revealed RBS- 42mg/dl, C peptide -15.7ng/ml (reference range 0.9-7.1ng/ml), serum insulin >300 µIU/ml (1.9-23µIU/ml fasting state) and serum cortisol 20.65 ug/dl(5-25ug/dl). His Sulphonyl urea screen was negative. Biochemically Endogenous hyperinsulinemia was thus proved but on imaging ultrasound examination was normal, CT scan abdomen did not reveal any pancreatic lesions. His endoscopic ultrasound also did not reveal any lesion in the pancreas. Insulin antibodies test showed positive antibodies >300 U/ml (reference range - <12U/ml). He is a known case of hypothyroidism on regular treatment. His thyroid profile was normal. There was no history of usage of methimazole, carbimazole, ALA, captopril, diltiazem, procainamide, Isoniazid, Imipenem, penicillin, Interferon, clopidogrel, gliclazide etc. He was treated with Diazoxide, Prednisolone and Acarbose. His blood glucose levels stabilized and he did not have a recurrence of hypoglycemia. Gradually he was tapered off diazoxide and prednisolone over a period of 6 months. His repeat insulin antibodies after 6 months showed 43.23U/ml (reference range<12). He is asymptomatic and is on follow up. IAS with PPI's is described 1 . Detailed history and physical examination did not reveal any other cause for IAS. CONCLUSION: PPIs are commonly prescribed agents used ubiquitously. Here we wanted to highlight the importance of IAS with PPIs. There is no specific regimen used to treat IAS. Our patient showed good response to a combination of prednisolone, diazoxide and acarbose. This triple combination addresses the multiple mechanisms of hypoglycemia in IAS. References: Simone Censi et al Insulin autoimmune syndrome: diagnosis to clinical management. Annals of Translational medicine 2018 sep;6(17): 335. Presentation: No date and time listed Oxford University Press 2022-11-01 /pmc/articles/PMC9629212/ http://dx.doi.org/10.1210/jendso/bvac150.665 Text en © The Author(s) 2022. Published by Oxford University Press on behalf of the Endocrine Society. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence (https://creativecommons.org/licenses/by-nc-nd/4.0/), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Diabetes & Glucose Metabolism
Paladugu, Sridevi
ODP213 Insulin Autoimmune Syndrome a rare case report
title ODP213 Insulin Autoimmune Syndrome a rare case report
title_full ODP213 Insulin Autoimmune Syndrome a rare case report
title_fullStr ODP213 Insulin Autoimmune Syndrome a rare case report
title_full_unstemmed ODP213 Insulin Autoimmune Syndrome a rare case report
title_short ODP213 Insulin Autoimmune Syndrome a rare case report
title_sort odp213 insulin autoimmune syndrome a rare case report
topic Diabetes & Glucose Metabolism
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9629212/
http://dx.doi.org/10.1210/jendso/bvac150.665
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