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Searching for the Culprit: When Diabetic Ketoacidosis Presents With Insulin Autoantibodies

OBJECTIVE: The main objective was to describe and review a unique case that presented with diabetic ketoacidosis, positive insulin autoantibodies (IAAbs, which are found in Hirata disease and are usually present with hypoglycemia), and laboratory findings characteristic of type B insulin resistance...

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Autores principales: Garcia-Avila, Scarlette, Samuel, Anish, Farooqi, Iqra, Manickam, Rajapriya, Ismail, Mourad, Khaddash, Saleh I., Gibiezaite, Sandra
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Association of Clinical Endocrinology 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8053616/
https://www.ncbi.nlm.nih.gov/pubmed/34095477
http://dx.doi.org/10.1016/j.aace.2020.12.004
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author Garcia-Avila, Scarlette
Samuel, Anish
Farooqi, Iqra
Manickam, Rajapriya
Ismail, Mourad
Khaddash, Saleh I.
Gibiezaite, Sandra
author_facet Garcia-Avila, Scarlette
Samuel, Anish
Farooqi, Iqra
Manickam, Rajapriya
Ismail, Mourad
Khaddash, Saleh I.
Gibiezaite, Sandra
author_sort Garcia-Avila, Scarlette
collection PubMed
description OBJECTIVE: The main objective was to describe and review a unique case that presented with diabetic ketoacidosis, positive insulin autoantibodies (IAAbs, which are found in Hirata disease and are usually present with hypoglycemia), and laboratory findings characteristic of type B insulin resistance syndrome (TBIRS) and systemic lupus erythematosus. Confirmation of TBIRS was obtained in Germany as immunoassay for insulin receptor antibodies (IRAbs) is not available in the United States. METHODS: A literature review on TBIRS and cases that present with IAAbs and IRAbs simultaneously was conducted. RESULTS: We found 6 cases presenting with hypoglycemia, both antibodies, and treatment attempts with various management approaches that were different from the proposed National Institutes of Health (NIH) protocol for TBIRS. Our case is distinct because of the demographic background, presentation with diabetic ketoacidosis, comparatively lower insulin requirement, and no significant hypoglycemic episodes in the third phase. CONCLUSION: We propose that access to IRAb immunoassays may be important for diagnosing milder cases of TBIRS, while IAAbs may provide prognostic and therapeutic insights. Despite completely different presentation from other TBIRS patients reviewed, we observed that the proposed NIH protocol consisting of dexamethasone, rituximab, and cyclophosphamide was successfully employed in our patient. Thus, we propose that our case and the findings regarding antibody testing and the NIH treatment regimen may assist clinicians with earlier recognition and effective management of milder cases of TBIRS.
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spelling pubmed-80536162021-06-03 Searching for the Culprit: When Diabetic Ketoacidosis Presents With Insulin Autoantibodies Garcia-Avila, Scarlette Samuel, Anish Farooqi, Iqra Manickam, Rajapriya Ismail, Mourad Khaddash, Saleh I. Gibiezaite, Sandra AACE Clin Case Rep Case Report OBJECTIVE: The main objective was to describe and review a unique case that presented with diabetic ketoacidosis, positive insulin autoantibodies (IAAbs, which are found in Hirata disease and are usually present with hypoglycemia), and laboratory findings characteristic of type B insulin resistance syndrome (TBIRS) and systemic lupus erythematosus. Confirmation of TBIRS was obtained in Germany as immunoassay for insulin receptor antibodies (IRAbs) is not available in the United States. METHODS: A literature review on TBIRS and cases that present with IAAbs and IRAbs simultaneously was conducted. RESULTS: We found 6 cases presenting with hypoglycemia, both antibodies, and treatment attempts with various management approaches that were different from the proposed National Institutes of Health (NIH) protocol for TBIRS. Our case is distinct because of the demographic background, presentation with diabetic ketoacidosis, comparatively lower insulin requirement, and no significant hypoglycemic episodes in the third phase. CONCLUSION: We propose that access to IRAb immunoassays may be important for diagnosing milder cases of TBIRS, while IAAbs may provide prognostic and therapeutic insights. Despite completely different presentation from other TBIRS patients reviewed, we observed that the proposed NIH protocol consisting of dexamethasone, rituximab, and cyclophosphamide was successfully employed in our patient. Thus, we propose that our case and the findings regarding antibody testing and the NIH treatment regimen may assist clinicians with earlier recognition and effective management of milder cases of TBIRS. American Association of Clinical Endocrinology 2020-12-28 /pmc/articles/PMC8053616/ /pubmed/34095477 http://dx.doi.org/10.1016/j.aace.2020.12.004 Text en © 2020 AACE. Published by Elsevier Inc. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Garcia-Avila, Scarlette
Samuel, Anish
Farooqi, Iqra
Manickam, Rajapriya
Ismail, Mourad
Khaddash, Saleh I.
Gibiezaite, Sandra
Searching for the Culprit: When Diabetic Ketoacidosis Presents With Insulin Autoantibodies
title Searching for the Culprit: When Diabetic Ketoacidosis Presents With Insulin Autoantibodies
title_full Searching for the Culprit: When Diabetic Ketoacidosis Presents With Insulin Autoantibodies
title_fullStr Searching for the Culprit: When Diabetic Ketoacidosis Presents With Insulin Autoantibodies
title_full_unstemmed Searching for the Culprit: When Diabetic Ketoacidosis Presents With Insulin Autoantibodies
title_short Searching for the Culprit: When Diabetic Ketoacidosis Presents With Insulin Autoantibodies
title_sort searching for the culprit: when diabetic ketoacidosis presents with insulin autoantibodies
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8053616/
https://www.ncbi.nlm.nih.gov/pubmed/34095477
http://dx.doi.org/10.1016/j.aace.2020.12.004
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