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Case Report: Fulminant Celiac Disease With Combination Immune Checkpoint Therapy

Since the first approval of immune checkpoint inhibitors (ICIs) in 2011, these agents have rapidly become an integral treatment option across tumor types. However, with the increased adoption of ICIs, the incidence of immune-related adverse events (irAEs) continues to rise, and rare toxicity continu...

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Autores principales: Falade, Ayo S., Reynolds, Kerry L., Zubiri, Leyre, Deshpande, Vikram, Fintelmann, Florian J., Dougan, Michael, Mooradian, Meghan J.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9049212/
https://www.ncbi.nlm.nih.gov/pubmed/35493494
http://dx.doi.org/10.3389/fimmu.2022.871452
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author Falade, Ayo S.
Reynolds, Kerry L.
Zubiri, Leyre
Deshpande, Vikram
Fintelmann, Florian J.
Dougan, Michael
Mooradian, Meghan J.
author_facet Falade, Ayo S.
Reynolds, Kerry L.
Zubiri, Leyre
Deshpande, Vikram
Fintelmann, Florian J.
Dougan, Michael
Mooradian, Meghan J.
author_sort Falade, Ayo S.
collection PubMed
description Since the first approval of immune checkpoint inhibitors (ICIs) in 2011, these agents have rapidly become an integral treatment option across tumor types. However, with the increased adoption of ICIs, the incidence of immune-related adverse events (irAEs) continues to rise, and rare toxicity continues to be reported. Here, we present a case of a 70-year-old male patient with widespread metastatic melanoma who developed rapid onset anasarca and transaminitis after initiation of dual anti-PD-1/CTLA-4 inhibition with nivolumab and ipilimumab. An extensive workup was performed with serologies returning positive for anti-tissue transglutaminase immunoglobulin (tTG-IgA) and endoscopy revealing duodenal mucosal atrophy with duodenal biopsies confirming celiac disease. All symptoms resolved after initiation of a gluten-free diet without the addition of immunosuppression. This case highlights the importance of considering celiac disease in patients with suspected protein-losing enteropathy on ICI, the fulminant nature this uncommon irAE can present with, and underscores the broad differential clinicians must maintain when managing presumed irAEs.
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spelling pubmed-90492122022-04-29 Case Report: Fulminant Celiac Disease With Combination Immune Checkpoint Therapy Falade, Ayo S. Reynolds, Kerry L. Zubiri, Leyre Deshpande, Vikram Fintelmann, Florian J. Dougan, Michael Mooradian, Meghan J. Front Immunol Immunology Since the first approval of immune checkpoint inhibitors (ICIs) in 2011, these agents have rapidly become an integral treatment option across tumor types. However, with the increased adoption of ICIs, the incidence of immune-related adverse events (irAEs) continues to rise, and rare toxicity continues to be reported. Here, we present a case of a 70-year-old male patient with widespread metastatic melanoma who developed rapid onset anasarca and transaminitis after initiation of dual anti-PD-1/CTLA-4 inhibition with nivolumab and ipilimumab. An extensive workup was performed with serologies returning positive for anti-tissue transglutaminase immunoglobulin (tTG-IgA) and endoscopy revealing duodenal mucosal atrophy with duodenal biopsies confirming celiac disease. All symptoms resolved after initiation of a gluten-free diet without the addition of immunosuppression. This case highlights the importance of considering celiac disease in patients with suspected protein-losing enteropathy on ICI, the fulminant nature this uncommon irAE can present with, and underscores the broad differential clinicians must maintain when managing presumed irAEs. Frontiers Media S.A. 2022-04-14 /pmc/articles/PMC9049212/ /pubmed/35493494 http://dx.doi.org/10.3389/fimmu.2022.871452 Text en Copyright © 2022 Falade, Reynolds, Zubiri, Deshpande, Fintelmann, Dougan and Mooradian https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Immunology
Falade, Ayo S.
Reynolds, Kerry L.
Zubiri, Leyre
Deshpande, Vikram
Fintelmann, Florian J.
Dougan, Michael
Mooradian, Meghan J.
Case Report: Fulminant Celiac Disease With Combination Immune Checkpoint Therapy
title Case Report: Fulminant Celiac Disease With Combination Immune Checkpoint Therapy
title_full Case Report: Fulminant Celiac Disease With Combination Immune Checkpoint Therapy
title_fullStr Case Report: Fulminant Celiac Disease With Combination Immune Checkpoint Therapy
title_full_unstemmed Case Report: Fulminant Celiac Disease With Combination Immune Checkpoint Therapy
title_short Case Report: Fulminant Celiac Disease With Combination Immune Checkpoint Therapy
title_sort case report: fulminant celiac disease with combination immune checkpoint therapy
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9049212/
https://www.ncbi.nlm.nih.gov/pubmed/35493494
http://dx.doi.org/10.3389/fimmu.2022.871452
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