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Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review

Cutaneous vesiculobullous eruptions associated with multiple myeloma (MM) are rare. Although the development of blisters is mostly driven by amyloid deposits of paraproteins in the skin, autoimmunity may play a role. In this study, we report an unusual case of an MM patient with blisters who present...

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Autores principales: Li, Tong, Liu, Hongjie, Li, Wei
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9950124/
https://www.ncbi.nlm.nih.gov/pubmed/36845094
http://dx.doi.org/10.3389/fimmu.2023.1121380
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author Li, Tong
Liu, Hongjie
Li, Wei
author_facet Li, Tong
Liu, Hongjie
Li, Wei
author_sort Li, Tong
collection PubMed
description Cutaneous vesiculobullous eruptions associated with multiple myeloma (MM) are rare. Although the development of blisters is mostly driven by amyloid deposits of paraproteins in the skin, autoimmunity may play a role. In this study, we report an unusual case of an MM patient with blisters who presented with both flaccid and tense vesicles and bullae. Direct immunofluorescence revealed IgA autoantibody deposits in the basement membrane zone (BMZ) and intercellular space of the epidermis, which displayed an atypical autoantibody deposition pattern. The patient showed rapid disease progression and died during follow-up. We performed a literature review of autoimmune bullous diseases (AIBDs) associated with MM or its precursors and found 17 previously reported cases. Together with the present case, cutaneous involvement of the skin folds was frequently reported, and mucous membranes were barely affected. IgA pemphigus, with consistent IgA monoclonality, was observed in half of the cases. Five patients displayed atypical autoantibody deposition patterns in the skin; the prognosis of these patients appeared to be poorer than that of other patients. We aim to increase our understanding of AIBDs associated with MM or its precursors.
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spelling pubmed-99501242023-02-25 Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review Li, Tong Liu, Hongjie Li, Wei Front Immunol Immunology Cutaneous vesiculobullous eruptions associated with multiple myeloma (MM) are rare. Although the development of blisters is mostly driven by amyloid deposits of paraproteins in the skin, autoimmunity may play a role. In this study, we report an unusual case of an MM patient with blisters who presented with both flaccid and tense vesicles and bullae. Direct immunofluorescence revealed IgA autoantibody deposits in the basement membrane zone (BMZ) and intercellular space of the epidermis, which displayed an atypical autoantibody deposition pattern. The patient showed rapid disease progression and died during follow-up. We performed a literature review of autoimmune bullous diseases (AIBDs) associated with MM or its precursors and found 17 previously reported cases. Together with the present case, cutaneous involvement of the skin folds was frequently reported, and mucous membranes were barely affected. IgA pemphigus, with consistent IgA monoclonality, was observed in half of the cases. Five patients displayed atypical autoantibody deposition patterns in the skin; the prognosis of these patients appeared to be poorer than that of other patients. We aim to increase our understanding of AIBDs associated with MM or its precursors. Frontiers Media S.A. 2023-02-10 /pmc/articles/PMC9950124/ /pubmed/36845094 http://dx.doi.org/10.3389/fimmu.2023.1121380 Text en Copyright © 2023 Li, Liu and Li 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
Li, Tong
Liu, Hongjie
Li, Wei
Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review
title Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review
title_full Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review
title_fullStr Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review
title_full_unstemmed Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review
title_short Case report: Vesiculobullous eruption with an atypical IgA deposition pattern in a patient with multiple myeloma—A case report and literature review
title_sort case report: vesiculobullous eruption with an atypical iga deposition pattern in a patient with multiple myeloma—a case report and literature review
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9950124/
https://www.ncbi.nlm.nih.gov/pubmed/36845094
http://dx.doi.org/10.3389/fimmu.2023.1121380
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