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Mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report

BACKGROUND: Mononeuritis multiplex is a rare autoimmune peripheral neuropathy that typically presents in the context of vasculitis, diabetes, infection, or as a paraneoplastic syndrome. Adverse immune-related neurological conditions have been increasingly reported with the use of immune checkpoint i...

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Autores principales: Abdelhakim, Safa, Klapholz, Jonah D., Roy, Bhaskar, Weiss, Sarah A., McGuone, Declan, Corbin, Zachary A.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8867751/
https://www.ncbi.nlm.nih.gov/pubmed/35197122
http://dx.doi.org/10.1186/s13256-022-03290-1
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author Abdelhakim, Safa
Klapholz, Jonah D.
Roy, Bhaskar
Weiss, Sarah A.
McGuone, Declan
Corbin, Zachary A.
author_facet Abdelhakim, Safa
Klapholz, Jonah D.
Roy, Bhaskar
Weiss, Sarah A.
McGuone, Declan
Corbin, Zachary A.
author_sort Abdelhakim, Safa
collection PubMed
description BACKGROUND: Mononeuritis multiplex is a rare autoimmune peripheral neuropathy that typically presents in the context of vasculitis, diabetes, infection, or as a paraneoplastic syndrome. Adverse immune-related neurological conditions have been increasingly reported with the use of immune checkpoint inhibitors against cytotoxic T-lymphocyte antigen-4 and/or the programmed cell death protein 1/programmed death ligand-1 axis. Mononeuritis multiplex has only been reported twice from treatment of cancers with immunotherapy. CASE PRESENTATION: Here we report a case of mononeuritis multiplex as a complication of immune checkpoint inhibitor therapy for melanoma. An 80-year-old non-Hispanic white female with recurrent melanoma was treated with combination ipilimumab and nivolumab and subsequently presented with progressive leg weakness, back pain, and difficulty ambulating. The diagnosis of mononeuritis multiplex was made, which was resistant to steroid pulses, chronic steroids, intravenous immunoglobulin, and rituximab. She developed progressive neurologic dysfunction and elected for hospice care. We found only two other cases reported in the literature. CONCLUSIONS: Increased awareness, prompt recognition, and aggressive treatments are likely the best opportunity for improved outcomes in this severe side effect.
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spelling pubmed-88677512022-02-25 Mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report Abdelhakim, Safa Klapholz, Jonah D. Roy, Bhaskar Weiss, Sarah A. McGuone, Declan Corbin, Zachary A. J Med Case Rep Case Report BACKGROUND: Mononeuritis multiplex is a rare autoimmune peripheral neuropathy that typically presents in the context of vasculitis, diabetes, infection, or as a paraneoplastic syndrome. Adverse immune-related neurological conditions have been increasingly reported with the use of immune checkpoint inhibitors against cytotoxic T-lymphocyte antigen-4 and/or the programmed cell death protein 1/programmed death ligand-1 axis. Mononeuritis multiplex has only been reported twice from treatment of cancers with immunotherapy. CASE PRESENTATION: Here we report a case of mononeuritis multiplex as a complication of immune checkpoint inhibitor therapy for melanoma. An 80-year-old non-Hispanic white female with recurrent melanoma was treated with combination ipilimumab and nivolumab and subsequently presented with progressive leg weakness, back pain, and difficulty ambulating. The diagnosis of mononeuritis multiplex was made, which was resistant to steroid pulses, chronic steroids, intravenous immunoglobulin, and rituximab. She developed progressive neurologic dysfunction and elected for hospice care. We found only two other cases reported in the literature. CONCLUSIONS: Increased awareness, prompt recognition, and aggressive treatments are likely the best opportunity for improved outcomes in this severe side effect. BioMed Central 2022-02-24 /pmc/articles/PMC8867751/ /pubmed/35197122 http://dx.doi.org/10.1186/s13256-022-03290-1 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Abdelhakim, Safa
Klapholz, Jonah D.
Roy, Bhaskar
Weiss, Sarah A.
McGuone, Declan
Corbin, Zachary A.
Mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report
title Mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report
title_full Mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report
title_fullStr Mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report
title_full_unstemmed Mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report
title_short Mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report
title_sort mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8867751/
https://www.ncbi.nlm.nih.gov/pubmed/35197122
http://dx.doi.org/10.1186/s13256-022-03290-1
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