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Pembrolizumab on pre-existing inclusion body myositis: a case report

BACKGROUND: Cases of exacerbation of pre-existing neuromuscular diseases induced by immune checkpoint inhibitors (ICIs) have rarely been reported because patients with autoimmune diseases have generally been excluded from ICI therapy due to the increased risk of exacerbation. We describe the first c...

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Autores principales: Uchio, Naohiro, Unuma, Atsushi, Kakumoto, Toshiyuki, Osaki, Masao, Zenke, Yoshitaka, Sakuta, Kenichi, Kubota, Akatsuki, Uesaka, Yoshikazu, Toda, Tatsushi, Shimizu, Jun
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2020
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Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7493364/
https://www.ncbi.nlm.nih.gov/pubmed/32944686
http://dx.doi.org/10.1186/s41927-020-00144-5
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author Uchio, Naohiro
Unuma, Atsushi
Kakumoto, Toshiyuki
Osaki, Masao
Zenke, Yoshitaka
Sakuta, Kenichi
Kubota, Akatsuki
Uesaka, Yoshikazu
Toda, Tatsushi
Shimizu, Jun
author_facet Uchio, Naohiro
Unuma, Atsushi
Kakumoto, Toshiyuki
Osaki, Masao
Zenke, Yoshitaka
Sakuta, Kenichi
Kubota, Akatsuki
Uesaka, Yoshikazu
Toda, Tatsushi
Shimizu, Jun
author_sort Uchio, Naohiro
collection PubMed
description BACKGROUND: Cases of exacerbation of pre-existing neuromuscular diseases induced by immune checkpoint inhibitors (ICIs) have rarely been reported because patients with autoimmune diseases have generally been excluded from ICI therapy due to the increased risk of exacerbation. We describe the first case of an elderly patient who experienced exacerbation of a previously undiagnosed sporadic inclusion body myositis (sIBM), the most common myopathy in the geriatric population, which was triggered by anti-programmed cell death-1 therapy. CASE PRESENTATION: A 75-year-old man who was receiving pembrolizumab presented with limb weakness. Three years prior, he had noticed slowly progressive limb weakness, but he received no diagnosis. After the first infusion of pembrolizumab, his creatine kinase (CK) levels had increased. The neurological examination and muscle biopsy findings confirmed the diagnosis of sIBM and suggested exacerbation of sIBM induced by pembrolizumab. After the patient’s CK levels decreased, pembrolizumab was restarted. The tumor progressed after its treatment with pembrolizumab. The patient died after 15 months of follow-up. CONCLUSIONS: In patients with slowly progressive limb weakness, sIBM should be explored before ICI therapy. In addition, if patients show high CK levels after ICI introduction, it is necessary to confirm whether they have sIBM in order to avoid unnecessary immunosuppressive therapies and assess whether they can tolerate ICI reintroduction.
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spelling pubmed-74933642020-09-16 Pembrolizumab on pre-existing inclusion body myositis: a case report Uchio, Naohiro Unuma, Atsushi Kakumoto, Toshiyuki Osaki, Masao Zenke, Yoshitaka Sakuta, Kenichi Kubota, Akatsuki Uesaka, Yoshikazu Toda, Tatsushi Shimizu, Jun BMC Rheumatol Case Report BACKGROUND: Cases of exacerbation of pre-existing neuromuscular diseases induced by immune checkpoint inhibitors (ICIs) have rarely been reported because patients with autoimmune diseases have generally been excluded from ICI therapy due to the increased risk of exacerbation. We describe the first case of an elderly patient who experienced exacerbation of a previously undiagnosed sporadic inclusion body myositis (sIBM), the most common myopathy in the geriatric population, which was triggered by anti-programmed cell death-1 therapy. CASE PRESENTATION: A 75-year-old man who was receiving pembrolizumab presented with limb weakness. Three years prior, he had noticed slowly progressive limb weakness, but he received no diagnosis. After the first infusion of pembrolizumab, his creatine kinase (CK) levels had increased. The neurological examination and muscle biopsy findings confirmed the diagnosis of sIBM and suggested exacerbation of sIBM induced by pembrolizumab. After the patient’s CK levels decreased, pembrolizumab was restarted. The tumor progressed after its treatment with pembrolizumab. The patient died after 15 months of follow-up. CONCLUSIONS: In patients with slowly progressive limb weakness, sIBM should be explored before ICI therapy. In addition, if patients show high CK levels after ICI introduction, it is necessary to confirm whether they have sIBM in order to avoid unnecessary immunosuppressive therapies and assess whether they can tolerate ICI reintroduction. BioMed Central 2020-09-16 /pmc/articles/PMC7493364/ /pubmed/32944686 http://dx.doi.org/10.1186/s41927-020-00144-5 Text en © The Author(s) 2020 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/. The Creative Commons Public Domain Dedication waiver (http://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
Uchio, Naohiro
Unuma, Atsushi
Kakumoto, Toshiyuki
Osaki, Masao
Zenke, Yoshitaka
Sakuta, Kenichi
Kubota, Akatsuki
Uesaka, Yoshikazu
Toda, Tatsushi
Shimizu, Jun
Pembrolizumab on pre-existing inclusion body myositis: a case report
title Pembrolizumab on pre-existing inclusion body myositis: a case report
title_full Pembrolizumab on pre-existing inclusion body myositis: a case report
title_fullStr Pembrolizumab on pre-existing inclusion body myositis: a case report
title_full_unstemmed Pembrolizumab on pre-existing inclusion body myositis: a case report
title_short Pembrolizumab on pre-existing inclusion body myositis: a case report
title_sort pembrolizumab on pre-existing inclusion body myositis: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7493364/
https://www.ncbi.nlm.nih.gov/pubmed/32944686
http://dx.doi.org/10.1186/s41927-020-00144-5
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