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Case Report: The Neuromusclar Triad of Immune Checkpoint Inhibitors: A Case Report of Myositis, Myocarditis, and Myasthenia Gravis Overlap Following Toripalimab Treatment

The neuromuscular adverse events of immune checkpoint inhibitor (ICI) treatment include myositis, polymyalgia rheumatica, myocarditis, and myasthenia syndrome. We report a 47-year old female presenting with external ophthalmoplegia, generalized muscle weakness, and third-degree atrioventricular bloc...

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Autores principales: Luo, Yue-Bei, Tang, Weiting, Zeng, Qiuming, Duan, Weiwei, Li, Shuyu, Yang, Xiaosu, Bi, Fangfang
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8415306/
https://www.ncbi.nlm.nih.gov/pubmed/34485412
http://dx.doi.org/10.3389/fcvm.2021.714460
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author Luo, Yue-Bei
Tang, Weiting
Zeng, Qiuming
Duan, Weiwei
Li, Shuyu
Yang, Xiaosu
Bi, Fangfang
author_facet Luo, Yue-Bei
Tang, Weiting
Zeng, Qiuming
Duan, Weiwei
Li, Shuyu
Yang, Xiaosu
Bi, Fangfang
author_sort Luo, Yue-Bei
collection PubMed
description The neuromuscular adverse events of immune checkpoint inhibitor (ICI) treatment include myositis, polymyalgia rheumatica, myocarditis, and myasthenia syndrome. We report a 47-year old female presenting with external ophthalmoplegia, generalized muscle weakness, and third-degree atrioventricular block 4 weeks after toripalimab treatment for metastatic thymoma. Creatine kinase was elevated to 25,200 U/l and cardiac troponin I to 2.796 ng/ml. Autoantibody profiling shows positive anti-ryanodine receptor and anti-acetylcholine receptor antibodies and negative myositis specific antibodies. Repetitive nerve stimulation did not reveal decrement of compound muscle action potentials. Pulse methylprednisolone and immunoglobulin infusion, together with temporary pacemaker insertion normalized her muscle enzyme levels and cardiac rhythm. This is the first report of overlaping neuromuscular adverse event of toripalimab.
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spelling pubmed-84153062021-09-04 Case Report: The Neuromusclar Triad of Immune Checkpoint Inhibitors: A Case Report of Myositis, Myocarditis, and Myasthenia Gravis Overlap Following Toripalimab Treatment Luo, Yue-Bei Tang, Weiting Zeng, Qiuming Duan, Weiwei Li, Shuyu Yang, Xiaosu Bi, Fangfang Front Cardiovasc Med Cardiovascular Medicine The neuromuscular adverse events of immune checkpoint inhibitor (ICI) treatment include myositis, polymyalgia rheumatica, myocarditis, and myasthenia syndrome. We report a 47-year old female presenting with external ophthalmoplegia, generalized muscle weakness, and third-degree atrioventricular block 4 weeks after toripalimab treatment for metastatic thymoma. Creatine kinase was elevated to 25,200 U/l and cardiac troponin I to 2.796 ng/ml. Autoantibody profiling shows positive anti-ryanodine receptor and anti-acetylcholine receptor antibodies and negative myositis specific antibodies. Repetitive nerve stimulation did not reveal decrement of compound muscle action potentials. Pulse methylprednisolone and immunoglobulin infusion, together with temporary pacemaker insertion normalized her muscle enzyme levels and cardiac rhythm. This is the first report of overlaping neuromuscular adverse event of toripalimab. Frontiers Media S.A. 2021-08-16 /pmc/articles/PMC8415306/ /pubmed/34485412 http://dx.doi.org/10.3389/fcvm.2021.714460 Text en Copyright © 2021 Luo, Tang, Zeng, Duan, Li, Yang and Bi. 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 Cardiovascular Medicine
Luo, Yue-Bei
Tang, Weiting
Zeng, Qiuming
Duan, Weiwei
Li, Shuyu
Yang, Xiaosu
Bi, Fangfang
Case Report: The Neuromusclar Triad of Immune Checkpoint Inhibitors: A Case Report of Myositis, Myocarditis, and Myasthenia Gravis Overlap Following Toripalimab Treatment
title Case Report: The Neuromusclar Triad of Immune Checkpoint Inhibitors: A Case Report of Myositis, Myocarditis, and Myasthenia Gravis Overlap Following Toripalimab Treatment
title_full Case Report: The Neuromusclar Triad of Immune Checkpoint Inhibitors: A Case Report of Myositis, Myocarditis, and Myasthenia Gravis Overlap Following Toripalimab Treatment
title_fullStr Case Report: The Neuromusclar Triad of Immune Checkpoint Inhibitors: A Case Report of Myositis, Myocarditis, and Myasthenia Gravis Overlap Following Toripalimab Treatment
title_full_unstemmed Case Report: The Neuromusclar Triad of Immune Checkpoint Inhibitors: A Case Report of Myositis, Myocarditis, and Myasthenia Gravis Overlap Following Toripalimab Treatment
title_short Case Report: The Neuromusclar Triad of Immune Checkpoint Inhibitors: A Case Report of Myositis, Myocarditis, and Myasthenia Gravis Overlap Following Toripalimab Treatment
title_sort case report: the neuromusclar triad of immune checkpoint inhibitors: a case report of myositis, myocarditis, and myasthenia gravis overlap following toripalimab treatment
topic Cardiovascular Medicine
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8415306/
https://www.ncbi.nlm.nih.gov/pubmed/34485412
http://dx.doi.org/10.3389/fcvm.2021.714460
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