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度伐利尤单抗诱导的三联征综合征。

Durvalumab-Induced Triple-M Syndrome.

作者信息

Adeoye Femi Williams, Jaffar Nida, Surandran Sanggeeta, Begum Gulshad, Islam Mohammad Rafiqul

机构信息

Southend University Hospital, Southend-on-Sea, United Kingdom.

Oncology Department, Southend University Hospital, Southend-on-Sea, United Kingdom.

出版信息

Eur J Case Rep Intern Med. 2024 Jul 30;11(8):004729. doi: 10.12890/2024_004729. eCollection 2024.

DOI:10.12890/2024_004729
PMID:39130070
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11313117/
Abstract

BACKGROUND

While the use of immunotherapy has revolutionised the treatment of various cancers, it is often associated with a myriad of immune-related adverse effects.

CASE PRESENTATION

In this article, we report a rare case of durvalumab-induced triple-M syndrome in a 69-year-old woman with stage III lung adenocarcinoma. She was admitted with profound generalised muscle weakness, myalgia, and exertional breathlessness, about a week into her second cycle of durvalumab, an immune checkpoint inhibitor. She had clinicopathological features of myositis, myasthenia and myocarditis with acute onset symptomatic tri-fascicular block on electrocardiogram, requiring urgent cardiology intervention. Durvalumab was discontinued and she was treated with a combination of high-dose steroids and intravenous immunoglobulin after which she had clinical and biochemical improvement, albeit with residual muscle weakness.

CONCLUSION

Myocarditis-myositis-myasthenia complex is a rare side effect of immunotherapy which has been reported in other immune checkpoint inhibitors, but less so with durvalumab. We report this clinical case to raise awareness of this rare and potentially life-threatening adverse effect of this agent.

LEARNING POINTS

Triple-M syndrome is a rare immune-related adverse effect, which has been noted in other immune checkpoint inhibitors, but less so with durvalumab specifically.Immunotherapy-induced myositis, myocarditis and myasthenia can occur in isolation or, rarely, in association as a syndrome.This case demonstrates the potentially life-threatening nature of this entity, the need for early recognition, and multi-specialist teamwork to ensure good outcome.

摘要

背景

虽然免疫疗法的应用彻底改变了各种癌症的治疗方式,但它常常与众多免疫相关的不良反应有关。

病例报告

在本文中,我们报告了一例罕见的由度伐利尤单抗引起的三 M 综合征,患者为一名 69 岁的 III 期肺腺癌女性。在接受免疫检查点抑制剂度伐利尤单抗的第二个周期治疗约一周后,她因严重的全身肌肉无力、肌痛和劳力性呼吸困难入院。她具有肌炎、重症肌无力和心肌炎的临床病理特征,心电图显示急性发作的症状性三分支传导阻滞,需要心脏科紧急干预。停用度伐利尤单抗后,她接受了大剂量类固醇和静脉注射免疫球蛋白的联合治疗,此后临床和生化指标有所改善,尽管仍有残留的肌肉无力。

结论

心肌炎 - 肌炎 - 重症肌无力综合征是免疫疗法的一种罕见副作用,在其他免疫检查点抑制剂中已有报道,但度伐利尤单抗引起的较少。我们报告这个临床病例是为了提高对这种药物罕见且可能危及生命的不良反应的认识。

经验教训

三 M 综合征是一种罕见的免疫相关不良反应,在其他免疫检查点抑制剂中已有发现,但度伐利尤单抗引起的情况较少。免疫疗法引起的肌炎、心肌炎和重症肌无力可单独发生,或很少作为一种综合征联合出现。本病例证明了该病症的潜在致命性、早期识别的必要性以及多专科团队合作以确保良好预后的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/47ce/11313117/f3e3bfff913e/4729_Fig4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/47ce/11313117/22316e9e75c6/4729_Fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/47ce/11313117/813c8dc0874c/4729_Fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/47ce/11313117/318cb2e7490f/4729_Fig3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/47ce/11313117/f3e3bfff913e/4729_Fig4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/47ce/11313117/22316e9e75c6/4729_Fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/47ce/11313117/813c8dc0874c/4729_Fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/47ce/11313117/318cb2e7490f/4729_Fig3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/47ce/11313117/f3e3bfff913e/4729_Fig4.jpg

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本文引用的文献

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Drug-Induced Myopathies: A Comprehensive Review and Update.药物性肌病:全面综述与更新
Biomedicines. 2024 Apr 30;12(5):987. doi: 10.3390/biomedicines12050987.
2
Immunotherapy-Induced Overlap Syndrome: Myositis, Myasthenia Gravis, and Myocarditis-A Case Series.免疫疗法诱导的重叠综合征:肌炎、重症肌无力和心肌炎——病例系列
Case Rep Med. 2024 Mar 29;2024:5399073. doi: 10.1155/2024/5399073. eCollection 2024.
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A comprehensive review on immune checkpoint inhibitors induced cardiotoxicity characteristics and associated factors.
免疫检查点抑制剂引起的心脏毒性特征及相关因素的全面综述。
Eur J Med Res. 2023 Nov 8;28(1):495. doi: 10.1186/s40001-023-01464-1.
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Immune Checkpoint Inhibitor-Induced Myocarditis with Myositis/Myasthenia Gravis Overlap Syndrome: A Systematic Review of Cases.免疫检查点抑制剂相关性心肌炎合并肌炎/重症肌无力重叠综合征:病例系统综述。
Oncologist. 2021 Dec;26(12):1052-1061. doi: 10.1002/onco.13931. Epub 2021 Aug 25.
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Myasthenia Gravis: Autoantibody Specificities and Their Role in MG Management.重症肌无力:自身抗体特异性及其在重症肌无力管理中的作用。
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Immune Checkpoint Inhibitor-Induced Myasthenia Gravis.免疫检查点抑制剂诱发的重症肌无力
Front Neurol. 2020 Jul 16;11:634. doi: 10.3389/fneur.2020.00634. eCollection 2020.
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Pembrolizumab-induced myasthenia gravis with myositis and presumable myocarditis in a patient with bladder cancer.一名膀胱癌患者发生帕博利珠单抗诱导的重症肌无力伴肌炎及可能的心肌炎。
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