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病例报告:免疫检查点抑制剂的神经肌肉三联征:托匹单抗治疗后肌炎、心肌炎和重症肌无力重叠的病例报告

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

作者信息

Luo Yue-Bei, Tang Weiting, Zeng Qiuming, Duan Weiwei, Li Shuyu, Yang Xiaosu, Bi Fangfang

机构信息

Department of Neurology, Xiangya Hospital, Central South University, Changsha, China.

出版信息

Front Cardiovasc Med. 2021 Aug 16;8:714460. doi: 10.3389/fcvm.2021.714460. eCollection 2021.

Abstract

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.

摘要

免疫检查点抑制剂(ICI)治疗的神经肌肉不良事件包括肌炎、风湿性多肌痛、心肌炎和肌无力综合征。我们报告一名47岁女性,在接受托瑞帕利单抗治疗转移性胸腺瘤4周后出现眼外肌麻痹、全身肌肉无力和三度房室传导阻滞。肌酸激酶升高至25200 U/l,心肌肌钙蛋白I升高至2.796 ng/ml。自身抗体谱显示抗兰尼碱受体和抗乙酰胆碱受体抗体阳性,肌炎特异性抗体阴性。重复神经刺激未显示复合肌肉动作电位衰减。静脉注射甲泼尼龙和免疫球蛋白,以及临时植入起搏器使她的肌肉酶水平和心律恢复正常。这是托瑞帕利单抗重叠性神经肌肉不良事件的首例报告。

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