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托泊替康治疗后以小脑受累为主的抗 GAD65 抗体相关自身免疫性脑炎:托泊替康一种新型 irAE 的病例报告。

Anti-GAD65 Antibody-Associated Autoimmune Encephalitis With Predominant Cerebellar Involvement Following Toripalimab Treatment: A Case Report of a Novel irAE of Toripalimab.

机构信息

Department of Neurology, West China Hospital, Sichuan University, Chengdu, China.

Department of Gastroenterology, West China Hospital, Sichuan University, Chengdu, China.

出版信息

Front Immunol. 2022 Mar 25;13:850540. doi: 10.3389/fimmu.2022.850540. eCollection 2022.

DOI:10.3389/fimmu.2022.850540
PMID:35401563
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8990884/
Abstract

Toripalimab (Junshi Bioscience Co., Ltd) is a new immune checkpoint inhibitor (ICI) that targets programmed cell death protein 1 (PD-1) in various cancers, including metastatic melanoma. No neurological immune-related adverse events (n-irAEs) of toripalimab have been reported, except for neuromuscular involvement. We report a case of a 63-year-old woman who presented with severe vertigo, vomiting, nystagmus, cerebellar ataxia, and cognitive impairment after toripalimab treatment for metastatic melanoma. Compared with the concomitant cognitive dysfunction and a pathological reflex involving the cerebral cortex, the signs and symptoms of cerebellar involvement were much more prominent. Anti-glutamic acid decarboxylase 65 (anti-GAD65) antibody was positive in both serum and cerebrospinal fluid (CSF). After intravenous immunoglobulin (IVIG) and methylprednisolone (IVMP) administration, the symptoms of vertigo and vomiting resolved, with cognitive impairment and cerebellar ataxia remaining. This is the first report of autoimmune encephalitis (AIE) as an n-irAE of toripalimab.

摘要

特瑞普利单抗(君实生物)是一种新型的免疫检查点抑制剂(ICI),可靶向多种癌症中的程序性死亡蛋白 1(PD-1),包括转移性黑色素瘤。除了神经肌肉受累外,尚未报道特瑞普利单抗的神经免疫相关不良事件(n-irAEs)。我们报告了一例 63 岁女性病例,在转移性黑色素瘤接受特瑞普利单抗治疗后出现严重眩晕、呕吐、眼球震颤、小脑共济失调和认知障碍。与同时存在的认知功能障碍和涉及大脑皮层的病理反射相比,小脑受累的体征和症状更为明显。血清和脑脊液(CSF)中均检测到抗谷氨酸脱羧酶 65(anti-GAD65)抗体阳性。静脉注射免疫球蛋白(IVIG)和甲基强的松龙(IVMP)治疗后,眩晕和呕吐症状缓解,认知障碍和小脑共济失调仍存在。这是特瑞普利单抗引起的自身免疫性脑炎(AIE)作为 n-irAE 的首例报告。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/92a5/8990884/3774d3e9ea9d/fimmu-13-850540-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/92a5/8990884/3774d3e9ea9d/fimmu-13-850540-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/92a5/8990884/3774d3e9ea9d/fimmu-13-850540-g001.jpg

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本文引用的文献

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J Neuroimmune Pharmacol. 2022 Dec;17(3-4):377-379. doi: 10.1007/s11481-021-10026-3. Epub 2021 Oct 22.
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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.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
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Management of Paraneoplastic Syndromes in the Era of Immune Checkpoint Inhibitors.免疫检查点抑制剂时代的副肿瘤综合征的管理。
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