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目的:探讨免疫检测点抑制剂（ICI）相关无菌性脑膜炎的临床特点、诊断及治疗。方法:总结2019年12月11日就诊于北京协和医院的1例帕博利珠单抗相关无菌性脑膜炎患者的临床资料和诊治过程，同时系统性回顾国内外文献所发表的ICI相关无菌性脑膜炎的病例报告或病例系列研究，分析其性别、年龄、基础肿瘤、发病时间、临床表现、治疗及预后等情况。结果:本例患者为45岁男性，因患食管癌使用帕博利珠单抗，2年后出现无菌性脑膜炎，经停用帕博利珠单抗，并接受糖皮质激素治疗，症状很快好转。分析报道的全部9例ICI相关无菌性脑膜炎病例，中位年龄55岁，男女比例为6∶3，多数患者出现头痛而无脑膜刺激征，少数患者出现发热，所有患者均接受糖皮质激素治疗，症状均好转。结论:ICI相关无菌性脑膜炎是接受ICI治疗的患者中罕见而重要的不良反应。对于接受ICI治疗的患者，若出现头痛、发热等症状，需警惕该病，并与脑膜癌病、感染性脑膜炎等疾病相鉴别。“,”Objective:To investigate the clinical features, diagnosis, therapy and prognosis of patients with immune checkpoint inhibitor (ICI)-associated aseptic meningitis.Methods:A case of pembrolizumab-associated aseptic meningitis hospitalized in Peking Union Medical College Hospital on December 11, 2019 was reported. Additionally, the literature was systematically reviewed to identify patients with ICI-associated aseptic meningitis, and the clinical characteristics, therapy and prognosis of these patients were analyzed.Results:The present case is a 45-year-old man who developed ICI-associated aseptic meningitis two years after the use of pembrolizumab for his esophageal cancer. He responded well to treatment with corticosteroids in addition to the cessation of pembrolizumab. Nine patients with ICI-associated aseptic meningitis were identified in the literature, with a median onset age of 55 years. Six patients were male and three were female. Most of the patients presented with headache without neck stiffness, while a few patients presented with fever. All patients responded well to treatment with corticosteroids.Conclusions:ICI-associated aseptic meningitis is a rare but important adverse event in patients treated with ICI. This disease should be suspected when patients treated with ICI present with headache and fever, and should be differentiated from meningeal carcinomatosis and infective meningitis.