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小儿抗 N-甲基-D-天冬氨酸受体(抗 NMDAR)脑炎中的复发性视神经炎:病例报告及文献综述

Recurrent Optic Neuritis in Paediatric Anti-N-Methyl-D-Aspartate Receptor (Anti-NMDAR) Encephalitis: Case Report and Literature Review.

作者信息

Chan Jinn Shian, Nurul-Ain Masnon, Mohamad Nor Fadhilah, Wan Hitam Wan Hazabbah, Thavaratnam Lakana Kumar

机构信息

UM Eye Research Centre, Department of Ophthalmology, Faculty of Medicine, University of Malaya, Kuala Lumpur, Malaysia.

Department of Ophthalmology, Hospital Kuala Lumpur, Ministry of Health, Kuala Lumpur, Malaysia.

出版信息

Neuroophthalmology. 2022 Jan 13;46(4):258-263. doi: 10.1080/01658107.2021.2014892. eCollection 2022.

Abstract

Optic neuritis associated with anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis poses a novel challenge and is recognised as being part of overlapping antibody-associated demyelinating syndromes. We report a case of a 10-year-old girl who first presented with anti-NMDAR encephalitis and later developed four episodes of left optic neuritis. Blurring of vision, pain on eye movement, and headache were the striking features in all episodes of optic neuritis. For each recurrent episode of optic neuritis she was treated with intravenous methylprednisolone, following which visual acuity, colour vision, and visual field improved to normal. Neuroimaging was unremarkable. Myelin oligodendrocyte glycoprotein and aquaporin-4 antibody tests were negative. She developed a relapse of anti-NMDAR encephalitis after recurrent episodes of optic neuritis despite being on azathioprine. Intravenous immunoglobulin and cycles of cyclophosphamide were started, and she has been free from optic neuritis and encephalitis for more than one year since. Optic neuritis and anti-NMDAR encephalitis are reversible with intensive immunotherapy. Early and accurate diagnosis can lead to better management of this condition.

摘要

与抗 N-甲基-D-天冬氨酸受体(NMDAR)脑炎相关的视神经炎带来了新的挑战,并且被认为是重叠性抗体相关脱髓鞘综合征的一部分。我们报告一例 10 岁女孩,她最初表现为抗 NMDAR 脑炎,后来出现了四次左侧视神经炎发作。视力模糊、眼球运动时疼痛和头痛是所有视神经炎发作的显著特征。每次视神经炎复发时,她都接受静脉注射甲基泼尼松龙治疗,之后视力、色觉和视野恢复正常。神经影像学检查无异常。髓鞘少突胶质细胞糖蛋白和水通道蛋白-4 抗体检测均为阴性。尽管服用硫唑嘌呤,但在视神经炎反复发作后,她出现了抗 NMDAR 脑炎复发。开始静脉注射免疫球蛋白和环磷酰胺疗程,自那以后她已超过一年没有出现视神经炎和脑炎。强化免疫治疗可使视神经炎和抗 NMDAR 脑炎可逆。早期准确诊断可更好地管理这种疾病。

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