儿童抗MOG抗体合并抗NMDAR抗体双阳性自身免疫性脑炎2例并文献复习

doi:10.3969/j.issn.1004-583X.2022.08.011

摘要/Abstract

摘要：

目的探讨儿童抗髓鞘少突胶质细胞糖蛋白(myelin oligodendrocyte glycoprotein,MOG)抗体合并抗N-甲基-D-门冬氨酸受体(N-methyl-D-aspartate receptor,NMDAR)抗体双阳性自身免疫性脑炎的临床特点。方法回顾性分析邯郸市中心医院收治的2例抗MOG抗体合并抗NMDAR抗体双阳性自身免疫性脑炎患儿的病例资料,并复习相关文献。结果 2例脑脊液白细胞数目均增多,1例颅脑磁共振成像(magnetic resonance imaging,MRI)同时存在幕上及幕下病变,1例仅有幕上病变且其视频脑电图结果与抗NMDAR脑炎典型脑电图相似。经甲泼尼龙冲击、人免疫球蛋白治疗后,2例症状均得到改善,3个月后随访,均无复发。结论该病多发于青少年,既有抗MOG抗体相关疾病的脑病表现,也有抗NMDAR脑炎的表现,且以抗NMDAR脑炎的表现更易被识别。颅脑MRI及视频脑电图等无创检查对此病诊断有一定提示意义,但确诊仍需神经抗体检查,诊疗方面一线治疗为激素、免疫球蛋白、血浆置换,多数患儿对此治疗反应良好,但发病机制仍需进一步研究。

关键词: 脑炎, 抗N-甲基-D-门冬氨酸受体脑炎, 抗髓鞘少突胶质细胞糖蛋白抗体, 儿童

Abstract:

Objective To explore clinical features of double-positive autoimmune encephalitis of children's anti-myelin oligodendrocyte glycoprotein (MOG) antibodies complicated with anti-N-methyl-D-aspartate receptor (NMDAR) antibodies. Methods The case data of 2 children with anti-MOG antibodies combined with anti-NMDAR antibody double-positive autoimmune encephalitis admitted to Handan Central Hospital were retrospectively analyzed, and the relevant literature was reviewed. Results The number of leukocytes in cerebrospinal fluid increased in 2 cases, both supratentorial and infratentorial lesions were found in the head magnetic resonance imaging (MRI) of one case, one case was found to have supratentorial lesions only, and the video electroencephalogram outcome was similar to that of anti-NMDAR encephalitis. The symptoms of both two cases were improved after being treated with methylprednisolone and human immunoglobulin, and no recurrence was found with either case after 3 months of follow-up. Conclusion The disease is more common in adolescents, both encephalopathic manifestations of anti-MOG antibody-related diseases and anti-NMDAR encephalitis are noted, and the manifestations of anti-NMDAR encephalitis are also noted. Noninvasive examinations including cranial MRI and video electroencephalogram show certain indications, and neuroantibody examination becomes necessary for confirmation. The first-line therapies involve the hormone, immunoglobulin and plasma exchange. Most children respond well to the above therapies, while the pathogenesis remains further study.

Key words: encephalitis, anti-N-methyl-D-aspartate receptor encephalitis, anti-myelin oligodendrocyte glycoprotein antibodies, children

中图分类号:

R512.31

胡文聪, 刘秀珍. 儿童抗MOG抗体合并抗NMDAR抗体双阳性自身免疫性脑炎2例并文献复习[J]. 临床荟萃, 2022, 37(8): 733-737.

Hu Wencong, Liu Xiuzhen. Two cases of children's anti-MOG antibodies combined with anti-NMDAR antibody double positive autoimmune encephalitis and literature review[J]. Clinical Focus, 2022, 37(8): 733-737.

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链接本文: https://huicui.hebmu.edu.cn/CN/10.3969/j.issn.1004-583X.2022.08.011

https://huicui.hebmu.edu.cn/CN/Y2022/V37/I8/733

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参考文献 20

[1]	刘斌, 任海涛, 关鸿志. 抗体相关自身免疫性脑炎的重叠综合征[J]. 中华神经科杂志, 2021, 54(1):71-74.
[2]	Fan S, Xu Y, Ren H, et al. Comparison of myelin oligodendrocyte glycoprotein (MOG)-antibody disease and AQP4-IgG-positive neuromyelitis optica spectrum disorder (NMOSD) when they coexist with anti-NMDA (N-methyl-D-aspartate) receptor encephalitis[J]. Mult Scler Relat Disord, 2018, 20:144-152. doi: 10.1016/j.msard.2018.01.007 URL
[3]	董会卿. MOG抗体介导的特发性炎性脱髓鞘疾病[J]. 中国神经免疫学和神经病学杂志, 2017, 24(2):88-91.
[4]	中国免疫学会神经免疫分会, 邱伟, 徐雁. 抗髓鞘少突胶质细胞糖蛋白免疫球蛋白G抗体相关疾病诊断和治疗中国专家共识[J]. 中国神经免疫学和神经病学杂志, 2020, 27(2):86-95.
[5]	Ramanathan S, Dale RC, Brilot F. Anti-MOG antibody: The history, clinical phenotype, and pathogenicity of a serum biomarker for demyelination[J]. Autoimmun Rev, 2016, 15(4):307-324. doi: 10.1016/j.autrev.2015.12.004 pmid: 26708342
[6]	武文卿, 袁征, 何允刚, 等. NMDA受体的结构及其生理作用[J]. 生物技术通讯, 2013, 24(4):584-586.
[7]	Dale RC, Gorman MP, Lim M. Autoimmune encephalitis in children: Clinical phenomenology, therapeutics, and emerging challenges[J]. Curr Opin Neurol, 2017, 30(3):334-344. doi: 10.1097/WCO.0000000000000443 pmid: 28234797
[8]	Florance NR, Davis RL, Lam C, et al. Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis in children and adolescents[J]. Ann Neurol, 2009, 66(1):11-18. doi: 10.1002/ana.21756 pmid: 19670433
[9]	关鸿志, 王佳伟. 中国自身免疫性脑炎诊治专家共识[J]. 中华神经科杂志, 2017, 50(2):91-98.
[10]	Chen C, Liu C, Fang L, et al. Different magnetic resonance imaging features between MOG antibody- and AQP4 antibody-mediated disease: A Chinese cohort study[J]. J Neurol Sci, 2019, 405:116430. doi: 10.1016/j.jns.2019.116430 URL
[11]	Titulaer MJ, Höftberger R, Iizuka T, et al. Overlapping demyelinating syndromes and anti-N-methyl-D-aspartate receptor encephalitis[J]. Ann Neurol, 2014, 75(3):411-428. pmid: 24700511
[12]	Hou C, Wu W, Tian Y, et al. Clinical analysis of anti-NMDAR encephalitis combined with MOG antibody in children[J]. Mult Scler Relat Disord, 2020, 42:102018. doi: 10.1016/j.msard.2020.102018 URL
[13]	李清晨, 卢沛琦, 滕军放. MOGAD合并抗NMDAR脑炎的临床特点分析[J]. 中华神经医学杂志, 2022, 21(1):54-62.
[14]	曹丽平, 马晓宇, 吴远华, 等. 抗N-甲基-D-天冬氨酸受体脑炎与髓鞘少突胶质细胞糖蛋白抗体相关疾病并存的临床特征[J]. 中山大学学报(医学科学版), 2020, 41(6):858-866.
[15]	Zhang J, Ji T, Chen Q, et al. Pediatric autoimmune encephalitis: Case series from two chinese tertiary pediatric neurology centers[J]. Front Neurol, 2019, 10:906. doi: 10.3389/fneur.2019.00906 pmid: 31507515
[16]	杨丹, 马红梅, 闫鹤立. NMDA重叠MOG抗体阳性的脑炎2例及文献复习[J]. 中风与神经疾病杂志, 2021, 38(7):643-646.
[17]	朱红敏, 孙丹, 吴革菲, 等. 儿童MOG抗体病合并抗NMDAR脑炎重叠综合征二例[J]. 中华儿科杂志, 2020, 58(4):324-326.
[18]	刘雅微, 韩婉君, 吕亚楠, 等. 抗NMDAR脑炎合并抗MOG炎性脱髓鞘疾病的研究进展[J]. 中国免疫学杂志, 2021, 37(24):3062-3064.
[19]	Sarigecili E, Cobanogullari MD, Komur M, et al. A rare concurrence: Antibodies against myelin oligodendrocyte glycoprotein and N-methyl-D-aspartate receptor in a child[J]. Mult Scler Relat Disord, 2019, 28:101-103. doi: 10.1016/j.msard.2018.12.017 URL
[20]	Konuskan B, Yildirim M, Gocmen R, et al. Retrospective analysis of children with myelin oligodendrocyte glycoprotein antibody-related disorders[J]. Mult Scler Relat Disord, 2018, 26:1-7. doi: 10.1016/j.msard.2018.07.022 URL