儿童髓鞘少突胶质细胞糖蛋白抗体病复发因素及预防复发方案疗效的回顾性分析

doi:10.7499/j.issn.1008-8830.2104017

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摘要

目的探讨儿童髓鞘少突胶质细胞糖蛋白（myelin oligodendrocyte glycoprotein，MOG）抗体病的临床特点、复发因素及预防复发方案的疗效。方法回顾性分析2014年12月至2020年9月在中南大学湘雅医院小儿神经专科住院的41例儿童MOG抗体病患儿的临床资料，根据患儿是否复发分为单相病程组（n=19）、复发组（n=22），复发患儿根据是否采用预防治疗分为预防治疗组及未预防治疗组。分析各组患儿的临床特征及比较各预防方案治疗前、治疗中年复发率（annualized relapse rate，ARR）的差异。结果 41例患儿中最常见的首发表现为急性播散性脑脊髓炎（56%，23/41）。54%（22/41）患儿复发，共有57次复发事件，最常见的复发事件为视神经炎（30%，17/57）。复发组急性期皮质激素疗程不足3个月的患儿比例高于单相病程组（64% vs 32%，P < 0.05）。预防治疗组和未预防治疗组ARR差异无统计学意义（P > 0.05）。评估32例次预防治疗方案发现，使用利妥昔单抗、硫唑嘌呤的治疗中ARR较治疗前ARR降低（P < 0.05）。结论超半数儿童MOG抗体病会出现复发，多数复发患儿急性期皮质激素疗程不足3个月；利妥昔单抗、硫唑嘌呤可能减少复发风险。

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	朱飒英
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	邓小鹿
	张慈柳
	杨丽芬
	尹飞
	何芳

关键词 ： MOG抗体病, 临床特点, 复发, 免疫治疗, 预防, 儿童

Abstract：Objective To study the clinical features and recurrence factors of myelin oligodendrocyte glycoprotein (MOG) antibody disease in children and the effect of recurrence prevention regimens. Methods A retrospective analysis was performed on the medical data of 41 children with MOG antibody disease who were hospitalized in the Department of Pediatric Neurology, Xiangya Hospital of Central South University, from December 2014 to September 2020. According to the presence or absence of recurrence, they were divided into a monophasic course group (n=19) and a recurrence group (n=22). According to whether preventive treatment for recurrence was given, the children with recurrence were further divided into a preventive treatment group and a non-preventive treatment group. The clinical features were analyzed for all groups, and the annualized relapse rate (ARR) was compared before and after treatment with prevention regimens. Results For these 41 children, acute disseminated encephalomyelitis was the most common initial manifestation and was observed in 23 children (56%). Of the 41 children, 22 (54%) experienced recurrence, with 57 recurrence events in total, among which optic neuritis was the most common event (17/57, 30%). The proportion of children in the recurrence group who were treated with corticosteroids for less than 3 months in the acute phase was higher than that in the monophasic course group (64% vs 32%; P < 0.05). There was no significant difference in the ARR between the preventive treatment and non-preventive treatment groups (P > 0.05). The assessment of preventive treatment regimens for 32 cases showed that the children treated with rituximab or azathioprine had a significant reduction in the ARR during treatment (P < 0.05). Conclusions More than half of the children with MOG antibody disease may experience recurrence. Most children with recurrence are treated with corticosteroids for less than 3 months in the acute phase. Rituximab and azathioprine may reduce the risk of recurrence.

Key words： Myelin oligodendrocyte glycoprotein antibody disease Clinical feature Recurrence Immunotherapy Prevention Child

收稿日期: 2021-04-06

通讯作者: 何芳,女,主治医师。Email:bubbly_ho@163.com。 E-mail: bubbly_ho@163.com

作者简介: 朱飒英,女,硕士研究生,住院医师。

引用本文:

朱飒英,彭镜,毛蕾蕾等. 儿童髓鞘少突胶质细胞糖蛋白抗体病复发因素及预防复发方案疗效的回顾性分析[J]. 中国当代儿科杂志, 2021, 23(7): 724-729.

ZHU Sa-Ying,PENG Jing,MAO Lei-Lei et al. Recurrence factors for myelin oligodendrocyte glycoprotein antibody disease in children and the effect of recurrence prevention regimens[J]. CJCP, 2021, 23(7): 724-729.

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http://www.zgddek.com/CN/10.7499/j.issn.1008-8830.2104017 或 http://www.zgddek.com/CN/Y2021/V23/I7/724

[1]	Bruijstens AL, Lechner C, Flet-Berliac L, et al. E.U. Paediatric MOG consortium consensus:part 1-classification of clinical phenotypes of paediatric myelin oligodendrocyte glycoprotein antibody-associated disorders[J]. Eur J Paediatr Neurol, 2020, 29:2-13. DOI:10.1016/j.ejpn.2020.10.006. PMID:33162302.
[2]	Armangue T, Olivé-Cirera G, Martínez-Hernandez E, et al. Associations of paediatric demyelinating and encephalitic syndromes with myelin oligodendrocyte glycoprotein antibodies:a multicentre observational study[J]. Lancet Neurol, 2020, 19(3):234-246. DOI:10.1016/S1474-4422(19)30488-0. PMID:32057303.
[3]	Jarius S, Ruprecht K, Kleiter I, et al. MOG-IgG in NMO and related disorders:a multicenter study of 50 patients. Part 2:epidemiology, clinical presentation, radiological and laboratory features, treatment responses, and long-term outcome[J]. J Neuroinflammation, 2016, 13(1):280. DOI:10.1186/s12974-016-0718-0. PMID:27793206. PMCID:PMC5086042.
[4]	Wynford-Thomas R, Jacob A, Tomassini V. Neurological update:MOG antibody disease[J]. J Neurol, 2019, 266(5):1280-1286. DOI:10.1007/s00415-018-9122-2. PMID:30569382. PMCID:PMC6469662.
[5]	Bruijstens AL, Wendel EM, Lechner C, et al. E.U. Paediatric MOG consortium consensus:part 5-treatment of paediatric myelin oligodendrocyte glycoprotein antibody-associated disorders[J]. Eur J Paediatr Neurol, 2020, 29:41-53. DOI:10.1016/j.ejpn.2020.10.005. PMID:33176999.
[6]	中国免疫学会神经免疫分会. 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. DOI:10.3969/j.issn.1006-2963.2020.02.002.
[7]	Takai Y, Misu T, Kaneko K, et al. Myelin oligodendrocyte glycoprotein antibody-associated disease:an immunopathological study[J]. Brain, 2020, 143(5):1431-1446. DOI:10.1093/brain/awaa102. PMID:32412053.
[8]	Jarius S, Paul F, Aktas O, et al. MOG encephalomyelitis:international recommendations on diagnosis and antibody testing[J]. J Neuroinflammation, 2018, 15(1):134. DOI:10.1186/s12974-018-1144-2. PMID:29724224. PMCID:PMC5932838.
[9]	Reindl M, Jarius S, Rostasy K, et al. Myelin oligodendrocyte glycoprotein antibodies:how clinically useful are they?[J]. Curr Opin Neurol, 2017, 30(3):295-301. DOI:10.1097/WCO.0000000000000446. PMID:28248700.
[10]	de Mol CL, Wong Y, van Pelt ED, et al. The clinical spectrum and incidence of anti-MOG-associated acquired demyelinating syndromes in children and adults[J]. Mult Scler, 2020, 26(7):806-814. DOI:10.1177/1352458519845112. PMID:31094288. PMCID:PMC7294530.
[11]	侯池, 李小晶, 张雅妮, 等. 儿童髓鞘少突胶质细胞糖蛋白抗体相关复发性脱髓鞘病临床分析[J]. 中华实用儿科临床杂志, 2019, 34(23):1807-1811. DOI:10.3760/cma.j.issn.2095-428X.2019.23.012.
[12]	张炜华, 任晓暾, 韩彤立, 等. 儿童抗髓鞘少突胶质细胞糖蛋白抗体相关中枢神经系统炎性脱髓鞘病临床特征[J]. 中华实用儿科临床杂志, 2019, 34(24):1858-1861. DOI:10.3760/cma.j.issn.2095-428X.2019.24.005.
[13]	Zhou J, Lu XP, Zhang Y, et al. Follow-up study on Chinese children with relapsing MOG-IgG-associated central nervous system demyelination[J]. Mult Scler Relat Disord, 2019, 28:4-10. DOI:10.1016/j.msard.2018.12.001. PMID:30529926.
[14]	周季, 张尧, 季涛云, 等. 儿童视神经脊髓炎谱系疾病临床分析[J]. 中华儿科杂志, 2019, 57(2):118-124. DOI:10.3760/cma.j.issn.0578-1310.2019.02.011. PMID:30695886.
[15]	Krupp LB, Tardieu M, Amato MP, et al. International Pediatric Multiple Sclerosis Study Group criteria for pediatric multiple sclerosis and immune-mediated central nervous system demyelinating disorders:revisions to the 2007 definitions[J]. Mult Scler, 2013, 19(10):1261-1267. DOI:10.1177/1352458513484547. PMID:23572237.
[16]	Wingerchuk DM, Banwell B, Bennett JL, et al. International consensus diagnostic criteria for neuromyelitis optica spectrum disorders[J]. Neurology, 2015, 85(2):177-189. DOI:10.1212/WNL.0000000000001729. PMID:26092914. PMCID:PMC4515040.
[17]	Ramanathan S, Mohammad S, Tantsis E, et al. Clinical course, therapeutic responses and outcomes in relapsing MOG antibody-associated demyelination[J]. J Neurol Neurosurg Psychiatry, 2018, 89(2):127-137. DOI:10.1136/jnnp-2017-316880. PMID:29142145. PMCID:PMC5800335.
[18]	Salama S, Khan M, Shanechi A, et al. MRI differences between MOG antibody disease and AQP4 NMOSD[J]. Mult Scler, 2020, 26(14):1854-1865. DOI:10.1177/1352458519893093. PMID:31937191. PMCID:PMC7363520.
[19]	孙红, 谭建敏, 刘静临, 等. 儿童髓鞘少突胶质细胞糖蛋白抗体相关脱髓鞘病57例临床特征及预后分析[J]. 临床儿科杂志, 2020, 38(11):824-830. DOI:10.3969/j.issn.1000-3606.2020.11.006.
[20]	Hacohen Y, Wong YY, Lechner C, et al. Disease course and treatment responses in children with relapsing myelin oligodendrocyte glycoprotein antibody-associated disease[J]. JAMA Neurol, 2018, 75(4):478-487. DOI:10.1001/jamaneurol.2017.4601. PMID:29305608. PMCID:PMC5885190.