中国实用儿科杂志

中国实用儿科杂志

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利妥昔单抗治疗复发性眼球阵挛-肌阵挛综合征的疗效观察

  

  1. 1.北京大学第一医院儿科，北京  100034； 2.成都市妇女儿童中心医院神经内科，四川  成都  610074
  • 出版日期:2018-04-06 发布日期:2018-04-16

Efficacy of rituximab therapy for relapsed opsoclonus myoclonus syndrome

  1. *Department of Pediatrics，Peking University First Hospital，Beijing  100034，China
  • Online:2018-04-06 Published:2018-04-16

摘要：

目的　总结利妥昔单抗对复发性眼球阵挛-肌阵挛综合征（OMS）的疗效。方法　分析北京大学第一医院儿科2012年9月至2015年10月收治的5例复发性OMS应用利妥昔单抗治疗前后的临床资料及随访情况。结果　5例发病年龄为13～20个月（平均16.8个月），入院时病程10～31个月（平均病程16.8个月）。5例均在接受利妥昔单抗治疗前曾接受过激素、大剂量人免疫球蛋白单用或联合治疗，均疗效欠佳并复发。5例均接受利妥昔单抗治疗，随访最短14个月，最长46个月；其中4例临床症状基本控制，1例在随访中复发，但症状较前有减轻。用药期间及随访中未观察到明显不良反应。结论　对复发性的OMS患儿，利妥昔单抗可能是一种安全、有效的治疗手段，但需要大样本的随机对照研究进一步证实。

关键词: 眼球阵挛-肌阵挛综合征, 复发, 利妥昔单抗, 治疗, 疗效

Abstract:

Objective　To summarize the efficacy of rituximab therapy for the relapsed opsoclonus myoclonus syndrome （OMS）. Methods　Analyze the clinical data collected from 5 patients with the relapsed OMS treated with rituximab between 2012 and 2015 at Peking University First Hospital. Results　The onset age ranged from 13 months to 20 months （mean age 16.8 months）. The course of the disease ranged from 10 months to 31 months on admission（average duration 16.8 months）. Before the rituximab therapy，all of them received hormone alone or hormone combined with high dose immunoglobulin therapy，but the disease relapsed due to a poor efficacy. Five patients were treated with rituximab. The follow-up time was 14 months to 46 months. The clinical symptoms of 4 patients were controlled. In 1 case the disease relapsed during follow-up，but the clinical symptoms were improved. There were no severe side effects during medication period or follow-up stage. Conclusion　Our data have shown that rituximab tends to be a safe and effective treatment for the replapsed OMS. An RCT with larger samples is necessary in the future.

Key words: opsoclonus myoclonus syndrome, relapse, rituximab, treatment, efficacy