MDUK Neuromuscular Centre, Department of Paediatrics, University of Oxford, United Kingdom; Department of Paediatric Neurology, John Radcliffe Hospital, Oxford, United Kingdom.
Department of Neuropediatrics, Developmental Neurology and Social Pediatrics, Children's Hospital, University of Duisburg-Essen, Germany.
Eur J Paediatr Neurol. 2022 Sep;40:5-10. doi: 10.1016/j.ejpn.2022.06.009. Epub 2022 Jun 25.
Juvenile myasthenia gravis (JMG) is a rare, antibody-mediated disorder of the neuromuscular junction. Treatment strategies in JMG are largely informed by adult MG treatments as the pathophysiology is similar. Rituximab is increasingly considered as a treatment option in refractory JMG but has not yet been systematically investigated in this patient group We conducted a retrospective study from five international centres with expertise in paediatric myasthenia. 10 JMG patients treated with rituximab were identified. Following rituximab treatment all patients had a reduction in JMG-related hospital admissions. At 24 month follow up, 6 patients (60%) had achieved complete stable remission or pharmacological remission and 7 patients were able to reduce immunomodulatory treatment(s). The main side-effect was infusion-related reactions (30%) which resolved in all patients with symptomatic treatment. We compared our cohort to previously reported JMG cases treated with rituximab and noted similar response rates but a slightly higher side-effect profile. Rituximab is a safe and effective treatment option in moderate to severe JMG and most patients have an improvement in MG symptoms post treatment.
青少年重症肌无力(JMG)是一种罕见的、由神经肌肉接头抗体介导的疾病。由于其病理生理学相似,JMG 的治疗策略在很大程度上借鉴了成人 MG 的治疗方法。利妥昔单抗作为难治性 JMG 的治疗选择越来越受到关注,但尚未在该患者群体中进行系统研究。我们对五个在儿科重症肌无力方面具有专业知识的国际中心进行了回顾性研究。确定了 10 例接受利妥昔单抗治疗的 JMG 患者。接受利妥昔单抗治疗后,所有患者的 JMG 相关住院次数均减少。在 24 个月的随访中,6 名患者(60%)达到完全稳定缓解或药物缓解,7 名患者能够减少免疫调节治疗。主要副作用是输液相关反应(30%),所有患者均通过对症治疗得到缓解。我们将我们的队列与之前报道的接受利妥昔单抗治疗的 JMG 病例进行了比较,发现相似的缓解率,但副作用略高。利妥昔单抗是中重度 JMG 的一种安全有效的治疗选择,大多数患者在治疗后 MG 症状得到改善。