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Evolution of brain MRI lesions in paediatric myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD) and its relevance to disease course
Journal of Neurology, Neurosurgery, and Psychiatry ( IF 11.0 ) Pub Date : 2024-05-01 , DOI: 10.1136/jnnp-2023-332542
Omar Abdel-Mannan 1 , Dimitrios Champsas 1, 2 , Carmen Tur 1, 3 , Vanessa Lee 4 , Sharmila Manivannan 5 , Haroon Usman 6 , Alison Skippen 7 , Ishita Desai 8 , Manali Chitre 9 , Rob Forsyth 10 , Rachel Kneen 11, 12 , Dipak Ram 8 , Sithara Ramdas 7 , Thomas Rossor 4 , Siobhan West 8 , Sukhvir Wright 13, 14 , Jacqueline Palace 15 , Evangeline Wassmer 13, 14 , Cheryl Hemingway 16 , Ming J Lim 4 , Kshitij Mankad 17 , Olga Ciccarelli 1, 18 , Yael Hacohen 2, 19 ,
Affiliation  

Background Lesion resolution is often observed in children with myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD), and asymptomatic lesions are less commonly reported in MOGAD than in multiple sclerosis (MS). Objective We aimed to evaluate brain MRI changes over time in paediatric MOGAD. Methods Retrospective study in eight UK paediatric neuroscience centres. Acute brain MRI and available follow-up MRIs were reviewed. Predictors for lesion dynamic were evaluated using multivariable regression and Kaplan-Meier survival analyses were used to predict risk of relapse, disability and MOG-Ab status. Results 200 children were included (MOGAD 97; MS 103). At first MRI post attack, new symptomatic and asymptomatic lesions were seen more often in MS versus MOGAD (52/103 vs 28/97; p=0.002 and 37/103 vs 11/97; p<0.001); 83% of patients with MOGAD showed at least one lesion’s resolution at first follow‐up scan, and 23% had normal MRI. Only 1 patient with MS had single lesion resolution; none had normal MRI. Disappearing lesions in MOGAD were seen in 40% after the second attack, 21% after third attack and none after the fourth attack. New lesions at first follow-up scan were associated with increased likelihood of relapse (p=0.02) and persistent MOG-Ab serostatus (p=0.0016) compared with those with no new lesions. Plasma exchange was associated with increased likelihood of lesion resolution (p=0.01). Longer time from symptom onset to steroids was associated with increased likelihood of new lesions; 50% increase at 20 days (p=0.01). Conclusions These striking differences in lesion dynamics between MOGAD and MS suggest greater potential to repair. Early treatment with steroids and plasma exchange is associated with reduced likelihood of new lesions. Data are available upon reasonable request. For purposes of replicating procedures and results, any qualified investigator can request anonymised data after ethics clearance and approval by all authors.

中文翻译:

儿科髓鞘少突胶质细胞糖蛋白抗体相关疾病(MOGAD)脑部 MRI 病变的演变及其与病程的相关性

背景 髓鞘少突胶质细胞糖蛋白抗体相关疾病 (MOGAD) 儿童中经常观察到病变消退,与多发性硬化症 (MS) 相比,MOGAD 中无症状病变的报道较少。目的 我们的目的是评估儿科 MOGAD 随时间的推移脑部 MRI 变化。方法 在英国八个儿科神经科学中心进行回顾性研究。对急性脑部 MRI 和可用的后续 MRI 进行了审查。使用多变量回归评估病变动态的预测因子,并使用 Kaplan-Meier 生存分析来预测复发、残疾和 MOG-Ab 状态的风险。结果 纳入了 200 名儿童(MOGAD 97;MS 103)。发作后首次 MRI 发现,与 MOGAD 相比,MS 中更常见新的有症状和无症状病变(52/103 vs 28/97;p=0.002 和 37/103 vs 11/97;p<0.001); 83% 的 MOGAD 患者在第一次随访扫描时显示至少一个病灶已消退,23% 的 MRI 正常。只有 1 名 MS 患者的单个病灶得到缓解;没有人的 MRI 检查结果正常。 MOGAD 中 40% 的病灶在第二次发作后消失,21% 在第三次发作后消失,第四次发作后无病灶消失。与没有新病灶的患者相比,首次随访扫描时出现的新病灶与复发可能性增加 (p=0.02) 和持续 MOG-Ab 血清状态 (p=0.0016) 相关。血浆置换与病变消退的可能性增加相关(p=0.01)。从症状出现到使用类固醇的时间越长,新病变的可能性就会增加; 20 天时增加 50% (p=0.01)。结论 MOGAD 和 MS 之间病变动态的这些显着差异表明修复潜力更大。早期使用类固醇和血浆置换治疗可降低新病变的可能性。数据可根据合理要求提供。为了复制程序和结果,任何合格的研究者都可以在道德审查和所有作者批准后请求匿名数据。
更新日期:2024-04-12
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