JNNP:儿童因血清AQP4 IgG阳性视神经脊髓炎谱系疾病致残的早期预测因子

2021-10-07 MedSci原创 MedSci原创

视神经脊髓炎谱系障碍(NMOSD)是一种中枢神经系统(CNS)的炎性脱髓鞘疾病,主要影响视神经和脊髓。约60%–80%的患者血清中含有疾病特异性水通道蛋白-4抗体(AQP4 IgG),导致

视神经脊髓炎谱系障碍(NMOSD)是一种中枢神经系统(CNS)的炎性脱髓鞘疾病,主要影响视神经和脊髓。约60%–80%的患者血清中含有疾病特异性水通道蛋白-4抗体(AQP4 IgG),导致原发性自身免疫性星形细胞病,并伴有特征性复发过程。

只有大约5%的AQP4 IgG血清阳性NMODS是儿科病例。于该疾病的罕见性,AQP4 IgG血清阳性儿科患者的长期临床结果数据很少,并且需要进一步的证据,特别是因为新的治疗方法正在出现,本文是一项长期结果研究,研究对象是从几个英国神经病学中心招募的相对较大的儿童发病AQP4 IgG NMOSD队列。本文发表在《神经病学,神经外科学和精神病学杂志》上()。

回顾性分析了从六个英国视神经脊髓炎中心。所有数据均为各自中心的标准临床护理数据(6个月神经和眼科随访、血清AQP4 IgG检测、诊断磁共振成像和脑脊液-CSF)。随后收集了关于性别、发病年龄、种族、发病类型和免疫抑制治疗、首次复发时间、长期视力残疾时间的信息(最佳视力低于6/36,持续时间超过6个月)、运动障碍(步行)≤500米长,6个月以上无人帮助,扩展残疾状态量表(EDSS)得分)和认知障碍(由神经心理学评估或记录在案的学习障碍定义,需要学校额外支持)。

神经认知测试采用不同的适合年龄的标准测试与三级成人和/或儿科神经科学中心合作的临床心理学家/神经心理学家概述了由临床需要决定的认知能力。这些测试通常包括情景记忆、语言、注意力和运动功能的测量。如果这些领域内的任何测试不合格,则认为认知能力受损。在未进行正式认知评估的患者中,认知障碍被定义为需要学校额外支持的学习障碍。

总计纳入了49例儿童AQP4 IgG患者(38.8%为白人,34.7%为黑人,20.4%为亚洲人,6.1%为混血儿),患者平均发病年龄为12±4.1岁,87.7%为女性。26.5%的患者出现多灶性表现,其中视神经(47%)、末梢/脑干(48.9%)和脑(28.6%)是最易受累的区域。

总的来说,52.3%的儿童在发病后1年内首次复发。发病年龄<12岁的儿童首次复发的可能性更大(p=0.030),尽管与发病年龄为12-18岁的儿童相比,免疫抑制时间没有差异。在79个月的队列中位数疾病持续时间中,34.3%患有永久性视力残疾,20.7%的EDSS评分为4,25.8%的认知障碍。视力障碍与白人(p=0.032)和视神经炎表现(p=0.002)相关。大脑综合征表现可预测认知损害(p=0.048),尤其是对类固醇耐药(p=0.034)。

影响首次复发、视力残疾、EDSS评分和认知障碍概率的早期预测因素

12岁以下的儿童比12-18岁的儿童更早复发。与12岁以下的人相比,12-18岁的人中黑人占占比更大。黑人儿童也更有可能对IVMP不耐受,需要急性二线治疗。发病时发作的儿童和未升级到二线急性治疗的儿童也更早出现视力残疾。长期残疾结果的生存分析证实了年龄对视觉和运动残疾的影响。尽管疾病持续时间的中位数相似,但与以成人为主的队列相比,儿童的视觉残疾风险增加了一倍(36.7%的儿童和18%以成人为主)但达到EDSS评分6的可能性要低三倍(儿童的10.2%比成人占多数的34%),达到EDSS评分8的可能性要低十倍(儿童的2%比成人占多数的23%)。4%的儿童和9%的成年人在相似的疾病持续时间内死亡。

总之,发病年龄、种族、发病症状和发病时对急性治疗的抵抗力可预测首次复发和长期残疾。对这些预测因子的认识可能有助于推动未来的儿科临床试验,并指导AQP4 IgG NMOSD的早期治疗决策。

Camera VMessina SElhadd KT, et al Early predictors of disability of paediatric-onset AQP4-IgG-seropositive neuromyelitis optica spectrum disorders

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    2021-10-14 xlysu

    #脊髓炎#

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    2021-12-18 ms7421025882867189

    #致残#

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    2021-12-27 snowpeakxu
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    2022-05-29 gj0740
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    2021-10-09 宋威
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    2021-10-09 ms5768352621950538

    #IgG#

    0

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