Scarlett L Harris, Sophie N M Binks, Donal Skelly, Hanine Fourie, Phoebe Cherrington-Walker, Tomasz Bajorek, Sarosh R Irani, M Isabel Leite, Adam E Handel, Adam Al-Diwani
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引用次数: 0
Abstract
Background: N-methyl-D-aspartate receptor antibody encephalitis (NMDAR-Ab-E) can have an onset during, after or prior to a pregnancy. In animal models, transplacental NMDAR immunoglobulin G transfer can affect neurodevelopment. In contrast, clinical reports of mothers affected by NMDAR-Ab-E typically are reassuring. We systematically reviewed maternal, infant and childhood clinical data pertaining to NMDAR-Ab-E with an onset before, during or after pregnancy and compared this to our single autoimmune neurology centre experience.
Methods: After pre-registration on PROSPERO (CRD42023408447), we searched PubMed and Scopus for NMDAR-Ab-E case reports/series with an onset before, during or after pregnancy (last search 19/10/2023). We extracted maternal, neonatal and childhood outcomes using an idealised checklist to derive summary statistics.
Results: After quality control, we identified 66 pregnancies in 61 women from 48 reports or series. 72% of women recovered with minimal or no neurological deficits, comparable to non-pregnancy-associated NMDAR-Ab-E. Likewise, 80% of pregnancies resulted in live births with a single neonatal death reported. Data on neonatal outcome measures were frequently unreported, and childhood follow-up was provided in only 60%. Our centre's experience is consistent: 3/4 mothers recovered with no functional deficits and 7/8 children without evidence of compromise at a median follow-up of 2 years.
Conclusions: Current evidence does not overall suggest unfavourable maternal, fetal or childhood outcomes after NMDAR-Ab-E. However, the available sample is small, predominantly single case reports with modest follow-up, lacks standardisation, and data are often incomplete. Future approaches should address these caveats: developing multi-centre collaboration towards an international registry.
背景:n -甲基- d -天冬氨酸受体抗体脑炎(NMDAR-Ab-E)可在妊娠期间、妊娠后或妊娠前发病。在动物模型中,经胎盘NMDAR免疫球蛋白G转移可影响神经发育。相比之下,NMDAR-Ab-E影响母亲的临床报告通常是令人放心的。我们系统地回顾了与NMDAR-Ab-E有关的孕妇、婴儿和儿童在怀孕前、怀孕期间或怀孕后发病的临床数据,并将其与我们单一的自身免疫神经学中心的经验进行了比较。方法:在PROSPERO (CRD42023408447)预注册后,检索PubMed和Scopus,检索在妊娠前、妊娠中或妊娠后发病的NMDAR-Ab-E病例报告/系列(最后检索19/10/2023)。我们使用理想的检查表提取产妇、新生儿和儿童的结局,得出汇总统计数据。结果:经过质量控制,我们从48份报告或系列中确定了61名妇女的66例妊娠。72%的妇女恢复时只有很少或没有神经功能障碍,与非妊娠相关的NMDAR-Ab-E相当。同样,80%的怀孕导致活产,报告有一例新生儿死亡。新生儿结局测量数据经常未报告,只有60%的儿童提供了随访。我们中心的经验是一致的:3/4的母亲康复后没有功能缺陷,7/8的孩子在中位随访2年期间没有损伤的证据。结论:目前的证据总体上并不表明NMDAR-Ab-E治疗对母体、胎儿或儿童的预后不利。然而,可获得的样本很小,主要是单个病例报告,随访时间不长,缺乏标准化,数据往往不完整。未来的方法应该解决这些问题:发展多中心合作,建立一个国际登记处。