Matricardi Sara, Farello Giovanni, Savasta Salvatore, Verrotti Alberto
Department of Neuropsychiatry, Children's Hospital "G. Salesi", Ospedali Riuniti Ancona, Ancona, Italy.
Pediatric Clinic, Department of Life, Health and Environmental Sciences, University of L'Aquila, L'Aquila, Italy.
Front Pediatr. 2019 Dec 19;7:511. doi: 10.3389/fped.2019.00511. eCollection 2019.
Immune-mediated diseases of the central nervous system (CNS) in childhood are a heterogeneous group of rare conditions sharing the inflammatory involvement of the CNS. This review highlights the growing knowledge of childhood neuroimmune diseases that primarily affect the CNS, outlining the clinical and diagnostic features, the pathobiological mechanisms and genetics, current treatment options, and emerging challenges. The clinical spectrum of these conditions is increasingly expanded, and the underlying mechanisms of dysregulation of the immune system could vary widely. Cell-mediated and antibody-mediated disorders, infection-triggered and paraneoplastic conditions, and genetically defined mechanisms can occur in previously healthy children and can contribute to different stages of the disease. The careful evaluation of the clinical presentation and temporal course of symptoms, the specific neuroimaging and immunological findings, and the exclusion of alternative causes are mandatory in clinical practice for the syndromic diagnosis. A common feature of these conditions is that immunotherapeutic agents could modulate the clinical course and outcomes of the disease. Furthermore, specific symptomatic treatments and comprehensive multidisciplinary care are needed in the overall management. We focus on recent advances on immune-mediated demyelinating CNS disorders, autoimmune encephalitis, interferonopathies, and possible neuroimmune disorders as Rasmussen encephalitis. Better knowledge of these conditions could allow prompt diagnosis and targeted immunotherapy, to decrease morbidity and mortality as well as to improve clinical outcomes, reducing the burden of the disease due to possible long-term neuropsychiatric sequelae. Persisting controversies remain in the rigorous characterization of each specific clinical entity because of the relative rarity in children; moreover, in a large proportion of suspected neuroimmune diseases, the immune "signature" remains unidentified; treatment guidelines are mostly based on retrospective cohort studies and expert opinions; then advances in specific molecular therapies are required. In the future, a better characterization of specific immunological biomarkers may provide a useful understanding of the underlying pathobiological mechanisms of these conditions in order to individualize more tailored therapeutic options and paradigms. Multicenter collaborative research on homogeneous groups of patients who may undergo immunological studies and therapeutic trials could improve the characterization of the underlying mechanisms, the specific phenotypes, and tailored management.
儿童期中枢神经系统(CNS)免疫介导性疾病是一组异质性罕见病症,其共同特征是CNS存在炎症累及。本综述着重介绍了对主要影响CNS的儿童期神经免疫疾病的不断深入了解,概述了临床和诊断特征、病理生物学机制及遗传学、当前的治疗选择以及新出现的挑战。这些病症的临床谱在不断扩大,免疫系统失调的潜在机制可能差异很大。细胞介导和抗体介导的疾病、感染引发和副肿瘤性病症以及基因决定的机制可发生于既往健康的儿童,并可导致疾病的不同阶段。在临床实践中,对综合征诊断而言,仔细评估临床表现和症状的时间进程、特定的神经影像学和免疫学发现以及排除其他病因是必不可少的。这些病症的一个共同特点是免疫治疗药物可调节疾病的临床进程和结局。此外,在整体管理中需要特定的对症治疗和全面的多学科护理。我们重点关注免疫介导的中枢神经系统脱髓鞘疾病、自身免疫性脑炎、干扰素病以及如拉斯穆森脑炎等可能的神经免疫疾病的最新进展。对这些病症有更深入的了解可实现早期诊断和靶向免疫治疗,以降低发病率和死亡率,并改善临床结局,减少因可能的长期神经精神后遗症导致的疾病负担。由于这些病症在儿童中相对罕见,因此在对每个特定临床实体进行严格特征描述方面仍存在争议;此外,在很大一部分疑似神经免疫疾病中,免疫“特征”仍未明确;治疗指南大多基于回顾性队列研究和专家意见;因此需要在特定分子疗法方面取得进展。未来,对特定免疫生物标志物进行更好的特征描述可能有助于深入了解这些病症的潜在病理生物学机制,从而使治疗选择和模式更加个体化。针对可能接受免疫学研究和治疗试验的同类患者群体开展多中心合作研究,可能会改善对潜在机制、特定表型和个体化管理的特征描述。