Pohl Daniela, Alper Gulay, Van Haren Keith, Kornberg Andrew J, Lucchinetti Claudia F, Tenembaum Silvia, Belman Anita L
From the Division of Neurology (D.P.), Children's Hospital of Eastern Ontario, University of Ottawa, Canada; Department of Pediatrics (G.A.), Division of Child Neurology, University of Pittsburgh School of Medicine, Children's Hospital of Pittsburgh, PA; Department of Neurology (K.V.H.), Stanford University; Division of Child Neurology (K.V.H.), Lucile Packard Children's Hospital, Palo Alto, CA; Department of Neurology (A.J.K.), Royal Children's Hospital, Parkville, Australia; Department of Neurology (C.F.L.), Mayo Clinic College of Medicine, Rochester, MN; Department of Neurology (S.T.), National Pediatric Hospital Dr. Juan P. Garrahan, Ciudad de Buenos Aires, Argentina; and Department of Neurology (A.L.B.), School of Medicine, Stony Brook University, Stony Brook, NY.
Neurology. 2016 Aug 30;87(9 Suppl 2):S38-45. doi: 10.1212/WNL.0000000000002825.
Acute disseminated encephalomyelitis (ADEM) is an immune-mediated demyelinating CNS disorder with predilection to early childhood. ADEM is generally considered a monophasic disease. However, recurrent ADEM has been described and defined as multiphasic disseminated encephalomyelitis. ADEM often occurs postinfectiously, although a causal relationship has never been established. ADEM and multiple sclerosis are currently viewed as distinct entities, generally distinguishable even at disease onset. However, pathologic studies have demonstrated transitional cases of yet unclear significance. ADEM is clinically defined by acute polyfocal neurologic deficits including encephalopathy. MRI typically demonstrates reversible, ill-defined white matter lesions of the brain and often also the spinal cord, along with frequent involvement of thalami and basal ganglia. CSF analysis may reveal a mild pleocytosis and elevated protein, but is generally negative for intrathecal oligoclonal immunoglobulin G synthesis. In the absence of a specific diagnostic test, ADEM is considered a diagnosis of exclusion, and ADEM mimics, especially those requiring a different treatment approach, have to be carefully ruled out. The role of biomarkers, including autoantibodies like anti-myelin oligodendrocyte glycoprotein, in the pathogenesis and diagnosis of ADEM is currently under debate. Based on the presumed autoimmune etiology of ADEM, the current treatment approach consists of early immunotherapy. Outcome of ADEM in pediatric patients is generally favorable, but cognitive deficits have been reported even in the absence of other neurologic sequelae. This review summarizes the current knowledge on epidemiology, pathology, clinical presentation, neuroimaging features, CSF findings, differential diagnosis, therapy, and outcome, with a focus on recent advances and controversies.
急性播散性脑脊髓炎(ADEM)是一种免疫介导的中枢神经系统脱髓鞘疾病,好发于儿童早期。ADEM通常被认为是单相疾病。然而,复发性ADEM已被描述并定义为多相性播散性脑脊髓炎。ADEM常发生于感染后,尽管因果关系尚未确立。目前,ADEM和多发性硬化被视为不同的疾病实体,即使在疾病发作时通常也可区分。然而,病理研究已证实存在意义尚不清楚的过渡病例。ADEM的临床定义为包括脑病在内的急性多灶性神经功能缺损。MRI通常显示大脑以及脊髓可逆性、边界不清的白质病变,丘脑和基底节也常受累。脑脊液分析可能显示轻度细胞增多和蛋白升高,但鞘内寡克隆免疫球蛋白G合成通常为阴性。在缺乏特异性诊断检测的情况下,ADEM被认为是一种排除性诊断,必须仔细排除ADEM的模仿疾病,尤其是那些需要不同治疗方法的疾病。包括抗髓鞘少突胶质细胞糖蛋白等自身抗体在内的生物标志物在ADEM发病机制和诊断中的作用目前仍存在争议。基于ADEM推测的自身免疫病因,目前的治疗方法包括早期免疫治疗。儿科患者ADEM的预后通常良好,但即使没有其他神经后遗症,也有认知缺陷的报道。本综述总结了目前关于流行病学、病理学、临床表现、神经影像学特征、脑脊液检查结果、鉴别诊断、治疗和预后的知识,重点关注最新进展和争议。