Wunderlich G, Abicht A, Brunn A, Daimagüler H-S, Schroeter M, Fink G R, Lehmann H C, Cirak S
Klinik und Poliklinik für Neurologie, Universitätsklinikum Köln, Kerpener Straße 62, 50937, Köln, Deutschland.
Zentrum für Seltene Erkrankungen, Universitätsklinikum Köln, Köln, Deutschland.
Nervenarzt. 2019 Feb;90(2):148-159. doi: 10.1007/s00115-018-0562-9.
The congenital myasthenic syndromes (CMS) represent a heterogeneous group of diseases with a broad spectrum of phenotypes. The common characteristic is an inherited genetic defect of the neuromuscular junction. Although in some patients the specific gene defect remains to be detected, the increasing identification of causative genes in recent years has already provided unique insights into the functionality of structural proteins at the neuromuscular junction. Neonatal and early childhood onset is observed in most CMS subtypes; however, late onset in adolescence or adulthood also occurs and establishing the diagnosis at these stages imposes particular challenges. To enable appropriate therapeutic interventions for an at least in principle treatable condition, determining the genetic cause is warranted. In this overview, the critical clinical and diagnostic features of the different CMS subtypes are presented and illustrated using typical cases. Furthermore, specific diagnostic clues are outlined. Finally, the overlap between CMS and muscular dystrophies is discussed. Illustrating characteristic patient examples, the essential clinical and additional diagnostic findings of various CMS subtypes and special diagnostic indications are presented.
先天性肌无力综合征(CMS)是一组具有广泛表型的异质性疾病。其共同特征是神经肌肉接头处存在遗传性基因缺陷。尽管在一些患者中仍有待检测出特定的基因缺陷,但近年来致病基因的鉴定不断增加,这已经为神经肌肉接头处结构蛋白的功能提供了独特的见解。大多数CMS亚型在新生儿期和儿童早期发病;然而,青春期或成年期的迟发性发病也会出现,在这些阶段进行诊断会带来特殊挑战。为了针对至少在原则上可治疗的疾病进行适当的治疗干预,确定遗传病因是必要的。在本综述中,将介绍不同CMS亚型的关键临床和诊断特征,并通过典型病例进行说明。此外,还概述了具体的诊断线索。最后,讨论了CMS与肌肉营养不良症之间的重叠。通过举例说明特征性患者,介绍了各种CMS亚型的基本临床和其他诊断结果以及特殊诊断指征。