Nogajski Joseph H, Kiernan Matthew C, Ouvrier Robert A, Andrews P Ian
Institute of Neurological Sciences, Prince of Wales Hospital, Randwick, New South Wales, Australia.
J Clin Neurosci. 2009 Jan;16(1):1-11. doi: 10.1016/j.jocn.2008.05.001. Epub 2008 Nov 18.
Congenital myasthenic syndromes (CMS) are a heterogeneous group of uncommon, inherited disorders affecting the neuromuscular junction. The defects interfere with presynaptic, synaptic, or postsynaptic function and compromise neuromuscular transmission. Most patients with CMS have similar clinical features regardless of the underlying defect, but attention to clinical and electrodiagnostic parameters can narrow the diagnostic spectrum. Recent advances in our understanding of the cellular mechanisms underlying specific syndromes allow DNA testing for some forms of CMS. Diagnosis of CMS enables a rationale for management to be developed. Two cases of genetically determined CMS as well as an undiagnosed infant are presented to highlight the clinical and electrophysiological difficulties associated with the diagnosis and management of such syndromes.
先天性肌无力综合征(CMS)是一组影响神经肌肉接头的罕见遗传性疾病,具有异质性。这些缺陷会干扰突触前、突触或突触后功能,损害神经肌肉传递。大多数CMS患者,无论潜在缺陷如何,都有相似的临床特征,但关注临床和电诊断参数可缩小诊断范围。我们对特定综合征潜在细胞机制认识的最新进展使得对某些形式的CMS能够进行DNA检测。CMS的诊断有助于制定合理的治疗方案。本文介绍了两例基因确诊的CMS病例以及一名未确诊的婴儿,以突出此类综合征在诊断和治疗方面所面临的临床和电生理难题。