Lunn Mitchell R, Wang Ching H
Department of Medicine, Stanford University School of Medicine, Stanford, CA, USA.
Lancet. 2008 Jun 21;371(9630):2120-33. doi: 10.1016/S0140-6736(08)60921-6.
Spinal muscular atrophy is an autosomal recessive neurodegenerative disease characterised by degeneration of spinal cord motor neurons, atrophy of skeletal muscles, and generalised weakness. It is caused by homozygous disruption of the survival motor neuron 1 (SMN1) gene by deletion, conversion, or mutation. Although no medical treatment is available, investigations have elucidated possible mechanisms underlying the molecular pathogenesis of the disease. Treatment strategies have been developed to use the unique genomic structure of the SMN1 gene region. Several candidate treatment agents have been identified and are in various stages of development. These and other advances in medical technology have changed the standard of care for patients with spinal muscular atrophy. In this Seminar, we provide a comprehensive review that integrates clinical manifestations, molecular pathogenesis, diagnostic strategy, therapeutic development, and evidence from clinical trials.
脊髓性肌萎缩症是一种常染色体隐性神经退行性疾病,其特征为脊髓运动神经元变性、骨骼肌萎缩和全身无力。它是由生存运动神经元1(SMN1)基因通过缺失、转换或突变导致的纯合性破坏引起的。尽管尚无医学治疗方法,但研究已阐明了该疾病分子发病机制的潜在可能机制。已制定治疗策略以利用SMN1基因区域独特的基因组结构。已鉴定出几种候选治疗药物,且它们正处于不同的研发阶段。这些以及医学技术的其他进展已经改变了脊髓性肌萎缩症患者的护理标准。在本次研讨会上,我们进行了全面综述,整合了临床表现、分子发病机制、诊断策略、治疗研发以及临床试验证据。