Edinburgh Medical School: Biomedical Sciences, University of Edinburgh, Edinburgh, UK.
Euan MacDonald Centre for Motor Neuron Disease Research, University of Edinburgh, Edinburgh, UK.
Nat Rev Neurol. 2018 Apr;14(4):214-224. doi: 10.1038/nrneurol.2018.4. Epub 2018 Feb 9.
Spinal muscular atrophy (SMA) is a devastating motor neuron disease that predominantly affects children and represents the most common cause of hereditary infant mortality. The condition results from deleterious variants in SMN1, which lead to depletion of the survival motor neuron protein (SMN). Now, 20 years after the discovery of this genetic defect, a major milestone in SMA and motor neuron disease research has been reached with the approval of the first disease-modifying therapy for SMA by US and European authorities - the antisense oligonucleotide nusinersen. At the same time, promising data from early-stage clinical trials of SMN1 gene therapy have indicated that additional therapeutic options are likely to emerge for patients with SMA in the near future. However, the approval of nusinersen has generated a number of immediate and substantial medical, ethical and financial implications that have the potential to resonate beyond the specific treatment of SMA. Here, we provide an overview of the rapidly evolving therapeutic landscape for SMA, highlighting current achievements and future opportunities. We also discuss how these developments are providing important lessons for the emerging second generation of combinatorial ('SMN-plus') therapies that are likely to be required to generate robust treatments that are effective across a patient's lifespan.
脊髓性肌萎缩症(SMA)是一种破坏性的运动神经元疾病,主要影响儿童,是遗传性婴儿死亡的最常见原因。这种疾病是由 SMN1 中的有害变异引起的,导致运动神经元存活蛋白(SMN)耗竭。如今,在发现这种遗传缺陷 20 年后,美国和欧洲当局批准了第一种用于治疗 SMA 的疾病修正疗法——反义寡核苷酸 nusinersen,这是 SMA 和运动神经元疾病研究的一个重要里程碑。与此同时,SMN1 基因治疗早期临床试验的有希望数据表明,SMA 患者可能很快会出现更多的治疗选择。然而,nusinersen 的批准带来了许多直接的、实质性的医学、伦理和财务影响,这些影响有可能超越 SMA 的具体治疗产生共鸣。在这里,我们提供了 SMA 治疗领域的快速发展概述,重点介绍了当前的成就和未来的机会。我们还讨论了这些发展如何为新兴的第二代组合(“SMN-plus”)疗法提供重要的经验教训,这些疗法可能需要生成有效的治疗方法,以在患者的整个生命周期内发挥作用。