Giovannelli Ilaria, Higginbottom Adrian, Kirby Janine, Azzouz Mimoun, Shaw Pamela J
Sheffield Institute for Translational Neuroscience (SITraN), University of Sheffield, Sheffield, UK.
Nat Rev Neurol. 2023 Jan;19(1):39-52. doi: 10.1038/s41582-022-00751-5. Epub 2022 Dec 8.
Amyotrophic lateral sclerosis (ALS) is a devastating and incurable neurodegenerative disease characterized by the progressive loss of upper and lower motor neurons. ALS causes death, usually within 2-5 years of diagnosis. Riluzole, the only drug currently approved in Europe for the treatment of this condition, offers only a modest benefit, increasing survival by 3 months on average. Recent advances in our understanding of causative or disease-modifying genetic variants and in the development of genetic therapy strategies present exciting new therapeutic opportunities for ALS. In addition, the approval of adeno-associated virus-mediated delivery of functional copies of the SMN1 gene to treat spinal muscular atrophy represents an important therapeutic milestone and demonstrates the potential of gene replacement therapies for motor neuron disorders. In this Review, we describe the current landscape of genetic therapies in ALS, highlighting achievements and critical challenges. In particular, we discuss opportunities for gene replacement therapy in subgroups of people with ALS, and we describe loss-of-function mutations that are known to contribute to the pathophysiology of ALS and could represent novel targets for gene replacement therapies.
肌萎缩侧索硬化症(ALS)是一种毁灭性的、无法治愈的神经退行性疾病,其特征是上下运动神经元逐渐丧失。ALS会导致死亡,通常在确诊后的2至5年内。利鲁唑是目前欧洲唯一被批准用于治疗这种疾病的药物,其疗效有限,平均仅能延长3个月的生存期。我们对致病或疾病修饰基因变异的理解以及基因治疗策略的发展取得了最新进展,为ALS带来了令人兴奋的新治疗机会。此外,腺相关病毒介导的SMN1基因功能拷贝递送用于治疗脊髓性肌萎缩症的获批是一个重要的治疗里程碑,证明了基因替代疗法对运动神经元疾病的潜力。在本综述中,我们描述了ALS基因治疗的现状,突出了成就和关键挑战。特别是,我们讨论了ALS患者亚组中基因替代疗法的机会,并描述了已知导致ALS病理生理学且可能成为基因替代疗法新靶点的功能丧失突变。