Shi Tianyu, Zhou Zijie, Xiang Taiyang, Suo Yinxuan, Shi Xiaoyan, Li Yaoyao, Zhang Peng, Dai Jun, Sheng Lei
Department of Orthopedics, The Second Affiliated Hospital of Soochow University, No. 1055, Sanxiang Road, Suzhou, 215004, Jiangsu, China.
Department of Neurology, Children's Hospital of Soochow University, Suzhou, 215025, Jiangsu, China.
J Neurol. 2024 Dec 12;272(1):19. doi: 10.1007/s00415-024-12724-3.
Spinal muscular atrophy (SMA) is a neurodegenerative disease caused by deletions or mutations of survival of motor neuron 1 (SMN1) gene. To date, the mechanism of selective cell death of motor neurons as a hallmark of SMA is still unclear. The severity of SMA is dependent on the amount of survival motor neuron (SMN) protein, which is an essential and ubiquitously expressed protein involved in various cellular processes including regulation of cytoskeletal dynamics. In this review, we discuss the effect of SMN ablation on cytoskeleton organization including actin dynamics, growth cone formation, axonal stability, neurite outgrowth, microtubule stability, synaptic vesicle dynamics and neurofilament protein release in SMA. We also summarized a list of critical proteins such as profilin-2 (PFN2), plastin-3 (PLS3), stathmin-1 (STMN1), microtubule-associated protein 1B (MAP1B) and neurofilament which play an important role in modulating cytoskeleton in SMA. Our aim is to highlight how cytoskeletal defects contribute to motor neuron degeneration in SMA disease progression and concentrating on cytoskeleton dynamics may be a promising approach to develop new therapy or biomarker.
脊髓性肌萎缩症(SMA)是一种由运动神经元存活1(SMN1)基因缺失或突变引起的神经退行性疾病。迄今为止,作为SMA标志的运动神经元选择性细胞死亡机制仍不清楚。SMA的严重程度取决于存活运动神经元(SMN)蛋白的量,SMN蛋白是一种必需且广泛表达的蛋白,参与包括细胞骨架动力学调节在内的各种细胞过程。在本综述中,我们讨论了SMN缺失对SMA中细胞骨架组织的影响,包括肌动蛋白动力学、生长锥形成、轴突稳定性、神经突生长、微管稳定性、突触小泡动力学和神经丝蛋白释放。我们还总结了一系列关键蛋白,如丝切蛋白-2(PFN2)、质体素-3(PLS3)、微管相关蛋白1B(MAP1B)和神经丝,它们在调节SMA细胞骨架中起重要作用。我们的目的是强调细胞骨架缺陷如何在SMA疾病进展中导致运动神经元变性,专注于细胞骨架动力学可能是开发新疗法或生物标志物的一种有前景的方法。