Picchiarelli Gina, Dupuis Luc
Université de Strasbourg, INSERM, Mécanismes Centraux et Périphériques de la Neurodégénérescence, UMR_S 1118, Strasbourg, France.
Cell Stress. 2020 Mar 10;4(4):76-91. doi: 10.15698/cst2020.04.217.
A number of neuromuscular and muscular diseases, including amyotrophic lateral sclerosis (ALS), spinal muscular atrophy (SMA) and several myopathies, are associated to mutations in related RNA-binding proteins (RBPs), including TDP-43, FUS, MATR3 or hnRNPA1/B2. These proteins harbor similar modular primary sequence with RNA binding motifs and low complexity domains, that enables them to phase separate and create liquid microdomains. These RBPs have been shown to critically regulate multiple events of RNA lifecycle, including transcriptional events, splicing and RNA trafficking and sequestration. Here, we review the roles of these disease-related RBPs in muscle and motor neurons, and how their dysfunction in these cell types might contribute to disease.
许多神经肌肉疾病和肌肉疾病,包括肌萎缩侧索硬化症(ALS)、脊髓性肌萎缩症(SMA)和几种肌病,都与相关RNA结合蛋白(RBP)的突变有关,这些蛋白包括TDP-43、FUS、MATR3或hnRNPA1/B2。这些蛋白质具有相似的模块化一级序列,带有RNA结合基序和低复杂性结构域,这使它们能够发生相分离并形成液体微结构域。这些RBP已被证明对RNA生命周期的多个事件起着关键调节作用,包括转录事件、剪接以及RNA运输和隔离。在这里,我们综述了这些与疾病相关的RBP在肌肉和运动神经元中的作用,以及它们在这些细胞类型中的功能障碍如何可能导致疾病。