Niewiadomska-Cimicka Anna, Hache Antoine, Trottier Yvon
Institut de Génétique et de Biologie Moléculaire et Cellulaire (IGBMC), Illkirch, France.
Centre National de la Recherche Scientifique, UMR7104, Illkirch, France.
Front Neurosci. 2020 Jun 9;14:571. doi: 10.3389/fnins.2020.00571. eCollection 2020.
Polyglutamine spinocerebellar ataxias (polyQ SCAs) include SCA1, SCA2, SCA3, SCA6, SCA7, and SCA17 and constitute a group of adult onset neurodegenerative disorders caused by the expansion of a CAG repeat sequence located within the coding region of specific genes, which translates into polyglutamine tract in the corresponding proteins. PolyQ SCAs are characterized by degeneration of the cerebellum and its associated structures and lead to progressive ataxia and other diverse symptoms. In recent years, gene and epigenetic deregulations have been shown to play a critical role in the pathogenesis of polyQ SCAs. Here, we provide an overview of the functions of wild type and pathogenic polyQ SCA proteins in gene regulation, describe the extent and nature of gene expression changes and their pathological consequences in diseases, and discuss potential avenues to further investigate converging and distinct disease pathways and to develop therapeutic strategies.
多聚谷氨酰胺脊髓小脑共济失调(polyQ SCAs)包括SCA1、SCA2、SCA3、SCA6、SCA7和SCA17,是一组成年期发病的神经退行性疾病,由特定基因编码区内CAG重复序列的扩增引起,该序列在相应蛋白质中翻译为多聚谷氨酰胺链。polyQ SCAs的特征是小脑及其相关结构的退化,并导致进行性共济失调和其他各种症状。近年来,基因和表观遗传失调已被证明在polyQ SCAs的发病机制中起关键作用。在此,我们概述野生型和致病性polyQ SCA蛋白在基因调控中的功能,描述疾病中基因表达变化的程度和性质及其病理后果,并讨论进一步研究共同和不同疾病途径以及制定治疗策略的潜在途径。