Department of Psychiatry and Human Behavior, Department of Neurobiology and Behavior, Department of Biological Chemistry, Institute of Memory Impairments and Neurological Disorders, Sue and Bill Gross Stem Cell Center, University of California Irvine, Irvine, CA 92697, USA.
Department of Laboratory Medicine and Pathology, Institute for Translational Neuroscience, University of Minnesota, Minneapolis and Saint Paul, MN 55455, USA.
Neuron. 2023 Nov 15;111(22):3517-3530. doi: 10.1016/j.neuron.2023.09.036. Epub 2023 Oct 19.
One of the more transformative findings in human genetics was the discovery that the expansion of unstable nucleotide repeats underlies a group of inherited neurological diseases. A subset of these unstable repeat neurodegenerative diseases is due to the expansion of a CAG trinucleotide repeat encoding a stretch of glutamines, i.e., the polyglutamine (polyQ) repeat neurodegenerative diseases. Among the CAG/polyQ repeat diseases are Huntington's disease (HD) and spinocerebellar ataxia type 1 (SCA1), in which the expansions are within widely expressed proteins. Although both HD and SCA1 are autosomal dominantly inherited, and both typically cause mid- to late-life-onset movement disorders with cognitive decline, they each are characterized by distinct clinical characteristics and predominant sites of neuropathology. Importantly, the respective affected proteins, Huntingtin (HTT, HD) and Ataxin 1 (ATXN1, SCA1), have unique functions and biological properties. Here, we review HD and SCA1 with a focus on how their disease-specific and shared features may provide informative insights.
在人类遗传学中,最具变革性的发现之一是发现不稳定核苷酸重复的扩展是一组遗传性神经疾病的基础。这些不稳定重复神经退行性疾病的一部分是由于编码谷氨酸的 CAG 三核苷酸重复的扩展,即聚谷氨酰胺(polyQ)重复神经退行性疾病。在 CAG/polyQ 重复疾病中,有亨廷顿病(HD)和脊髓小脑共济失调 1 型(SCA1),其中扩展存在于广泛表达的蛋白质中。尽管 HD 和 SCA1 都是常染色体显性遗传的,并且通常都会导致中年至晚年发病的运动障碍伴认知能力下降,但它们各自具有独特的临床特征和主要的神经病理学部位。重要的是,各自受影响的蛋白质,亨廷顿蛋白(HTT,HD)和 Ataxin 1(ATXN1,SCA1),具有独特的功能和生物学特性。在这里,我们重点回顾 HD 和 SCA1,探讨它们的疾病特异性和共同特征如何提供有价值的见解。