Dipartimento Universitario di Scienze Biotecnologiche di Base, Cliniche Intensivologiche e Perioperatorie, Sezione di Biochimica e Biochimica Clinica, Università Cattolica del Sacro Cuore di Roma, Largo F. Vito 1, 00168 Roma, Italy.
Istituto di Scienze e Tecnologie Chimiche "Giulio Natta"- SCITEC Sede di Roma, Largo F. Vito 1, 00168 Roma, Italy.
Int J Mol Sci. 2020 Nov 5;21(21):8314. doi: 10.3390/ijms21218314.
Huntington's disease (HD) is an autosomal dominant neurodegenerative disorder caused by pathogenic expansions of the triplet cytosine-adenosine-guanosine (CAG) within the Huntingtin gene. These expansions lead to a prolongation of the poly-glutamine stretch at the N-terminus of Huntingtin causing protein misfolding and aggregation. Huntingtin and its pathological variants are widely expressed, but the central nervous system is mainly affected, as proved by the wide spectrum of neurological symptoms, including behavioral anomalies, cognitive decline and motor disorders. Other hallmarks of HD are loss of body weight and muscle atrophy. This review highlights some key elements that likely provide a major contribution to muscle atrophy, namely, alteration of the transcriptional processes, mitochondrial dysfunction, which is strictly correlated to loss of energy homeostasis, inflammation, apoptosis and defects in the processes responsible for the protein quality control. The improvement of muscular symptoms has proven to slow the disease progression and extend the life span of animal models of HD, underlining the importance of a deep comprehension of the molecular mechanisms driving deterioration of muscular tissue.
亨廷顿病(HD)是一种常染色体显性神经退行性疾病,由亨廷顿基因内三核苷酸胞嘧啶-腺嘌呤-鸟嘌呤(CAG)的致病扩展引起。这些扩展导致亨廷顿蛋白 N 端的多谷氨酰胺延伸延长,导致蛋白质错误折叠和聚集。亨廷顿及其病理性变体广泛表达,但主要受中枢神经系统影响,这已被广泛的神经症状所证明,包括行为异常、认知能力下降和运动障碍。HD 的其他特征还包括体重减轻和肌肉萎缩。本综述强调了一些可能对肌肉萎缩有重大贡献的关键因素,即转录过程的改变、线粒体功能障碍,这与能量稳态的丧失密切相关,炎症、细胞凋亡和负责蛋白质质量控制的过程缺陷。肌肉症状的改善已被证明可以减缓疾病的进展并延长 HD 动物模型的寿命,这强调了深入了解导致肌肉组织恶化的分子机制的重要性。