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亨廷顿病中的线粒体异常与突触损伤:聚焦于有缺陷的线粒体自噬及线粒体靶向治疗

Mitochondrial Abnormalities and Synaptic Damage in Huntington's Disease: a Focus on Defective Mitophagy and Mitochondria-Targeted Therapeutics.

作者信息

Sawant Neha, Morton Hallie, Kshirsagar Sudhir, Reddy Arubala P, Reddy P Hemachandra

机构信息

Department of Internal Medicine, Texas Tech University Health Sciences Center, Lubbock, TX, USA.

Nutritional Sciences Department, College of Human Sciences, Texas Tech University, 1301 Akron Ave, Lubbock, TX, USA.

出版信息

Mol Neurobiol. 2021 Dec;58(12):6350-6377. doi: 10.1007/s12035-021-02556-x. Epub 2021 Sep 14.

Abstract

Huntington's disease (HD) is a fatal and pure genetic disease with a progressive loss of medium spiny neurons (MSN). HD is caused by expanded polyglutamine repeats in the exon 1 of HD gene. Clinically, HD is characterized by chorea, seizures, involuntary movements, dystonia, cognitive decline, intellectual impairment, and emotional disturbances. Several years of intense research revealed that multiple cellular changes, including defective axonal transport, protein-protein interactions, defective bioenergetics, calcium dyshomeostasis, NMDAR activation, synaptic damage, mitochondrial abnormalities, and selective loss of medium spiny neurons are implicated in HD. Recent research on mutant huntingtin (mHtt) and mitochondria has found that mHtt interacts with the mitochondrial division protein, dynamin-related protein 1 (DRP1), enhances GTPase DRP1 enzymatic activity, and causes excessive mitochondrial fragmentation and abnormal distribution, leading to defective axonal transport of mitochondria and selective synaptic degeneration. Recent research also revealed that failure to remove dead and/or dying mitochondria is an early event in the disease progression. Currently, efforts are being made to reduce abnormal protein interactions and enhance synaptic mitophagy as therapeutic strategies for HD. The purpose of this article is to discuss recent research in HD progression. This article also discusses recent developments of cell and mouse models, cellular changes, mitochondrial abnormalities, DNA damage, bioenergetics, oxidative stress, mitophagy, and therapeutics strategies in HD.

摘要

亨廷顿舞蹈病(HD)是一种致命的纯基因疾病,会导致中等棘状神经元(MSN)进行性丧失。HD是由HD基因外显子1中多聚谷氨酰胺重复序列扩增引起的。临床上,HD的特征为舞蹈症、癫痫发作、不自主运动、肌张力障碍、认知衰退、智力损害和情绪障碍。数年的深入研究表明,多种细胞变化与HD有关,包括轴突运输缺陷、蛋白质-蛋白质相互作用、生物能量学缺陷、钙稳态失调、NMDAR激活、突触损伤、线粒体异常以及中等棘状神经元的选择性丧失。最近对突变型亨廷顿蛋白(mHtt)和线粒体的研究发现,mHtt与线粒体分裂蛋白动力相关蛋白1(DRP1)相互作用,增强GTP酶DRP1的酶活性,并导致线粒体过度碎片化和异常分布,从而导致线粒体轴突运输缺陷和选择性突触退化。最近的研究还表明,无法清除死亡和/或濒死的线粒体是疾病进展中的早期事件。目前,人们正在努力减少异常的蛋白质相互作用并增强突触线粒体自噬,将其作为HD的治疗策略。本文的目的是讨论HD进展方面的最新研究。本文还讨论了HD中细胞和小鼠模型、细胞变化、线粒体异常、DNA损伤、生物能量学、氧化应激、线粒体自噬以及治疗策略的最新进展。

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