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继发性线粒体功能障碍作为溶酶体贮积病神经退行性功能障碍的病因,以及潜在治疗方法概述。

Secondary Mitochondrial Dysfunction as a Cause of Neurodegenerative Dysfunction in Lysosomal Storage Diseases and an Overview of Potential Therapies.

机构信息

Adult Inherited Metabolic Disorders, Salford Royal NHS Foundation Trust, Salford M6 8HD, UK.

School of Pharmacy and Biomolecular Sciences, Liverpool John Moores University, Liverpool L3 3AF, UK.

出版信息

Int J Mol Sci. 2022 Sep 12;23(18):10573. doi: 10.3390/ijms231810573.

Abstract

Mitochondrial dysfunction has been recognised a major contributory factor to the pathophysiology of a number of lysosomal storage disorders (LSDs). The cause of mitochondrial dysfunction in LSDs is as yet uncertain, but appears to be triggered by a number of different factors, although oxidative stress and impaired mitophagy appear to be common inhibitory mechanisms shared amongst this group of disorders, including Gaucher's disease, Niemann-Pick disease, type C, and mucopolysaccharidosis. Many LSDs resulting from defects in lysosomal hydrolase activity show neurodegeneration, which remains challenging to treat. Currently available curative therapies are not sufficient to meet patients' needs. In view of the documented evidence of mitochondrial dysfunction in the neurodegeneration of LSDs, along with the reciprocal interaction between the mitochondrion and the lysosome, novel therapeutic strategies that target the impairment in both of these organelles could be considered in the clinical management of the long-term neurodegenerative complications of these diseases. The purpose of this review is to outline the putative mechanisms that may be responsible for the reported mitochondrial dysfunction in LSDs and to discuss the new potential therapeutic developments.

摘要

线粒体功能障碍已被认为是许多溶酶体贮积症(LSD)病理生理学的主要致病因素。LSD 中线粒体功能障碍的原因尚不确定,但似乎是由许多不同的因素触发的,尽管氧化应激和受损的线粒体自噬似乎是这一组疾病共有的常见抑制机制,包括戈谢病、尼曼-匹克病 C 型和黏多糖贮积症。许多由于溶酶体水解酶活性缺陷引起的 LSD 表现出神经退行性变,这仍然是难以治疗的。目前可用的治愈疗法不足以满足患者的需求。鉴于 LSD 神经退行性变中线粒体功能障碍的已有文献证据,以及线粒体和溶酶体之间的相互作用,针对这两个细胞器损伤的新型治疗策略可以考虑用于这些疾病的长期神经退行性并发症的临床管理。本文旨在概述可能导致 LSD 中报道的线粒体功能障碍的假定机制,并讨论新的潜在治疗进展。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5abb/9503973/0b401d0a2118/ijms-23-10573-g001.jpg

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