线粒体氧化应激促进糖脑苷酶突变小鼠α-突触核蛋白聚集和扩散

IF 6.7 1区 医学 Q1 NEUROSCIENCES NPJ Parkinson's Disease Pub Date : 2024-12-11 DOI:10.1038/s41531-024-00842-8
Pietro La Vitola, Eva M. Szegö, Rita Pinto-Costa, Angela Rollar, Eugenia Harbachova, Anthony HV Schapira, Ayse Ulusoy, Donato A. Di Monte
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摘要

在这项研究中,发现一种常见的帕金森相关GBA1变异L444P突变的杂合表达,在一个针对髓系迷走神经系统神经元的帕金森样病理小鼠模型中,加剧了α-突触核蛋白的聚集和扩散。这些神经元在表达L444P后也更容易受到氧化应激和硝化应激的影响。后者与活性氧的神经元形成平行,并导致硝化α-突触核蛋白的显著积累。突变诱导的氧化/硝化应激与α-突触核蛋白聚集和扩散增强之间的因果关系确实可以通过线粒体超氧化物歧化酶2的神经元过表达来挽救。进一步的证据支持线粒体作为活性氧的来源以及表达l444p的神经元中氧化和硝化损伤的靶标的关键参与。这些发现支持这样的结论,即线粒体氧化应激易感性的增强应被认为是GBA1突变携带者易发生病理转化的重要机制。
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Mitochondrial oxidant stress promotes α-synuclein aggregation and spreading in mice with mutated glucocerebrosidase

In this study, heterozygous expression of a common Parkinson-associated GBA1 variant, the L444P mutation, was found to exacerbate α-synuclein aggregation and spreading in a mouse model of Parkinson-like pathology targeting neurons of the medullary vagal system. These neurons were also shown to become more vulnerable to oxidative and nitrative stress after L444P expression. The latter paralleled neuronal formation of reactive oxygen species and led to a pronounced accumulation of nitrated α-synuclein. A causal relationship linked mutation-induced oxidative/nitrative stress to enhanced α-synuclein aggregation and spreading that could indeed be rescued by neuronal overexpression of mitochondrial superoxide dismutase 2. Further evidence supported a key involvement of mitochondria as sources of reactive oxygen species as well as targets of oxidative and nitrative damage within L444P-expressing neurons. These findings support the conclusion that enhanced vulnerability to mitochondrial oxidative stress should be considered an important mechanism predisposing to pathology conversion in carriers of GBA1 mutations.

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来源期刊
NPJ Parkinson's Disease
NPJ Parkinson's Disease Medicine-Neurology (clinical)
CiteScore
9.80
自引率
5.70%
发文量
156
审稿时长
11 weeks
期刊介绍: npj Parkinson's Disease is a comprehensive open access journal that covers a wide range of research areas related to Parkinson's disease. It publishes original studies in basic science, translational research, and clinical investigations. The journal is dedicated to advancing our understanding of Parkinson's disease by exploring various aspects such as anatomy, etiology, genetics, cellular and molecular physiology, neurophysiology, epidemiology, and therapeutic development. By providing free and immediate access to the scientific and Parkinson's disease community, npj Parkinson's Disease promotes collaboration and knowledge sharing among researchers and healthcare professionals.
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