Kleinknecht Alexandra, Popova Blagovesta, Lázaro Diana F, Pinho Raquel, Valerius Oliver, Outeiro Tiago F, Braus Gerhard H
Department of Molecular Microbiology and Genetics and Göttingen Center for Molecular Biosciences (GZMB), Institute of Microbiology and Genetics, Georg-August-Universität, Göttingen, Germany.
Center for Nanoscale Microscopy and Molecular Physiology of the Brain (CNMPB), Göttingen, Germany.
PLoS Genet. 2016 Jun 24;12(6):e1006098. doi: 10.1371/journal.pgen.1006098. eCollection 2016 Jun.
Parkinson´s disease (PD) is characterized by the presence of proteinaceous inclusions called Lewy bodies that are mainly composed of α-synuclein (αSyn). Elevated levels of oxidative or nitrative stresses have been implicated in αSyn related toxicity. Phosphorylation of αSyn on serine 129 (S129) modulates autophagic clearance of inclusions and is prominently found in Lewy bodies. The neighboring tyrosine residues Y125, Y133 and Y136 are phosphorylation and nitration sites. Using a yeast model of PD, we found that Y133 is required for protective S129 phosphorylation and for S129-independent proteasome clearance. αSyn can be nitrated and form stable covalent dimers originating from covalent crosslinking of two tyrosine residues. Nitrated tyrosine residues, but not di-tyrosine-crosslinked dimers, contributed to αSyn cytotoxicity and aggregation. Analysis of tyrosine residues involved in nitration and crosslinking revealed that the C-terminus, rather than the N-terminus of αSyn, is modified by nitration and di-tyrosine formation. The nitration level of wild-type αSyn was higher compared to that of A30P mutant that is non-toxic in yeast. A30P formed more dimers than wild-type αSyn, suggesting that dimer formation represents a cellular detoxification pathway in yeast. Deletion of the yeast flavohemoglobin gene YHB1 resulted in an increase of cellular nitrative stress and cytotoxicity leading to enhanced aggregation of A30P αSyn. Yhb1 protected yeast from A30P-induced mitochondrial fragmentation and peroxynitrite-induced nitrative stress. Strikingly, overexpression of neuroglobin, the human homolog of YHB1, protected against αSyn inclusion formation in mammalian cells. In total, our data suggest that C-terminal Y133 plays a major role in αSyn aggregate clearance by supporting the protective S129 phosphorylation for autophagy and by promoting proteasome clearance. C-terminal tyrosine nitration increases pathogenicity and can only be partially detoxified by αSyn di-tyrosine dimers. Our findings uncover a complex interplay between S129 phosphorylation and C-terminal tyrosine modifications of αSyn that likely participates in PD pathology.
帕金森病(PD)的特征是存在称为路易小体的蛋白质内含物,其主要由α-突触核蛋白(αSyn)组成。氧化应激或硝化应激水平升高与αSyn相关毒性有关。αSyn丝氨酸129(S129)位点的磷酸化调节内含物的自噬清除,且在路易小体中显著存在。相邻的酪氨酸残基Y125、Y133和Y136是磷酸化和硝化位点。利用PD酵母模型,我们发现Y133对于保护性S129磷酸化以及不依赖S129的蛋白酶体清除是必需的。αSyn可被硝化并形成稳定的共价二聚体,其源于两个酪氨酸残基的共价交联。硝化的酪氨酸残基而非二酪氨酸交联的二聚体导致αSyn细胞毒性和聚集。对参与硝化和交联的酪氨酸残基的分析表明,αSyn的C末端而非N末端被硝化和形成二酪氨酸修饰。与在酵母中无毒的A30P突变体相比,野生型αSyn的硝化水平更高。A30P形成的二聚体比野生型αSyn更多,这表明二聚体形成代表酵母中的一种细胞解毒途径。酵母黄素血红蛋白基因YHB1的缺失导致细胞硝化应激和细胞毒性增加,从而导致A30P αSyn聚集增强。Yhb1保护酵母免受A30P诱导的线粒体碎片化和过氧亚硝酸盐诱导的硝化应激。引人注目的是,YHB1的人类同源物神经球蛋白的过表达可防止哺乳动物细胞中αSyn内含物的形成。总之,我们的数据表明,C末端的Y133通过支持自噬的保护性S129磷酸化和促进蛋白酶体清除,在αSyn聚集体清除中起主要作用。C末端酪氨酸硝化增加致病性,并且只能被αSyn二酪氨酸二聚体部分解毒。我们的发现揭示了αSyn的S129磷酸化和C末端酪氨酸修饰之间复杂的相互作用,这可能参与了PD病理学过程。