Division of Neurosurgery, Neuro-Medical Scientific Center, Hualien Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation, Hualien 970, Taiwan.
School of Medicine, Tzu Chi University, Hualien 970, Taiwan.
Int J Mol Sci. 2023 Jan 19;24(3):2017. doi: 10.3390/ijms24032017.
Mutations in parkin, a neuroprotective protein, are the predominant cause of autosomal recessive juvenile Parkinson's disease. Neuroinflammation-derived nitrosative stress has been implicated in the etiology of the chronic neurodegeneration. However, the interactions between genetic predisposition and nitrosative stress contributing to the degeneration of dopaminergic (DA) neurons remain incompletely understood. Here, we used the SH-SY5Y neuroblastoma cells to investigate the function of parkin and its pathogenic mutants in relation to cell survival under nitric oxide (NO) exposure. The results showed that overexpression of wild-type parkin protected SH-SY5Y cells from NO-induced apoptosis in a reactive oxygen species-dependent manner. Under nitrosative stress conditions, parkin selectively upregulated the inositol-requiring enzyme 1α/X-box binding protein 1 (IRE1α/XBP1) signaling axis, an unfolded protein response signal through the sensor IRE1α, which controls the splicing of XBP1 mRNA. Inhibition of XBP1 mRNA splicing either by pharmacologically inhibiting IRE1α endoribonuclease activity or by genetically knocking down XBP1 interfered with the protective activity of parkin. Furthermore, pathogenic parkin mutants with a defective protective capacity showed a lower ability to activate the IRE1α/XBP1 signaling. Finally, we demonstrated that IRE1α activity augmented by parkin was possibly mediated through interacting with IRE1α to regulate its phosphorylation/oligomerization processes, whereas mutant parkin diminished its binding to and activation of IRE1α. Thus, these results support a direct link between the protective activity of parkin and the IRE1α/XBP1 pathway in response to nitrosative stress, and mutant parkin disrupts this function.
帕金森病是一种常见的神经退行性疾病,其主要的致病原因是抑癌基因 parkin 的突变。神经炎症引起的硝化应激在慢性神经退行性变的发病机制中起重要作用。然而,遗传易感性与硝化应激之间的相互作用如何导致多巴胺能(DA)神经元变性仍不完全清楚。在这里,我们使用 SH-SY5Y 神经母细胞瘤细胞来研究 parkin 及其致病变异体在一氧化氮(NO)暴露下与细胞存活的关系。结果表明,野生型 parkin 的过表达以依赖活性氧的方式保护 SH-SY5Y 细胞免受 NO 诱导的细胞凋亡。在硝化应激条件下,parkin 选择性地上调了未折叠蛋白反应信号通路的肌醇需求酶 1α/X 盒结合蛋白 1(IRE1α/XBP1),这是一种通过传感器 IRE1α 控制 XBP1 mRNA 剪接的信号。通过药理学抑制 IRE1α 内切核酸酶活性或基因敲低 XBP1 抑制 XBP1 mRNA 剪接,会干扰 parkin 的保护活性。此外,具有缺陷保护能力的致病变异体 parkin 显示出激活 IRE1α/XBP1 信号的能力较低。最后,我们证明 parkin 增强的 IRE1α 活性可能是通过与 IRE1α 相互作用来调节其磷酸化/寡聚化过程而介导的,而突变型 parkin 则减弱了其与 IRE1α 的结合和激活。因此,这些结果支持了 parkin 的保护活性与硝化应激反应中的 IRE1α/XBP1 通路之间的直接联系,并且突变型 parkin 破坏了这种功能。