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非特异性致死复合物调节与帕金森病遗传相关的基因和通路。

The non-specific lethal complex regulates genes and pathways genetically linked to Parkinson's disease.

机构信息

Department of Neurodegenerative Disease, UCL Queen Square Institute of Neurology, London WC1N 3BG, UK.

Aligning Science Across Parkinson's (ASAP) Collaborative Research Network, Chevy Chase, MD 20815, USA.

出版信息

Brain. 2023 Dec 1;146(12):4974-4987. doi: 10.1093/brain/awad246.

Abstract

Genetic variants conferring risks for Parkinson's disease have been highlighted through genome-wide association studies, yet exploration of their specific disease mechanisms is lacking. Two Parkinson's disease candidate genes, KAT8 and KANSL1, identified through genome-wide studies and a PINK1-mitophagy screen, encode part of the histone acetylating non-specific lethal complex. This complex localizes to the nucleus, where it plays a role in transcriptional activation, and to mitochondria, where it has been suggested to have a role in mitochondrial transcription. In this study, we sought to identify whether the non-specific lethal complex has potential regulatory relationships with other genes associated with Parkinson's disease in human brain. Correlation in the expression of non-specific lethal genes and Parkinson's disease-associated genes was investigated in primary gene co-expression networks using publicly-available transcriptomic data from multiple brain regions (provided by the Genotype-Tissue Expression Consortium and UK Brain Expression Consortium), whilst secondary networks were used to examine cell type specificity. Reverse engineering of gene regulatory networks generated regulons of the complex, which were tested for heritability using stratified linkage disequilibrium score regression. Prioritized gene targets were then validated in vitro using a QuantiGene multiplex assay and publicly-available chromatin immunoprecipitation-sequencing data. Significant clustering of non-specific lethal genes was revealed alongside Parkinson's disease-associated genes in frontal cortex primary co-expression modules, amongst other brain regions. Both primary and secondary co-expression modules containing these genes were enriched for mainly neuronal cell types. Regulons of the complex contained Parkinson's disease-associated genes and were enriched for biological pathways genetically linked to disease. When examined in a neuroblastoma cell line, 41% of prioritized gene targets showed significant changes in mRNA expression following KANSL1 or KAT8 perturbation. KANSL1 and H4K8 chromatin immunoprecipitation-sequencing data demonstrated non-specific lethal complex activity at many of these genes. In conclusion, genes encoding the non-specific lethal complex are highly correlated with and regulate genes associated with Parkinson's disease. Overall, these findings reveal a potentially wider role for this protein complex in regulating genes and pathways implicated in Parkinson's disease.

摘要

通过全基因组关联研究,已经发现了一些与帕金森病相关的遗传变异体,但对其具体的疾病机制的研究还很缺乏。通过全基因组研究和 PINK1-线粒体自噬筛选发现的两个帕金森病候选基因 KAT8 和 KANSL1,编码组蛋白乙酰转移酶非特异性致死复合物的一部分。该复合物定位于细胞核,在那里它在转录激活中发挥作用,也定位于线粒体,在那里它被认为在线粒体转录中发挥作用。在这项研究中,我们试图确定非特异性致死复合物是否与人类大脑中与帕金森病相关的其他基因具有潜在的调节关系。使用来自多个大脑区域的公共转录组数据(由基因型-组织表达联盟和英国大脑表达联盟提供),在初级基因共表达网络中研究非特异性致死基因和帕金森病相关基因的表达相关性,同时使用二级网络研究细胞类型特异性。使用基因调控网络的反向工程生成复合物的调控网络,使用分层连锁不平衡评分回归测试其遗传力。然后使用 QuantiGene 多重测定法和公开的染色质免疫沉淀测序数据在体外验证优先基因靶标。在额皮质初级共表达模块中,与帕金森病相关的基因与非特异性致死基因一起显著聚类,而在其他大脑区域也有类似的情况。包含这些基因的初级和二级共表达模块都富含主要的神经元细胞类型。复合物的调控网络包含帕金森病相关基因,并富集了与疾病相关的生物途径。在神经母细胞瘤细胞系中进行检查时,在 KANSL1 或 KAT8 扰动后,41%的优先基因靶标显示 mRNA 表达发生显著变化。KANSL1 和 H4K8 染色质免疫沉淀测序数据表明,在许多这些基因中,非特异性致死复合物具有活性。总之,编码非特异性致死复合物的基因与帕金森病相关基因高度相关,并调节这些基因。总的来说,这些发现揭示了这个蛋白质复合物在调节与帕金森病相关的基因和途径方面可能具有更广泛的作用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a3b8/10689904/13e2ee66453e/awad246f1.jpg

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