将痴呆症风险基因与脑脊液蛋白质组联系起来,可确定痴呆症的病理生理学线索。

IF 10.6 1区 医学 Q1 CLINICAL NEUROLOGY
Brain Pub Date : 2024-10-03 DOI:10.1093/brain/awae090
Lianne M Reus, Iris E Jansen, Betty M Tijms, Pieter Jelle Visser, Niccoló Tesi, Sven J van der Lee, Lisa Vermunt, Carel F W Peeters, Lisa A De Groot, Yanaika S Hok-A-Hin, Alice Chen-Plotkin, David J Irwin, William T Hu, Lieke H Meeter, John C van Swieten, Henne Holstege, Marc Hulsman, Afina W Lemstra, Yolande A L Pijnenburg, Wiesje M van der Flier, Charlotte E Teunissen, Marta Del Campo Milan
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引用次数: 0

摘要

全基因组关联研究已成功确定了许多痴呆症的遗传风险位点,但遗传风险因素导致痴呆症的确切生物学机制仍不清楚。将脑脊液蛋白质组数据与痴呆症风险位点相结合,可以揭示连接遗传变异与痴呆症发病的中间分子途径。我们在一个深度表型的混合记忆诊所队列(n=502,平均年龄(sd)=64.1[8.7]岁,181 名女性[35.4%]),包括阿尔茨海默病(AD,n=213)、路易体痴呆(DLB,n=50)和额颞叶痴呆(FTD,n=93)患者以及对照组(n=146)。在独立队列(PEA 平台 n=99,MRM 靶向质谱和多重检测 n=198)中进行了验证评估。我们还根据诊断状态(分别为 AD、DLB、FTD 和对照组)进行了分层分析,以探讨 CSF 蛋白质与遗传变异之间的关联是否针对特定疾病。我们确定了四个 AD 风险位点为蛋白质定量性状位点(pQTL):CR1-CR2(rs3818361,P=1.65e-08)、ZCWPW1-PILRB(rs1476679,P=2.73e-32)、CTSH-CTSH(rs3784539,P=2.88e-24)和 HESX1-RETN(rs186108507,P=8.39e-08),其中前三个 pQTL 在独立队列中显示出直接复制。我们在 TREM2 的一个罕见遗传变异与 CSF IL6 水平(rs75932628,P=3.90e-7)之间发现了一个 AD 特异性关联。仅在 DLB 患者中,DLB 风险基因座 GBA 对 7 个相互关联的 CSF 水平显示出正向反式效应。无论是总体样本还是仅在 FTD 中进行的分析,都没有发现额颞叶痴呆的 pQTL。我们在对AD和DLB的分层分析中进一步发现了pQTLs,这暗示了痴呆症中的疾病特异性pQTLs。剖析风险基因位点对神经生物学过程的贡献有助于理解痴呆症的疾病机制。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Connecting dementia risk loci to the CSF proteome identifies pathophysiological leads for dementia.

Genome-wide association studies have successfully identified many genetic risk loci for dementia, but exact biological mechanisms through which genetic risk factors contribute to dementia remains unclear. Integrating CSF proteomic data with dementia risk loci could reveal intermediate molecular pathways connecting genetic variance to the development of dementia. We tested to what extent effects of known dementia risk loci can be observed in CSF levels of 665 proteins [proximity extension-based (PEA) immunoassays] in a deeply-phenotyped mixed memory clinic cohort [n = 502, mean age (standard deviation, SD) = 64.1 (8.7) years, 181 female (35.4%)], including patients with Alzheimer's disease (AD, n = 213), dementia with Lewy bodies (DLB, n = 50) and frontotemporal dementia (FTD, n = 93), and controls (n = 146). Validation was assessed in independent cohorts (n = 99 PEA platform, n = 198, mass reaction monitoring-targeted mass spectroscopy and multiplex assay). We performed additional analyses stratified according to diagnostic status (AD, DLB, FTD and controls separately), to explore whether associations between CSF proteins and genetic variants were specific to disease or not. We identified four AD risk loci as protein quantitative trait loci (pQTL): CR1-CR2 (rs3818361, P = 1.65 × 10-8), ZCWPW1-PILRB (rs1476679, P = 2.73 × 10-32), CTSH-CTSH (rs3784539, P = 2.88 × 10-24) and HESX1-RETN (rs186108507, P = 8.39 × 10-8), of which the first three pQTLs showed direct replication in the independent cohorts. We identified one AD-specific association between a rare genetic variant of TREM2 and CSF IL6 levels (rs75932628, P = 3.90 × 10-7). DLB risk locus GBA showed positive trans effects on seven inter-related CSF levels in DLB patients only. No pQTLs were identified for FTD loci, either for the total sample as for analyses performed within FTD only. Protein QTL variants were involved in the immune system, highlighting the importance of this system in the pathophysiology of dementia. We further identified pQTLs in stratified analyses for AD and DLB, hinting at disease-specific pQTLs in dementia. Dissecting the contribution of risk loci to neurobiological processes aids in understanding disease mechanisms underlying dementia.

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来源期刊
Brain
Brain 医学-临床神经学
CiteScore
20.30
自引率
4.10%
发文量
458
审稿时长
3-6 weeks
期刊介绍: Brain, a journal focused on clinical neurology and translational neuroscience, has been publishing landmark papers since 1878. The journal aims to expand its scope by including studies that shed light on disease mechanisms and conducting innovative clinical trials for brain disorders. With a wide range of topics covered, the Editorial Board represents the international readership and diverse coverage of the journal. Accepted articles are promptly posted online, typically within a few weeks of acceptance. As of 2022, Brain holds an impressive impact factor of 14.5, according to the Journal Citation Reports.
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