Suppr超能文献

在单核苷酸分辨率下对复杂和分子性状变异进行功能解析。

Functional dissection of complex and molecular trait variants at single nucleotide resolution.

作者信息

Siraj Layla, Castro Rodrigo I, Dewey Hannah, Kales Susan, Nguyen Thanh Thanh L, Kanai Masahiro, Berenzy Daniel, Mouri Kousuke, Wang Qingbo S, McCaw Zachary R, Gosai Sager J, Aguet François, Cui Ran, Vockley Christopher M, Lareau Caleb A, Okada Yukinori, Gusev Alexander, Jones Thouis R, Lander Eric S, Sabeti Pardis C, Finucane Hilary K, Reilly Steven K, Ulirsch Jacob C, Tewhey Ryan

机构信息

Broad Institute of Harvard and MIT, Cambridge, MA, USA.

Program in Biophysics, Harvard Graduate School of Arts and Sciences, Boston, MA, USA.

出版信息

bioRxiv. 2024 May 6:2024.05.05.592437. doi: 10.1101/2024.05.05.592437.

Abstract

Identifying the causal variants and mechanisms that drive complex traits and diseases remains a core problem in human genetics. The majority of these variants have individually weak effects and lie in non-coding gene-regulatory elements where we lack a complete understanding of how single nucleotide alterations modulate transcriptional processes to affect human phenotypes. To address this, we measured the activity of 221,412 trait-associated variants that had been statistically fine-mapped using a Massively Parallel Reporter Assay (MPRA) in 5 diverse cell-types. We show that MPRA is able to discriminate between likely causal variants and controls, identifying 12,025 regulatory variants with high precision. Although the effects of these variants largely agree with orthogonal measures of function, only 69% can plausibly be explained by the disruption of a known transcription factor (TF) binding motif. We dissect the mechanisms of 136 variants using saturation mutagenesis and assign impacted TFs for 91% of variants without a clear canonical mechanism. Finally, we provide evidence that epistasis is prevalent for variants in close proximity and identify multiple functional variants on the same haplotype at a small, but important, subset of trait-associated loci. Overall, our study provides a systematic functional characterization of likely causal common variants underlying complex and molecular human traits, enabling new insights into the regulatory grammar underlying disease risk.

摘要

识别驱动复杂性状和疾病的因果变异及机制仍然是人类遗传学中的一个核心问题。这些变异中的大多数个体效应较弱,且位于非编码基因调控元件中,而我们对单核苷酸改变如何调节转录过程以影响人类表型尚缺乏全面的了解。为了解决这一问题,我们在5种不同的细胞类型中,使用大规模平行报告基因检测(MPRA)对221,412个经统计精细定位的性状相关变异的活性进行了测量。我们表明,MPRA能够区分可能的因果变异和对照,高精度地识别出12,025个调控变异。尽管这些变异的效应在很大程度上与功能的正交测量结果一致,但只有69%的变异可以通过已知转录因子(TF)结合基序的破坏得到合理的解释。我们使用饱和诱变剖析了136个变异的机制,并为91%没有明确典型机制的变异确定了受影响的TF。最后,我们提供证据表明,上位性在紧密相邻的变异中普遍存在,并在一小部分但重要的性状相关位点上,在同一单倍型上鉴定出多个功能变异。总体而言,我们的研究对复杂和分子人类性状潜在的可能因果常见变异进行了系统的功能表征,为疾病风险背后的调控语法提供了新的见解。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e982/11100724/53c16b8ffe04/nihpp-2024.05.05.592437v1-f0001.jpg

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验