Logo image
Dynamic incorporation of multiple in silico functional annotations empowers rare variant association analysis of large whole-genome sequencing studies at scale
Journal article   Peer reviewed

Dynamic incorporation of multiple in silico functional annotations empowers rare variant association analysis of large whole-genome sequencing studies at scale

Xihao Li, Barry I Freedman, Xiuqing Guo, Zilin Li, George Hindy, Hufeng Zhou, Sheila M Gaynor, Marguerite R Irvin, Yaowu Liu, Sharon L R Kardia, …
Nature genetics, Vol.52(9), pp.969-983
09/2020
DOI: 10.1038/s41588-020-0676-4
PMCID: PMC7483769
PMID: 32839606
url
https://www.ncbi.nlm.nih.gov/pmc/articles/7483769View
Open Access

Abstract

Large-scale whole-genome sequencing studies have enabled the analysis of rare variants (RVs) associated with complex phenotypes. Commonly used RV association tests have limited scope to leverage variant functions. We propose STAAR (variant-set test for association using annotation information), a scalable and powerful RV association test method that effectively incorporates both variant categories and multiple complementary annotations using a dynamic weighting scheme. For the latter, we introduce 'annotation principal components', multidimensional summaries of in silico variant annotations. STAAR accounts for population structure and relatedness and is scalable for analyzing very large cohort and biobank whole-genome sequencing studies of continuous and dichotomous traits. We applied STAAR to identify RVs associated with four lipid traits in 12,316 discovery and 17,822 replication samples from the Trans-Omics for Precision Medicine Program. We discovered and replicated new RV associations, including disruptive missense RVs of NPC1L1 and an intergenic region near APOC1P1 associated with low-density lipoprotein cholesterol.
Phenotype Cholesterol, LDL - genetics Computer Simulation Genetic Predisposition to Disease - genetics Genetic Variation - genetics Genome - genetics Genome-Wide Association Study - methods Humans Models, Genetic Molecular Sequence Annotation - methods Whole Genome Sequencing - methods

Details

Metrics

Logo image