A method to estimate the contribution of rare coding variants to complex trait heritability.

Pathan, Nazia; Deng, Wei Q; Di Scipio, Matteo; Khan, Mohammad; Mao, Shihong; Morton, Robert W; Lali, Ricky; Pigeyre, Marie et al. · Nat Commun · 2024

basic_science · Level V

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Abstract

It has been postulated that rare coding variants (RVs; MAF < 0.01) contribute to the "missing" heritability of complex traits. We developed a framework, the Rare variant heritability (RARity) estimator, to assess RV heritability (h<sup>2</sup><sub>RV</sub>) without assuming a particular genetic architecture. We applied RARity to 31 complex traits in the UK Biobank (n = 167,348) and showed that gene-level RV aggregation suffers from 79% (95% CI: 68-93%) loss of h<sup>2</sup><sub>RV</sub>. Using unaggregated variants, 27 traits had h<sup>2</sup><sub>RV</sub> > 5%, with height having the highest h<sup>2</sup><sub>RV</sub> at 21.9% (95% CI: 19.0-24.8%). The total heritability, including common and rare variants, recovered pedigree-based estimates for 11 traits. RARity can estimate gene-level h<sup>2</sup><sub>RV</sub>, enabling the assessment of gene-level characteristics and revealing 11, previously unreported, gene-phenotype relationships. Finally, we demonstrated that in silico pathogenicity prediction (variant-level) and gene-level annotations do not generally enrich for RVs that over-contribute to complex trait variance, and thus, innovative methods are needed to predict RV functionality.

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