Duncan NRI Faculty and Staff Publications

Language

English

Publication Date

9-4-2025

Journal

American Journal of Human Genetics

DOI

10.1016/j.ajhg.2025.07.012

PMID

40829599

PMCID

PMC12461002

PubMedCentral® Posted Date

8-18-2025

PubMedCentral® Full Text Version

Post-print

Abstract

We present the Causal Pivot (CP) as a structural causal model (SCM) for analyzing genetic heterogeneity in complex diseases. The CP leverages an established causal factor or factors to detect the contribution of additional suspected causes. Specifically, polygenic risk scores (PRSs) serve as known causes, while rare variants (RVs) or RV ensembles are evaluated as candidate causes. The CP incorporates outcome-induced association by conditioning on disease status. We derive a conditional maximum-likelihood procedure for binary and quantitative traits and develop the Causal Pivot likelihood ratio test (CP-LRT) to detect causal signals. Through simulations, we demonstrate the CP-LRT's robust power and superior error control compared to alternatives. We apply the CP-LRT to UK Biobank (UKB) data, analyzing three exemplar diseases: hypercholesterolemia (HC, low-density lipoprotein cholesterol ≥4.9 mmol/L; n

Keywords

Humans, Genetic Heterogeneity, Breast Neoplasms, Multifactorial Inheritance, Parkinson Disease, Genetic Predisposition to Disease, Female, Models, Genetic, Genetic Variation, Likelihood Functions, Genome-Wide Association Study, causal inference, genetic heterogeneity, complex disease, collider, rare variation, polygenic risk score

Published Open-Access

yes

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