Simplifying causal gene identification in GWAS loci

Genome-wide association studies (GWAS) help to identify disease-linked genetic variants, but pinpointing the most likely causal genes in GWAS loci remains challenging. Existing GWAS gene prioritization tools are powerful but often use complex black box models trained on datasets containing biases. Here, we used a data-driven approach to construct a truth set of causal genes in 200 GWAS loci. We found that a simple logistic regression model performed as well as a more complex XGBoost model, and that many commonly-used gene prioritization features could be removed without meaningfully affecting performance (e.g., expression quantitative trait locus colocalization and Mendelian randomization). We present CALDERA, a gene prioritization tool that uses a logistic regression model and uses just four input features. In independent benchmarking datasets of resolved GWAS loci, CALDERA achieved state-of-the-art performance in comparison with other methods (FLAMES, L2G, and cS2G). CALDERA outputs causal gene probabilities for all genes in a given GWAS locus and we show that these probabilities are well-calibrated. Applying CALDERA to 93 UK Biobank traits, we predicted 11,956 putative causal genes, potentially resolving up to 52% of loci. Overall, CALDERA provides a powerful solution for prioritizing potentially causal genes in GWAS loci that minimizes the data processing required to construct input features and generates an easily-interpretable output score.

Authors

Institutions

Publication Details

Journal
PLoS Genetics
Published
2026-03-17
DOI
https://doi.org/10.1371/journal.pgen.1012079
Primary Topic
Genetic Associations and Epidemiology
Type
article
Field-Weighted Citation Impact
0.00
Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

Simplifying causal gene identification in GWAS loci

Marijn Schipper, Stephan Ripke, Karl Heilbron, Jacob Ulirsch et al.
PLoS Genetics
Genetic Associations and Epidemiology
article

Simplifying causal gene identification in GWAS loci

Marijn Schipper, Stephan Ripke, Karl Heilbron, Jacob Ulirsch, Danielle Posthuma
article en

Abstract

Genome-wide association studies (GWAS) help to identify disease-linked genetic variants, but pinpointing the most likely causal genes in GWAS loci remains challenging. Existing GWAS gene prioritization tools are powerful but often use complex black box models trained on datasets containing biases. Here, we used a data-driven approach to construct a truth set of causal genes in 200 GWAS loci. We found that a simple logistic regression model performed as well as a more complex XGBoost model, and that many commonly-used gene prioritization features could be removed without meaningfully affecting performance (e.g., expression quantitative trait locus colocalization and Mendelian randomization). We present CALDERA, a gene prioritization tool that uses a logistic regression model and uses just four input features. In independent benchmarking datasets of resolved GWAS loci, CALDERA achieved state-of-the-art performance in comparison with other methods (FLAMES, L2G, and cS2G). CALDERA outputs causal gene probabilities for all genes in a given GWAS locus and we show that these probabilities are well-calibrated. Applying CALDERA to 93 UK Biobank traits, we predicted 11,956 putative causal genes, potentially resolving up to 52% of loci. Overall, CALDERA provides a powerful solution for prioritizing potentially causal genes in GWAS loci that minimizes the data processing required to construct input features and generates an easily-interpretable output score.

PLoS GeneticsVol. 22(3)
Broad Institute (US), Illumina (United States) (US), Amsterdam Neuroscience (NL), Charité - Universitätsmedizin Berlin (DE), Vrije Universiteit Amsterdam (NL)
Openalex Percentile: Top 20%
Genetic Associations and Epidemiology
AI Navigator

Ask Laika to Summarize, Analyze, and Connect papers live on the map.

Summarize Papers & Methodologies

Extract key findings, datasets, and comparative methods across publications.

Benchmark Rankings & Visual Analytics

Rank top research institutions, authors, funders, topics, and journals by Field-Weighted Citation Impact (FWCI) and paper volume with instant charts.

Connect Distant Disciplines

Bridge topological clusters on the map to find hidden collaborative intersections.