Tracing the evolutionary histories of ultra-rare variants using variational dating of large ancestral recombination graphs

Ultra-rare variants dominate whole-genome sequencing datasets, yet their interpretation is limited by allele frequency, which provides little information at very low counts and is highly sensitive to uneven ancestry representation. Allele age offers an ancestry-agnostic alternative but existing methods do not scale to biobank-sized cohorts. Here we present a scalable variational algorithm for dating Ancestral Recombination Graphs (ARGs), implemented in tsdate, together with new distributed methods enabling practical biobank-scale ARG inference using tsinfer. Applied to 47,535 genomes from the Genomics England 100,000 Genomes Project, we infer contiguous ARGs spanning 206 Mb and estimate ages for 23.2 million variants, including 11.8 million singletons. ARG-based allele ages remain accurate under extreme sampling imbalance and, in real data, reveal signatures of purifying selection and clinically relevant heterogeneity among variants with identical observed frequencies. Estimates for recent mutations are precise only at large sample sizes, highlighting the information accessible in the haplotype structure of large datasets. Biobank-scale ARGs therefore enable robust, ancestry-agnostic age estimation for ultra-rare variation with broad utility for statistical and clinical genomics.

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Publication Details

Journal
bioRxiv (Cold Spring Harbor Laboratory)
Published
2026-01-12
DOI
https://doi.org/10.64898/2026.01.07.698223
Primary Topic
Genomics and Rare Diseases
Type
article
Field-Weighted Citation Impact
0.00

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article

Tracing the evolutionary histories of ultra-rare variants using variational dating of large ancestral recombination graphs

Jerome Kelleher, Samuel Tallman, Pope Nathaniel, Peter L. Ralph et al.
bioRxiv (Cold Spring Harbor Laboratory)
Genomics and Rare Diseases
article

Tracing the evolutionary histories of ultra-rare variants using variational dating of large ancestral recombination graphs

Jerome Kelleher, Samuel Tallman, Pope Nathaniel, Peter L. Ralph, Benjamin Jeffery
article en

Abstract

Ultra-rare variants dominate whole-genome sequencing datasets, yet their interpretation is limited by allele frequency, which provides little information at very low counts and is highly sensitive to uneven ancestry representation. Allele age offers an ancestry-agnostic alternative but existing methods do not scale to biobank-sized cohorts. Here we present a scalable variational algorithm for dating Ancestral Recombination Graphs (ARGs), implemented in tsdate, together with new distributed methods enabling practical biobank-scale ARG inference using tsinfer. Applied to 47,535 genomes from the Genomics England 100,000 Genomes Project, we infer contiguous ARGs spanning 206 Mb and estimate ages for 23.2 million variants, including 11.8 million singletons. ARG-based allele ages remain accurate under extreme sampling imbalance and, in real data, reveal signatures of purifying selection and clinically relevant heterogeneity among variants with identical observed frequencies. Estimates for recent mutations are precise only at large sample sizes, highlighting the information accessible in the haplotype structure of large datasets. Biobank-scale ARGs therefore enable robust, ancestry-agnostic age estimation for ultra-rare variation with broad utility for statistical and clinical genomics.

bioRxiv (Cold Spring Harbor Laboratory)
University of Oregon (US), Health Data Research UK (GB), Genomics England (GB)
Robertson Foundation, Department of Health and Social Care, National Institutes of Health, National Human Genome Research Institute
Openalex Percentile: Top 91%
Genomics and Rare Diseases
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