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Record W4411753936 · doi:10.1101/2025.06.26.25330270

A flexible pipeline for reproducible exome-wide rare variant gene-trait associations

2025· preprint· en· W4411753936 on OpenAlexafffund
Kevin Y. H. Liang, Ethan Kreuzer, Yann Ilboudo, Yiheng Chen, J. Brent Richards, Guillaume Butler‐Laporte

Bibliographic record

VenuemedRxiv · 2025
Typepreprint
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomics and Rare Diseases
Canadian institutionsMcGill University Health CentreMcGill UniversityHEC MontréalJewish General Hospital
FundersFonds de Recherche du Québec - SantéCanadian Institutes of Health ResearchJewish General HospitalPublic Health AgencyCompute CanadaNational Institutes of HealthPublic Health Agency of CanadaCancer Research UKMcGill University
KeywordsExome sequencingPipeline (software)TraitExomeComputational biologyGeneGeneticsBiologyComputer sciencePhenotype

Abstract

fetched live from OpenAlex

Abstract Summary Exome-wide gene-burden association studies are widely used to assess gene – trait relationships. By focusing on coding variants, such analyses can directly quantify the magnitude and direction of a gene’s effect on a given trait across the proteome, information that cannot be easily derived from genome-wide associate studies. However, the lack of a standardized workflow poses a significant challenge for reproducibility. Here, we provide a customizable workflow implemented in Python 3 and Nextflow for performing exome-wide rare variant gene – trait association testing. We demonstrated its utility by replicating three recent studies. This workflow will also serve as a framework for performing similar analyses in a standardized and systematic manner. Availability and Implementation This workflow is publicly available at https://github.com/richardslab/EXWAS_pipeline under the MIT license. Supplementary information Supplementary information will be made available online.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.012
metaresearch head score (Gemma)0.018
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesMetaresearch
Consensus categoriesnone
DomainCandidate signal: Reproducibility · Consensus signal: none
Study designCandidate signal: Simulation or modeling · Consensus signal: none
GenreCandidate signal: Methods · Consensus signal: Methods
Teacher disagreement score0.988
Threshold uncertainty score0.127

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0120.018
Meta-epidemiology (narrow)0.0020.002
Meta-epidemiology (broad)0.0020.004
Bibliometrics0.0030.002
Science and technology studies0.0020.001
Scholarly communication0.0040.002
Open science0.0030.008
Research integrity0.0010.004
Insufficient payload (model declined to judge)0.0380.022

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.023
GPT teacher head0.286
Teacher spread0.264 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

Study designSimulation or modeling
DomainReproducibility
GenreMethods

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations0
Published2025
Admission routes2
Has abstractyes

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Same venuemedRxiv→Same topicGenomics and Rare Diseases→French-language works237,207→