Evidence map›Paper›PMID 42362888›Full record

ReviewHuman genetics2026

AI in variant analysis: fast track to genetic diagnoses.

Elizabeth J Wilk, Sasha Taluri, Timothy C Howton, Anthony B Crumley, Michal Mrug, Brittany N Lasseigne

Abstract readReview
In one paragraph

Review in Human genetics, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

6 authors.

Elizabeth J Wilk *The Department of Cell, Developmental and Integrative Biology, Heersink School of Medicine, The University of Alabama at Birmingham, Birmingham, AL, USA.ORCID http://orcid.org/0000-0002-7078-1215
Sasha Taluri *The Department of Cell, Developmental and Integrative Biology, Heersink School of Medicine, The University of Alabama at Birmingham, Birmingham, AL, USA.ORCID http://orcid.org/0009-0005-1964-2120
Timothy C HowtonThe Department of Cell, Developmental and Integrative Biology, Heersink School of Medicine, The University of Alabama at Birmingham, Birmingham, AL, USA.ORCID http://orcid.org/0000-0002-9423-0135
Anthony B CrumleyThe Department of Cell, Developmental and Integrative Biology, Heersink School of Medicine, The University of Alabama at Birmingham, Birmingham, AL, USA.ORCID http://orcid.org/0009-0004-7347-4722
Michal MrugThe Department of Medicine, Heersink School of Medicine, The University of Alabama at Birmingham, Birmingham, AL, USA.ORCID http://orcid.org/0000-0001-8981-1843
Brittany N LasseigneThe Department of Cell, Developmental and Integrative Biology, Heersink School of Medicine, The University of Alabama at Birmingham, Birmingham, AL, USA. bnp0001@uab.edu.ORCID http://orcid.org/0000-0002-1642-8904

Funding

UAB Pilot Center for Precision Animal Modeling (C-PAM) - Resource and Service SectionU54OD030167 · OD · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI Bradley K. Yoder · 2020 to 2026
$15.3M
UAB Childhood Cystic Kidney Disease Core Center (UAB-CCKDCC) - Therapeutic Development and Screening ResourceU54DK126087 · NIDDK · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI Bradley K. Yoder · 2020 to 2026
$6.7M
Biomedical Laboratory Research and Development, VA Office of Research and Development 1-I01-BX006266-01BLRD VA I01 BX006266NIDDK NIH HHS U54 DK126087NIH HHS U54 OD030167NIH HHS U54-OD030167
6 · The paper itself

Abstract

While falling costs have expanded access to genomic sequencing, clinical utility is frequently hindered by the challenge of interpreting complex genetic data. Variant analysis for rare disease patients especially requires significant time and expertise, creating a bottleneck that delays diagnostics. Although advances in genetic variant classification have improved diagnostic precision, they have also increased the identification of variants of uncertain significance (VUSs), widening the interpretation gap between data generation and clinical actionability. The high prevalence of VUSs can lead to false reassurance or psychological distress by misinterpretting inconclusive results. We propose that artificial intelligence (AI) is a critical clinical decision-support tool for bridging this gap, offering a scalable framework to optimize variant interpretation and shorten the diagnostic odyssey. While reclassification ultimately requires biological evidence that AI cannot replace, these tools serve as essential aggregators and prioritizers, especially as guidelines transition toward the upcoming quantitative ACMG v4 framework. We advocate integrating AI throughout the genetic diagnostic workflow-from initial phenotyping to variant prioritization-to facilitate data-driven, personalized treatment. We outline current AI-assisted approaches and discuss anticipated challenges in this pursuit, such as privacy, training data bias and quality, model explainability, and the necessity of a total product life cycle for validation. To address these challenges, we provide recommendations for "human-in-the-loop" design and intuitive workflow integration to ensure AI tools meet the highest standards of precision, reproducibility, and transparency to maximize adoption. By standardizing AI across the variant analysis pipeline, we can fast-track the path to genetic diagnoses, effectively bridging the interpretation gap and enabling rapid delivery of personalized medical interventions.

Indexed as

Artificial IntelligenceGenetic TestingGenetic VariationGenomicsHumans

Identifiers

PMID42362888
PMCPMC13309432

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.