Evidence map›Paper›PMID 40551016›Full record

ReviewNature genetics2025

Using large-scale population-based data to improve disease risk assessment of clinical variants.

Iain S Forrest, Kuan-Lin Huang, Julie M Eggington, Wendy K Chung, Daniel M Jordan, Ron Do

Abstract readReview
In one paragraph

Review in Nature genetics, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.

0numbers the graph read from it
0cells of the map it votes in
7citing 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

7 citing papers in PubMed.

  1. Genetic influences on haematopoiesis.Nature reviews. Genetics · 2026
    Review
  2. Article
  3. Article
  4. Review
  5. Article
  6. Article
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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.

Iain S ForrestThe Charles Bronfman Institute for Personalized Medicine, Icahn School of Medicine at Mount Sinai, New York, NY, USA.ORCID http://orcid.org/0000-0002-5642-422X
Kuan-Lin HuangDepartment of Genetics and Genomic Sciences, Icahn School of Medicine at Mount Sinai, New York, NY, USA.ORCID http://orcid.org/0000-0002-5537-5817
Julie M EggingtonCenter for Genomic Interpretation, Sandy, UT, USA.ORCID http://orcid.org/0000-0002-0703-9296
Wendy K ChungDepartment of Pediatrics, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.ORCID http://orcid.org/0000-0003-3438-5685
Daniel M JordanThe Charles Bronfman Institute for Personalized Medicine, Icahn School of Medicine at Mount Sinai, New York, NY, USA. daniel.jordan@mssm.edu.ORCID http://orcid.org/0000-0002-5318-8225
Ron DoThe Charles Bronfman Institute for Personalized Medicine, Icahn School of Medicine at Mount Sinai, New York, NY, USA. ron.do@mssm.edu.ORCID http://orcid.org/0000-0002-3144-3627

Funding

Genetic Analysis and Manipulation Core (GAEC)P50HD105351 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI Hisashi Umemori · 2021 to 2026
$9.4M
Towards an integrated map of causal connections for common, complex diseasesR35GM124836 · NIGMS · ICAHN SCHOOL OF MEDICINE AT MOUNT SINAI · PI Ron Do · 2017 to 2026
$3.9M
Integrative Approaches for Identifying Causal Gene-Cell Type Pairs of Complex DiseaseR35GM138113 · NIGMS · ICAHN SCHOOL OF MEDICINE AT MOUNT SINAI · PI Kuan-lin Huang · 2020 to 2026
$3.0M
Development of recommendations and policies for genetic variant reclassificationR01HG010365 · NHGRI · COLUMBIA UNIVERSITY HEALTH SCIENCES · PI APPELBAUM, PAUL STUART, CHUNG, WENDY K · 2018 to 2021
$2.9M
NHGRI NIH HHS R01 HG010365NICHD NIH HHS P50 HD105351NIGMS NIH HHS R35 GM124836NIGMS NIH HHS R35 GM138113U.S. Department of Health & Human Services | NIH | National Institute of General Medical Sciences (NIGMS) R35-GM124836
6 · The paper itself

Abstract

Understanding the disease risk of genetic variants is fundamental to precision medicine. Estimates of penetrance-the probability of disease for individuals with a variant allele-rely on disease-specific cohorts, clinical testing and emerging electronic health record (EHR)-linked biobanks. These data sources, while valuable, each have limitations in quality, representativeness and analyzability. Here, we provide a historical account of the currently accepted pathogenicity classification system and data available in ClinVar, a public archive that aggregates variant interpretations but lacks detailed data for accurate penetrance assessment, highlighting its oversimplification of disease risk. We propose an integrative Bayesian framework that unifies pathogenicity and penetrance, leveraging both functional and real-world evidence to refine risk predictions. In addition, we advocate for enhancing ClinVar with the inclusion of high-priority phenotypes, age-stratified data and population-based cohorts linked to EHRs. We suggest developing a community repository of population-based penetrance estimates to support the clinical application of genetic data.

Indexed as

Genetic Predisposition to DiseaseGenetic VariationBayes TheoremDatabases, GeneticElectronic Health RecordsHumansPenetrancePrecision MedicineRisk Assessment

Identifiers

PMID40551016
PMCPMC12321300

What Socratic holds

Textmetadata
LicenceTDM
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 Socratic graph.