Evidence map›Paper›PMID 40062617›Full record

ArticleBriefings in bioinformatics2025

Optimized phenotyping of complex morphological traits: enhancing discovery of common and rare genetic variants.

Meng Yuan, Seppe Goovaerts, Myoung K Lee, Jay Devine, Stephen Richmond, Susan Walsh, Mark D Shriver, John R Shaffer, Mary L Marazita, Hilde Peeters and 2 more

Abstract read
In one paragraph

Article in Briefings in bioinformatics, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing 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

1 citing paper in PubMed.

  1. Review
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

12 authors.

Meng YuanDepartment of Electrical Engineering, ESAT/PSI, KU Leuven, Oude Markt 13, 3000 Leuven, Belgium.ORCID 0000-0003-0922-8012
Seppe GoovaertsDepartment of Human Genetics, KU Leuven, Oude Markt 13, 3000 Leuven, Belgium.
Myoung K LeeCenter for Craniofacial and Dental Genetics, Department of Oral and Craniofacial Sciences, University of Pittsburgh, Pittsburgh, 4200 Fifth Ave, Pittsburgh, PA 15260, United States.
Jay DevineDepartment of Electrical Engineering, ESAT/PSI, KU Leuven, Oude Markt 13, 3000 Leuven, Belgium.
Stephen RichmondApplied Clinical Research and Public Health, School of Dentistry, Cardiff University, Cardiff CF10 3AT, United Kingdom.
Susan WalshDepartment of Biology, Indiana University Indianapolis, 420 University Blvd, Indianapolis 46202, IN, United States.
Mark D ShriverDepartment of Anthropology, Pennsylvania State University, 201 Old Main, University Park, PA 16802, United States.
John R ShafferCenter for Craniofacial and Dental Genetics, Department of Oral and Craniofacial Sciences, University of Pittsburgh, Pittsburgh, 4200 Fifth Ave, Pittsburgh, PA 15260, United States.
Mary L MarazitaCenter for Craniofacial and Dental Genetics, Department of Oral and Craniofacial Sciences, University of Pittsburgh, Pittsburgh, 4200 Fifth Ave, Pittsburgh, PA 15260, United States.
Hilde PeetersDepartment of Human Genetics, KU Leuven, Oude Markt 13, 3000 Leuven, Belgium.
Seth M WeinbergCenter for Craniofacial and Dental Genetics, Department of Oral and Craniofacial Sciences, University of Pittsburgh, Pittsburgh, 4200 Fifth Ave, Pittsburgh, PA 15260, United States.
Peter ClaesDepartment of Electrical Engineering, ESAT/PSI, KU Leuven, Oude Markt 13, 3000 Leuven, Belgium.

Funding

Research Foundation-Flanders FWO G017225NResearch Fund KU Leuven BOF-C1, C14/20/081UK Medical Research Council and Wellcome 217065/Z/19/ZWellcome TrustWellcome Trust 092731
6 · The paper itself

Abstract

Genotype-phenotype (G-P) analyses for complex morphological traits typically utilize simple, predetermined anatomical measures or features derived via unsupervised dimension reduction techniques (e.g. principal component analysis (PCA) or eigen-shapes). Despite the popularity of these approaches, they do not necessarily reveal axes of phenotypic variation that are genetically relevant. Therefore, we introduce a framework to optimize phenotyping for G-P analyses, such as genome-wide association studies (GWAS) of common variants or rare variant association studies (RVAS) of rare variants. Our strategy is two-fold: (i) we construct a multidimensional feature space spanning a wide range of phenotypic variation, and (ii) within this feature space, we use an optimization algorithm to search for directions or feature combinations that are genetically enriched. To test our approach, we examine human facial shape in the context of GWAS and RVAS. In GWAS, we optimize for phenotypes exhibiting high heritability, estimated from either family data or genomic relatedness measured in unrelated individuals. In RVAS, we optimize for the skewness of phenotype distributions, aiming to detect commingled distributions that suggest single or few genomic loci with major effects. We compare our approach with eigen-shapes as baseline in GWAS involving 8246 individuals of European ancestry and in gene-based tests of rare variants with a subset of 1906 individuals. After applying linkage disequilibrium score regression to our GWAS results, heritability-enriched phenotypes yielded the highest SNP heritability, followed by eigen-shapes, while commingling-based traits displayed the lowest SNP heritability. Heritability-enriched phenotypes also exhibited higher discovery rates, identifying the same number of independent genomic loci as eigen-shapes with a smaller effective number of traits. For RVAS, commingling-based traits resulted in more genes passing the exome-wide significance threshold than eigen-shapes, while heritability-enriched phenotypes lead to only a few associations. Overall, our results demonstrate that optimized phenotyping allows for the extraction of genetically relevant traits that can specifically enhance discovery efforts of common and rare variants, as evidenced by their increased power in facial GWAS and RVAS.

Indexed as

Genetic VariationGenome-Wide Association StudyPhenotypeAlgorithmsGenotypeHumansPolymorphism, Single NucleotidePrincipal Component Analysis3D shapecomplex traitfacial phenotypinggenotype–phenotype associationheritability

Identifiers

PMID40062617
PMCPMC11891655

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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.