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Record W4200036706 · doi:10.1161/circresaha.120.317107

Common Genetic Variants Contribute to Risk of Transposition of the Great Arteries

2021· article· en· W4200036706 on OpenAlexaff
Doris Škorić‐Milosavljević, Rafik Tadros, Fernanda M. Bosada, Federico Tessadori, Jan Hendrik van Weerd, Odilia I. Woudstra, Fleur V.Y. Tjong, Najim Lahrouchi, Fanny Bajolle, Heather J. Cordell, A. J. Agopian, Gillian M. Blue, Daniela Q.C.M. Barge‐Schaapveld, Marc Gewillig, Christoph Preuß, Elisabeth M. Lodder, Phil Barnett, Aho Ilgun, Leander Beekman, Karel van Duijvenboden, Regina Bökenkamp, Martina Müller‐Nurasyid, Hubert W. Vliegen, Thelma C. Konings, Joost P. van Melle, Arie P.J. van Dijk, Roland R.J. van Kimmenade, Jolien W. Roos‐Hesselink, Gertjan T. Sieswerda, Folkert J. Meijboom, Hashim Abdul‐Khaliq, Felix Berger, Sven Dittrich, Marc‐Phillip Hitz, Julia Moosmann, Frank‐Thomas Riede, Stephan Schubert, Pilar Galán, Mark Lathrop, Hans Markus Münter, Ammar Al‐Chalabi, Christopher E. Shaw, Pamela J. Shaw, Karen Morrison, Jan H. Veldink, Leonard H. van den Berg, Sylvia Μ. Evans, Marcelo A. Nóbrega, Ivy Aneas, Milena Radivojkov‐Blagojevic, Thomas Meitinger, Erwin Oechslin, Tapas Mondal, Lynn Bergin, John Smythe, Luis Altamirano‐Diaz, Jane Lougheed, Berto J. Bouma, Marie Chaix, Jennie Kline, Anne S. Bassett, Grégor Andelfinger, Roel L.F. van der Palen, Patrice Bouvagnet, Sally‐Ann B. Clur, Jeroen Breckpot, Wilhelmina S. Kerstjens‐Frederikse, David S. Winlaw, Ulrike Bauer, Seema Mital, Elizabeth Goldmuntz, Bernard Keavney, Damien Bonnet, Barbara J.M. Mulder, Michael W.T. Tanck, Jeroen Bakkers, Vincent M. Christoffels, Cornelis J. Boogerd, Alex V. Postma, Connie R. Bezzina

Bibliographic record

VenueCirculation Research · 2021
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicCongenital heart defects research
Canadian institutionsHospital for Sick ChildrenCentre for Addiction and Mental HealthMcMaster University Medical CentreUniversity Health NetworkChildren's Hospital of Eastern OntarioLondon Health Sciences CentreCentre Hospitalier Universitaire Sainte-JustineMcMaster UniversityWestern UniversityMcGill Genome CentreKingston General HospitalMontreal Heart Institute
FundersEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentNational Center for Advancing Translational SciencesWellcome TrustChildren's Heart FoundationNational Human Genome Research InstituteNational Center for Research ResourcesMedical Research CouncilNational Heart, Lung, and Blood InstituteHartstichtingFondation LeducqMotor Neurone Disease AssociationNational Institute for Health and Care ResearchBritish Heart Foundation
KeywordsGreat arteriesGeneticsSingle-nucleotide polymorphismBiologyLocus (genetics)Genome-wide association studyHaplotypeAlleleGeneZebrafishMendelian inheritanceSNPEnhancerTranscription factorGenotypeHeart diseaseMedicineInternal medicine

Abstract

fetched live from OpenAlex

Rationale: Dextro-transposition of the great arteries (D-TGA) is a severe congenital heart defect which affects approximately 1 in 4,000 live births. While there are several reports of D-TGA patients with rare variants in individual genes, the majority of D-TGA cases remain genetically elusive. Familial recurrence patterns and the observation that most cases with D-TGA are sporadic suggest a polygenic inheritance for the disorder, yet this remains unexplored. Objective: We sought to study the role of common single nucleotide polymorphisms (SNPs) in risk for D-TGA. Methods and Results: We conducted a genome-wide association study in an international set of 1,237 patients with D-TGA and identified a genome-wide significant susceptibility locus on chromosome 3p14.3, which was subsequently replicated in an independent case-control set (rs56219800, meta-analysis P=8.6x10 -10 , OR=0.69 per C allele). SNP-based heritability analysis showed that 25% of variance in susceptibility to D-TGA may be explained by common variants. A genome-wide polygenic risk score derived from the discovery set was significantly associated to D-TGA in the replication set (P=4x10 -5 ). The genome-wide significant locus (3p14.3) co-localizes with a putative regulatory element that interacts with the promoter of WNT5A , which encodes the Wnt Family Member 5A protein known for its role in cardiac development in mice. We show that this element drives reporter gene activity in the developing heart of mice and zebrafish and is bound by the developmental transcription factor TBX20. We further demonstrate that TBX20 attenuates Wnt5a expression levels in the developing mouse heart. Conclusions: This work provides support for a polygenic architecture in D-TGA and identifies a susceptibility locus on chromosome 3p14.3 near WNT5A . Genomic and functional data support a causal role of WNT5A at the locus.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.495
Threshold uncertainty score0.219

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

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 head0.031
GPT teacher head0.337
Teacher spread0.307 · 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 teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designBench or experimental
Domainnot available
GenreEmpirical

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

Citations38
Published2021
Admission routes1
Has abstractyes

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