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Record W4318706872 · doi:10.1101/2023.01.28.23285147

Large scale genome-wide association analyses identify novel genetic loci and mechanisms in hypertrophic cardiomyopathy

2023· preprint· en· W4318706872 on OpenAlexafffund
Rafik Tadros, Sean Zheng, Christopher Grace, Paloma Jordà, Catherine Francis, Sean J. Jurgens, Kate Thomson, Andrew R. Harper, Elizabeth Ormondroyd, Dominique M West, Xiao Yun Xu, Pantazis Theotokis, Rachel Buchan, Kathryn A. McGurk, Francesco Mazzarotto, Beatrice Boschi, Elisabetta Pelo, Michael Lee, Michela Noseda, Amanda Varnava, Alexa M.C. Vermeer, Roddy Walsh, Ahmad S. Amin, Marjon A. van Slegtenhorst, Nicole M. Roslin, Lisa J. Strug, Erika Salvi, Chiara Lanzani, Antonio de Marvao, Jason D. Roberts, Maxime Tremblay‐Gravel, Geneviève Giraldeau, Julia Cadrin‐Tourigny, Philippe L. L’Allier, Patrick Garceau, Mario Talajic, Yigal M. Pinto, Harry Rakowski, Antonis Pantazis, John Baksi, Brian P. Halliday, Sanjay Prasad, Paul J.R. Barton, Declan P. O’Regan, Stuart A. Cook, Rudolf A. de Boer, Imke Christiaans, Michelle Michels, Christopher M. Kramer, Carolyn Y. Ho, Stefan Neubauer, Paul M. Matthews, Arthur A.M. Wilde, Jean‐Claude Tardif, Iacopo Olivotto, Arnon Adler, Anuj Goel, James S. Ware, Connie R. Bezzina, Hugh Watkins

Bibliographic record

VenuemedRxiv · 2023
Typepreprint
Languageen
FieldMedicine
TopicCardiomyopathy and Myosin Studies
Canadian institutionsWestern UniversityUniversity Health NetworkSickKids FoundationUniversity of TorontoUniversité de MontréalHospital for Sick ChildrenMontreal Heart Institute
FundersAmsterdam Cardiovascular Sciences, Amsterdam University Medical CentersNational Institutes of HealthNational Heart, Lung, and Blood InstituteUK Dementia Research InstituteMedical Research CouncilInstitut de Cardiologie de MontréalFondation Institut de Cardiologie de MontréalAmsterdam University Medical CentersFondation LeducqDeutsches Zentrum für Herz-KreislaufforschungEuropean CommissionCanadian Institutes of Health ResearchImperial College LondonNational Institute for Health and Care ResearchZonMwNovo Nordisk FondenIsrael Cancer Research FundWellcome TrustAlzheimer's SocietyBritish Heart Foundation
KeywordsHypertrophic cardiomyopathyGenome-wide association studyMendelian randomizationGenetic associationDiseaseBiologyGeneticsCardiomyopathyInternal medicineGeneMedicineGenetic variantsGenotypeSingle-nucleotide polymorphismHeart failure

Abstract

fetched live from OpenAlex

Hypertrophic cardiomyopathy (HCM) is an important cause of morbidity and mortality with both monogenic and polygenic components. We here report results from the largest HCM genome-wide association study (GWAS) and multi-trait analysis (MTAG) including 5,900 HCM cases, 68,359 controls, and 36,083 UK Biobank (UKB) participants with cardiac magnetic resonance (CMR) imaging. We identified a total of 70 loci (50 novel) associated with HCM, and 62 loci (32 novel) as sociated with relevant left ventricular (LV) structural or functional traits. Amongst the common variant HCM loci, we identify a novel HCM disease gene, SVIL , which encodes the actin-binding protein supervillin, showing that rare truncating SVIL variants cause HCM. Mendelian randomization analyses support a causal role of increased LV contractility in both obstructive and non-obstructive forms of HCM, suggesting common disease mechanisms and anticipating shared response to therapy. Taken together, the findings significantly increase our understanding of the genetic basis and molecular mechanisms of HCM, with potential implications for disease management.

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)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.011
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.001
Research integrity0.0000.001
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 head0.047
GPT teacher head0.314
Teacher spread0.267 · 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.

Study designObservational
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

Citations12
Published2023
Admission routes2
Has abstractyes

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