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Genome-Wide Association Transethnic Meta-Analyses Identifies Novel Associations Regulating Coagulation Factor VIII and von Willebrand Factor Plasma Levels

2019· review· en· W2901097982 on OpenAlex
Maria Sabater‐Lleal, Jennifer E. Huffman, Paul S. de Vries, Jonathan Marten, Michael A. Mastrangelo, Ci Song, Nathan Pankratz, Cavin Ward‐Caviness, Lisa R. Yanek, Stella Trompet, Graciela E. Delgado, Xiuqing Guo, Traci M. Bartz, Ángel Martínez-Pérez, Marine Germain, Hugoline G. de Haan, Ayse Bilge Ozel, Ozren Polašek, Albert V. Smith, John D. Eicher, Alex P. Reiner, Weihong Tang, Neil M Davies, David J. Stott, Jerome I. Rotter, Geoffrey H. Tofler, Eric Boerwinkle, Moniek P.M. de Maat, Marcus E. Kleber, Paul Welsh, Jennifer A. Brody, Ming‐Huei Chen, Dhananjay Vaidya, José Manuel Soria, Pierre Suchon, Astrid van Hylckama Vlieg, Karl C. Desch, Ivana Kolčić, Peter K. Joshi, Lenore J. Launer, Tamara B. Harris, Harry Campbell, Igor Rudan, Diane M. Becker, Jun Z. Li, Fernando Rivadeneira, André G. Uitterlinden, Albert Hofman, Oscar H. Franco, Mary Cushman, Bruce M. Psaty, Pierre‐Emmanuel Morange, Barbara McKnight, Michael Chong, Israel Fernández‐Cadenas, Jonathan Rosand, Vilmundur Guðnason, James F. Wilson, Caroline Hayward, David Ginsburg, Myriam Fornage, Frits R. Rosendaal, Juan Carlos Souto, Lewis C. Becker, Nancy S. Jenny, Winfried März, J. Wouter Jukema, Abbas Dehghan, David‐Alexandre Trégouët, Alanna C. Morrison, Andrew D. Johnson, Christopher J. O’Donnell, David P. Strachan, Charles J. Lowenstein, Nicholas L. Smith

Why this work is in the frame

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

affAt least one author lists a Canadian institution in the pinned OpenAlex snapshot.

Bibliographic record

VenueCirculation · 2019
Typereview
Languageen
FieldMedicine
TopicPlatelet Disorders and Treatments
Canadian institutionsPopulation Health Research InstituteCentre for Global Health Research
FundersNational Heart, Lung, and Blood InstituteNational Institute of Neurological Disorders and StrokeMedical Research CouncilNational Institute on AgingNational Institutes of HealthEuropean Hematology Association
KeywordsMendelian randomizationVon Willebrand factorGenome-wide association studyMedicineGenetic associationVon Willebrand diseaseInternal medicineGeneticsSingle-nucleotide polymorphismGeneImmunologyBioinformaticsGenotypeBiologyPlateletGenetic variants

Abstract

fetched live from OpenAlex

BACKGROUND: Factor VIII (FVIII) and its carrier protein von Willebrand factor (VWF) are associated with risk of arterial and venous thrombosis and with hemorrhagic disorders. We aimed to identify and functionally test novel genetic associations regulating plasma FVIII and VWF. METHODS: We meta-analyzed genome-wide association results from 46 354 individuals of European, African, East Asian, and Hispanic ancestry. All studies performed linear regression analysis using an additive genetic model and associated ≈35 million imputed variants with natural log-transformed phenotype levels. In vitro gene silencing in cultured endothelial cells was performed for candidate genes to provide additional evidence on association and function. Two-sample Mendelian randomization analyses were applied to test the causal role of FVIII and VWF plasma levels on the risk of arterial and venous thrombotic events. RESULTS: ) associations, 7 with FVIII levels ( FCHO2/TMEM171/TNPO1, HLA, SOX17/RP1, LINC00583/NFIB, RAB5C-KAT2A, RPL3/TAB1/SYNGR1, and ARSA) and 11 with VWF levels ( PDHB/PXK/KCTD6, SLC39A8, FCHO2/TMEM171/TNPO1, HLA, GIMAP7/GIMAP4, OR13C5/NIPSNAP, DAB2IP, C2CD4B, RAB5C-KAT2A, TAB1/SYNGR1, and ARSA), beyond 10 previously reported associations with these phenotypes. Functional validation provided further evidence of association for all loci on VWF except ARSA and DAB2IP. Mendelian randomization suggested causal effects of plasma FVIII activity levels on venous thrombosis and coronary artery disease risk and plasma VWF levels on ischemic stroke risk. CONCLUSIONS: The meta-analysis identified 13 novel genetic loci regulating FVIII and VWF plasma levels, 10 of which we validated functionally. We provide some evidence for a causal role of these proteins in thrombotic events.

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.

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)0.000
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: none
GenreCandidate signal: Review · Consensus signal: none
Teacher disagreement score0.711
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0030.001
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.000
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 head0.276
GPT teacher head0.394
Teacher spread0.118 · 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