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Record W2989237060 · doi:10.1002/gepi.22265

Powerful rare variant association testing in a copula‐based joint analysis of multiple phenotypes

2019· article· en· W2989237060 on OpenAlex
Stefan Konigorski, Yildiz E. Yilmaz, Jürgen Janke, Manuela M. Bergmann, Heiner Boeing, Tobias Pischon

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.
fundA Canadian funder is recorded on the work.

Bibliographic record

VenueGenetic Epidemiology · 2019
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetic Associations and Epidemiology
Canadian institutionsMemorial University of Newfoundland
FundersNatural Sciences and Engineering Research Council of Canada
KeywordsUnivariateCopula (linguistics)Genome-wide association studyGenetic associationMultivariate statisticsPhenotypeComputational biologyType I and type II errorsStatisticBiologyStatistical powerStatisticsTest statisticGeneticsStatistical hypothesis testingMathematicsSingle-nucleotide polymorphismEconometricsGenotypeGene

Abstract

fetched live from OpenAlex

Abstract In genetic association studies of rare variants, the low power of association tests is one of the main challenges. In this study, we propose a new single‐marker association test called C‐JAMP (Copula‐based Joint Analysis of Multiple Phenotypes), which is based on a joint model of multiple phenotypes given genetic markers and other covariates. We evaluated its performance and compared its empirical type I error and power with existing univariate and multivariate single‐marker and multi‐marker rare‐variant tests in extensive simulation studies. C‐JAMP yielded unbiased genetic effect estimates and valid type I errors with an adjusted test statistic. When strongly dependent traits were jointly analyzed, C‐JAMP had the highest power in all scenarios except when a high percentage of variants were causal with moderate/small effect sizes. When traits with weak or moderate dependence were analyzed, whether C‐JAMP or competing approaches had higher power depended on the effect size. When C‐JAMP was applied with a misspecified copula function, it still achieved high power in some of the scenarios considered. In a real‐data application, we analyzed sequencing data using C‐JAMP and performed the first genome‐wide association studies of high‐molecular‐weight and medium‐molecular‐weight adiponectin plasma concentrations. C‐JAMP identified 20 rare variants with p ‐values smaller than 10 −5 , while all other tests resulted in the identification of fewer variants with higher p ‐values. In summary, the results indicate that C‐JAMP is a powerful, flexible, and robust method for association studies, and we identified novel candidate markers for adiponectin. C‐JAMP is implemented as an R package and freely available from https://cran.r‐project.org/package=CJAMP .

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)0.003
metaresearch head score (Gemma)0.012
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMetaresearch
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.044
Threshold uncertainty score0.997

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0030.012
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.001
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 head0.030
GPT teacher head0.279
Teacher spread0.249 · 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