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Record W4310338995 · doi:10.1038/s41398-022-02250-z

Hypothesis-driven genome-wide association studies provide novel insights into genetics of reading disabilities

2022· review· en· W4310338995 on OpenAlexafffundabout
Kaitlyn M. Price, Karen Wigg, Else Eising, Yu Feng, Kirsten Blokland, Margaret Wilkinson, Elizabeth N. Kerr, Sharon Guger, Filippo Abbondanza, Andrea G. Allegrini, Till F. M. Andlauer, Timothy C. Bates, Manon Bernard, Milene Bonte, Dorret I. Boomsma, Thomas Bourgeron, Daniel Brandeis, Manuel Carreiras, Fabiola Ceroni, Valéria Csépe, Philip S. Dale, John C. DeFries, Peter F. de Jong, Jean‐François Démonet, Eveline L. de Zeeuw, Marie-Christine Franken, Clyde Francks, Margot Gerritse, Alessandro Gialluisi, Scott D. Gordon, Jeffrey R. Gruen, Marianna E. Hayiou‐Thomas, Juan Hernández, Jouke‐Jan Hottenga, Charles Hulme, Philip R. Jansen, Juha Kere, Tanner Koomar, Karin Landerl, Gabriel Leonard, Zhijie Liao, Michelle Luciano, Heikki Lyytinen, Nicholas G. Martin, Angela Martinelli, Urs Maurer, Jacob J. Michaelson, Nazanin Mirza‐Schreiber, Kristina Moll, Anthony P. Monaco, Angela Morgan, Bertram Müller‐Myhsok, Dianne F. Newbury, Markus M. Nöthen, Richard K. Olson, Silvia Paracchini, Tomáš Paus, Zdenka Pausová, Craig E. Pennell, Bruce F. Pennington, Robert Plomin, Franck Ramus, Sheena Reilly, Louis Richer, Kaili Rimfeld, Gerd Schulte‐Körne, Chin Yang Shapland, Nuala H. Simpson, Shelley D. Smith, Margaret J. Snowling, Beaté St Pourcain, John Stein, Joel B. Talcott, Henning Tiemeier, J. Bruce Tomblin, Dongnhu T. Truong, Elsje van Bergen, Marc P. van der Schroeff, Marjolein van Donkelaar, Ellen Verhoef, Carol A. Wang, Kate E. Watkins, Andrew Whitehouse, Erik G. Willcutt, Margaret J. Wright, Gu Zhu, Simon E. Fisher, Maureen W. Lovett, Lisa J. Strug, Cathy L. Barr

Bibliographic record

VenueTranslational Psychiatry · 2022
Typereview
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetics and Neurodevelopmental Disorders
Canadian institutionsUniversité du Québec à ChicoutimiUniversité de MontréalCentre Hospitalier Universitaire Sainte-JustinePublic Health OntarioHospital for Sick ChildrenUniversity Health NetworkUniversity of TorontoMcGill University
FundersCIHR Skin Research Training CentreNational Institute of Mental HealthNational Institute on AgingNational Health and Medical Research CouncilMedical Research CouncilCanadian Institutes of Health ResearchHeart And Stroke Foundation Of QuebecUniversité de ParisNational Institute on Drug AbuseGovernment of Western AustraliaHospital for Sick ChildrenFondazione Umberto VeronesiMax-Planck-GesellschaftEusko JaurlaritzaHeart and Stroke Foundation of CanadaUniversiteit van AmsterdamNational Institutes of HealthUniversity of Notre Dame AustraliaMurdoch UniversityEdith Cowan UniversityAgencia Estatal de InvestigaciónSchweizerischer Nationalfonds zur Förderung der Wissenschaftlichen ForschungNational Institute on Deafness and Other Communication DisordersCurtin University of TechnologyEuropean CommissionAustrian Science FundEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentZonMwRaine Medical Research FoundationNational Institute of Child Health and Human DevelopmentGovernment of CanadaUniversity of BristolManton FoundationUniversity of Notre DameAvera Institute for Human GeneticsNederlandse Organisatie voor Wetenschappelijk OnderzoekAustralian GovernmentAgence Nationale de la RechercheWellcome TrustWomen and Infants Research FoundationChildren's Hospital of PhiladelphiaNational Science Foundation
KeywordsGenome-wide association studySingle-nucleotide polymorphismGeneticsLocus (genetics)Genetic associationBiologyAutismPsychologyGenePsychiatryGenotype

Abstract

fetched live from OpenAlex

Abstract Reading Disability (RD) is often characterized by difficulties in the phonology of the language. While the molecular mechanisms underlying it are largely undetermined, loci are being revealed by genome-wide association studies (GWAS). In a previous GWAS for word reading (Price, 2020), we observed that top single-nucleotide polymorphisms (SNPs) were located near to or in genes involved in neuronal migration/axon guidance (NM/AG) or loci implicated in autism spectrum disorder (ASD). A prominent theory of RD etiology posits that it involves disturbed neuronal migration, while potential links between RD-ASD have not been extensively investigated. To improve power to identify associated loci, we up-weighted variants involved in NM/AG or ASD, separately, and performed a new Hypothesis-Driven (HD)–GWAS. The approach was applied to a Toronto RD sample and a meta-analysis of the GenLang Consortium. For the Toronto sample (n = 624), no SNPs reached significance; however, by gene-set analysis, the joint contribution of ASD-related genes passed the threshold (p~1.45 × 10–2, threshold = 2.5 × 10–2). For the GenLang Cohort (n = 26,558), SNPs in DOCK7 and CDH4 showed significant association for the NM/AG hypothesis (sFDR q = 1.02 × 10–2). To make the GenLang dataset more similar to Toronto, we repeated the analysis restricting to samples selected for reading/language deficits (n = 4152). In this GenLang selected subset, we found significant association for a locus intergenic between BTG3-C21orf91 for both hypotheses (sFDR q < 9.00 × 10–4). This study contributes candidate loci to the genetics of word reading. Data also suggest that, although different variants may be involved, alleles implicated in ASD risk may be found in the same genes as those implicated in word reading. This finding is limited to the Toronto sample suggesting that ascertainment influences genetic associations.

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.029
metaresearch head score (Gemma)0.054
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Systematic review · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: none
Teacher disagreement score0.029
Threshold uncertainty score0.156

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0290.054
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0020.008
Bibliometrics0.0030.003
Science and technology studies0.0010.001
Scholarly communication0.0030.001
Open science0.0020.002
Research integrity0.0020.003
Insufficient payload (model declined to judge)0.0110.001

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 head0.055
GPT teacher head0.305
Teacher spread0.251 · 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 source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designSystematic review
Domainnot available
GenreReview

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

Citations14
Published2022
Admission routes3
Has abstractyes

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