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Record W4307657653 · doi:10.1038/s41588-022-01203-y

Statistical and functional convergence of common and rare genetic influences on autism at chromosome 16p

2022· article· en· W4307657653 on OpenAlexaff
Daniel J. Weiner, Emi Ling, Serkan Erdin, Derek J.C. Tai, Rachita Yadav, Jakob Grove, Jack Fu, Ajay Nadig, Caitlin E. Carey, Nikolas Baya, Jonas Bybjerg‐Grauholm, Preben Bo Mortensen, Thomas Werge, Ditte Demontis, Ole Mors, Merete Nordentoft, Thomas D. Als, Marie Bækvad‐Hansen, Anders Rosengren, Alexandra Havdahl, Anne Hedemand, Aarno Palotie, Aravinda Chakravarti, Dan E. Arking, Arvis Sulovari, Anna Starnawska, Bhooma Thiruvahindrapuram, Christiaan de Leeuw, Christine Ladd‐Acosta, Celia van der Merwe, Bernie Devlin, Edwin H. Cook, Evan E. Eichler, Elisabeth Corfield, Gwen Dieleman, Gerard D. Schellenberg, Håkon Håkonarson, Hilary Coon, Isabel Dziobek, Jacob Vorstman, Jessica B. Girault, James S. Sutcliffe, Jinjie Duan, John I. Nürnberger, Joachim Hallmayer, Joseph D. Buxbaum, Joseph Piven, Lauren A. Weiss, Lea K. Davis, Magdalena Janecka, Manuel Mattheisen, Matthew W. State, Michael Gill, Mark J. Daly, Mohammed Uddin, Ole A. Andreassen, Péter Szatmári, Phil Hyoun Lee, Richard Anney, Stephan Ripke, Kyle Satterstrom, Susan L. Santangelo, Susan S. Kuo, Ludger Tebartz van Elst, Thomas Rolland, Thomas Bougeron, Tinca J. C. Polderman, Tychele N. Turner, Jack F. G. Underwood, Veera Manikandan, Vamsee Pillalamarri, Varun Warrier, Alexandra Philipsen, Andreas Reif, Anke Hinney, Bru Cormand, Claiton H.D. Bau, Diego Luiz Rovaris, Edmund Sonuga‐Barke, Elizabeth C. Corfield, Eugênio H. Grevet, Giovanni Abrahão Salum, Henrik Larsson, Jan Buitelaar, Jan Haavik, James J. McGough, Jonna Kuntsi, Josephine Elia, Klaus‐Peter Lesch, Marieke Klein, Mark A. Bellgrove, Martin Tesli, Patrick W. L. Leung, Pedro Mário Pan, Søren Dalsgaard, Sandra K. Loo, Sarah E. Medland, Stephen V. Faraone, Ted Reichborn‐Kjennerud, Tobias Banaschewski, Ziarih Hawi, Sabina Berretta, Evan Z. Macosko, Jonathan Sebat, Luke J. O’Connor, David M. Hougaard, Anders D. Børglum, Michael E. Talkowski, Steven A. McCarroll, Elise Robinson

Bibliographic record

VenueNature Genetics · 2022
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomic variations and chromosomal abnormalities
Canadian institutionsUniversity of TorontoSickKids FoundationHospital for Sick Children
FundersNational Human Genome Research InstituteNovo Nordisk FondenNational Institute of Mental HealthEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentAarhus UniversitetNational Institute of Neurological Disorders and StrokeNational Institute of General Medical SciencesNovo NordiskNational Cancer InstituteU.S. Department of Health and Human ServicesNational Institutes of HealthU.S. National Library of MedicineH. Lundbeck A/SLundbeckfondenSimons Foundation Autism Research Initiative
KeywordsBiologyAutismGeneticsChromosomeEvolutionary biologyConvergence (economics)Computational biologyGeneDevelopmental psychology

Abstract

fetched live from OpenAlex

The canonical paradigm for converting genetic association to mechanism involves iteratively mapping individual associations to the proximal genes through which they act. In contrast, in the present study we demonstrate the feasibility of extracting biological insights from a very large region of the genome and leverage this strategy to study the genetic influences on autism. Using a new statistical approach, we identified the 33-Mb p-arm of chromosome 16 (16p) as harboring the greatest excess of autism's common polygenic influences. The region also includes the mechanistically cryptic and autism-associated 16p11.2 copy number variant. Analysis of RNA-sequencing data revealed that both the common polygenic influences within 16p and the 16p11.2 deletion were associated with decreased average gene expression across 16p. The transcriptional effects of the rare deletion and diffuse common variation were correlated at the level of individual genes and analysis of Hi-C data revealed patterns of chromatin contact that may explain this transcriptional convergence. These results reflect a new approach for extracting biological insight from genetic association data and suggest convergence of common and rare genetic influences on autism at 16p.

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)0.000
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: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.272
Threshold uncertainty score0.491

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
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 head0.006
GPT teacher head0.222
Teacher spread0.217 · 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 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

Citations55
Published2022
Admission routes1
Has abstractyes

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