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Record W3012538954 · doi:10.1002/ajmg.b.32785

Segregating patterns of copy number variations in extended autism spectrum disorder (<scp>ASD</scp>) pedigrees

2020· article· en· W3012538954 on OpenAlexafffund
Marc Woodbury‐Smith, Mehdi Zarrei, John Wei, Bhooma Thiruvahindrapuram, Irene O’Connor, Andrew D. Paterson, Ryan K. C. Yuen, Jila Dastan, Dimitri J. Stavropoulos, Jennifer Howe, Ann Thompson, Morgan Parlier, Bridget A. Fernandez, Joseph Piven, Evdokia Anagnostou, Stephen W. Scherer, Veronica J. Vieland, Péter Szatmári

Bibliographic record

VenueAmerican Journal of Medical Genetics Part B Neuropsychiatric Genetics · 2020
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomic variations and chromosomal abnormalities
Canadian institutionsCentre for Addiction and Mental HealthHolland Bloorview Kids Rehabilitation HospitalSt. John’s Health Sciences CentreHospital for Sick ChildrenPublic Health OntarioSickKids FoundationUniversity of TorontoMcMaster University
FundersNIH Clinical CenterCanadian Institutes of Health ResearchCanada Foundation for Innovation
KeywordsPedigree chartCopy-number variationAutism spectrum disorderAutismGeneticsPhenotypeHeritability of autismGene duplicationNeurodevelopmental disorderGenetic linkageBiologyGenePsychologyDevelopmental psychologyGenome

Abstract

fetched live from OpenAlex

Autism spectrum disorder (ASD) is a relatively common childhood onset neurodevelopmental disorder with a complex genetic etiology. While progress has been made in identifying the de novo mutational landscape of ASD, the genetic factors that underpin the ASD's tendency to run in families are not well understood. In this study, nine extended pedigrees each with three or more individuals with ASD, and others with a lesser autism phenotype, were phenotyped and genotyped in an attempt to identify heritable copy number variants (CNVs). Although these families have previously generated linkage signals, no rare CNV segregated with these signals in any family. A small number of clinically relevant CNVs were identified. Only one CNV was identified that segregated with ASD phenotype; namely, a duplication overlapping DLGAP2 in three male offspring each with an ASD diagnosis. This gene encodes a synaptic scaffolding protein, part of a group of proteins known to be pathologically implicated in ASD. On the whole, however, the heritable nature of ASD in the families studied remains poorly understood.

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 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 score0.074
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
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 science0.0010.000
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 head0.008
GPT teacher head0.244
Teacher spread0.236 · 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

Citations11
Published2020
Admission routes2
Has abstractyes

Explore more

Same venueAmerican Journal of Medical Genetics Part B Neuropsychiatric GeneticsSame topicGenomic variations and chromosomal abnormalitiesFrench-language works237,207