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Record W2810872365 · doi:10.1002/ajmg.a.40359

Variance of IQ is partially dependent on deletion type among 1,427 22q11.2 deletion syndrome subjects

2018· article· en· W2810872365 on OpenAlexafffund
Yingjie Zhao, Tingwei Guo, Ania Fiksinski, Elemi Breetvelt, Donna M. McDonald‐McGinn, T. Blaine Crowley, Alexander Diacou, Maude Schneider, Stéphan Eliez, Ann Swillen, Jeroen Breckpot, Joris Vermeesch, Eva W.C. Chow, Doron Gothelf, Sasja N. Duijff, Rens Evers, Thérèse A. van Amelsvoort, Marianne B. M. van den Bree, Michael J. Owen, Maria Niarchou, Carrie E. Bearden, Claudia Ornstein, Maria Pontillo, Antonino Buzzanca, Stefano Vicari, Marco Armando, Kieran C. Murphy, Clodagh M. Murphy, Sixto García‐Miñáur, Nicole Philip, Linda Campbell, Jaume Morey‐Cañellas, Jasna Raventos, Jordi Rosell, Damián Heine‐Suñer, Robert J. Shprintzen, Raquel E. Gur, Elaine H. Zackai, Beverly S. Emanuel, Tao Wang, Wendy R. Kates, Anne S. Bassett, Jacob Vorstman, Bernice E. Morrow

Bibliographic record

VenueAmerican Journal of Medical Genetics Part A · 2018
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicCongenital heart defects research
Canadian institutionsToronto General HospitalUniversity Health NetworkUniversity of TorontoCentre for Addiction and Mental HealthHospital for Sick Children
FundersEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentNational Institute of General Medical SciencesInstitute of Genetics and Genomics of GenevaNational Science FoundationAmerican Heart AssociationCanadian Institutes of Health ResearchUniversity of TorontoFonds Wetenschappelijk OnderzoekMedical Research CouncilNational Institute of Mental HealthNational Heart, Lung, and Blood InstituteInstitute of GeneticsNational Institutes of HealthSchweizerischer Nationalfonds zur Förderung der Wissenschaftlichen ForschungChildren's Hospital of Philadelphia
KeywordsIntelligence quotientHaploinsufficiencyDeletion syndromeGeneticsPopulationDiGeorge syndromeAllelePsychologyBiologyMedicineGenePhenotypePsychiatryCognition

Abstract

fetched live from OpenAlex

The 22q11.2 deletion syndrome is caused by non-allelic homologous recombination events during meiosis between low copy repeats (LCR22) termed A, B, C, and D. Most patients have a typical LCR22A-D (AD) deletion of 3 million base pairs (Mb). In this report, we evaluated IQ scores in 1,478 subjects with 22q11.2DS. The mean of full scale IQ, verbal IQ, and performance IQ scores in our cohort were 72.41 (standard deviation-SD of 13.72), 75.91(SD of 14.46), and 73.01(SD of 13.71), respectively. To investigate whether IQ scores are associated with deletion size, we examined individuals with the 3 Mb, AD (n = 1,353) and nested 1.5 Mb, AB (n = 74) deletions, since they comprised the largest subgroups. We found that full scale IQ was decreased by 6.25 points (p = .002), verbal IQ was decreased by 8.17 points (p = .0002) and performance IQ was decreased by 4.03 points (p = .028) in subjects with the AD versus AB deletion. Thus, individuals with the smaller, 1.5 Mb AB deletion have modestly higher IQ scores than those with the larger, 3 Mb AD deletion. Overall, the deletion of genes in the AB region largely explains the observed low IQ in the 22q11.2DS population. However, our results also indicate that haploinsufficiency of genes in the LCR22B-D region (BD) exert an additional negative impact on IQ. Furthermore, we did not find evidence of a confounding effect of severe congenital heart disease on IQ scores in our cohort.

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)0.001
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.449
Threshold uncertainty score0.591

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.001
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 head0.014
GPT teacher head0.309
Teacher spread0.295 · 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 designBench or experimental
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

Citations52
Published2018
Admission routes2
Has abstractyes

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