MétaCan
Menu
Back to cohort
Record W2794758161 · doi:10.1136/jmedgenet-2017-105222

Chitayat-Hall and Schaaf-Yang syndromes:a common aetiology: expanding the phenotype of <i>MAGEL2</i>-related disorders

2018· article· en· W2794758161 on OpenAlexafffund
Rebekah Jobling, Dimitri J. Stavropoulos, Christian R. Marshall, Cheryl Cytrynbaum, Michelle M. Axford, Vanessa Londero, Sharon Moalem, Jennifer Orr, Francis Rossignol, Fátima Lopes, Julie Gauthier, Nathalie Alos, Rosemarie Rupps, Margaret L. McKinnon, Shelin Adam, Małgorzata J.M. Nowaczyk, Susan Walker, Stephen W. Scherer, Christina Nassif, Fadi F. Hamdan, Cheri Deal, Jean‐François Soucy, Rosanna Weksberg, Patrick MacLeod, Jacques L. Michaud, David Chitayat

Bibliographic record

VenueJournal of Medical Genetics · 2018
Typearticle
Languageen
FieldMedicine
TopicNeurogenetic and Muscular Disorders Research
Canadian institutionsUniversity of VictoriaUniversité de MontréalMcMaster UniversityCentre Hospitalier Universitaire Sainte-JustineSickKids FoundationUniversity of TorontoUniversity of British ColumbiaHospital for Sick Children
FundersFundação para a Ciência e a TecnologiaCentre hospitalier universitaire Sainte-JustineHospital for Sick ChildrenUniversity of TorontoGlaxoSmithKline
KeywordsEtiologyExome sequencingPhenotypeMedicineGeneticsIntellectual disabilityExomeBioinformaticsGeneBiologyInternal medicine

Abstract

fetched live from OpenAlex

Background Chitayat-Hall syndrome, initially described in 1990, is a rare condition characterised by distal arthrogryposis, intellectual disability, dysmorphic features and hypopituitarism, in particular growth hormone deficiency. The genetic aetiology has not been identified. Methods and results We identified three unrelated families with a total of six affected patients with the clinical manifestations of Chitayat-Hall syndrome. Through whole exome or whole genome sequencing, pathogenic variants in the MAGEL2 gene were identified in all affected patients. All disease-causing sequence variants detected are predicted to result in a truncated protein, including one complex variant that comprised a deletion and inversion. Conclusions Chitayat-Hall syndrome is caused by pathogenic variants in MAGEL2 and shares a common aetiology with the recently described Schaaf-Yang syndrome. The phenotype of MAGEL2 -related disorders is expanded to include growth hormone deficiency as an important and treatable complication.

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: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.776
Threshold uncertainty score0.390

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 integrity0.0000.001
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 head0.022
GPT teacher head0.343
Teacher spread0.320 · 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

Citations38
Published2018
Admission routes2
Has abstractyes

Explore more

Same venueJournal of Medical GeneticsSame topicNeurogenetic and Muscular Disorders ResearchFrench-language works237,207