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Record W2755489603 · doi:10.1002/ajmg.a.38354

<i>HLX</i> is a candidate gene for a pattern of anomalies associated with congenital diaphragmatic hernia, short bowel, and asplenia

2017· article· en· W2755489603 on OpenAlexafffund
Sandra A. Farrell, Sandi Sodhi, Christian R. Marshall, Andrea Guerin, Anne Slavotinek, Tara Paton, Karen Chong, Wilma Sirkin, Stephen W. Scherer, Félix‐Antoine Bérubé‐Simard, Nicolas Pilon

Bibliographic record

VenueAmerican Journal of Medical Genetics Part A · 2017
Typearticle
Languageen
FieldMedicine
TopicCongenital Diaphragmatic Hernia Studies
Canadian institutionsUniversité du Québec à MontréalNorth York General HospitalKingston General HospitalMount Sinai HospitalUniversity of TorontoSickKids FoundationHospital for Sick ChildrenTrillium Health Centre
FundersUniversity of TorontoMcLaughlin Centre, University of Toronto
KeywordsAspleniaDiaphragmatic herniaHomeoboxMissense mutationBiologyCongenital diaphragmatic herniaGeneticsExome sequencingFetusAnatomyMedicineMutationGeneHerniaTranscription factorImmunologyPregnancySurgery

Abstract

fetched live from OpenAlex

Isolated congenital diaphragmatic hernia is often a sporadic event with a low recurrence risk. However, underlying genetic etiologies, such as chromosome anomalies or single gene disorders, are identified in a small number of individuals. We describe two fetuses with a unique pattern of multiple congenital anomalies, including diaphragmatic hernia, short bowel and asplenia, born to first-cousin parents. Whole exome sequencing showed that both were homozygous for a missense variant, c.950A>C, predicting p.Asp317Ala, in the H.20-Like Homeobox 1 (HLX1) gene. HLX is a homeobox transcription factor gene which is relatively conserved across species. Hlx homozygous null mice have a short bowel and reduced muscle cells in the diaphragm, closely resembling the anomalies in the two fetuses and we therefore suggest that the HLX mutation in this family could explain the fetal findings.

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: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.245
Threshold uncertainty score0.813

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.000
Science and technology studies0.0000.002
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 head0.018
GPT teacher head0.297
Teacher spread0.279 · 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

Citations12
Published2017
Admission routes2
Has abstractyes

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