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Record W2165672021 · doi:10.1136/jmg.2003.011429

Missense mutations in the β strands of the single A-domain of matrilin-3 result in multiple epiphyseal dysplasia

2004· letter· en· W2165672021 on OpenAlexaff
G C Jackson

Bibliographic record

VenueJournal of Medical Genetics · 2004
Typeletter
Languageen
FieldMedicine
TopicOsteoarthritis Treatment and Mechanisms
Canadian institutionsHospital for Sick Children
FundersFundacja na rzecz Nauki Polskiej
KeywordsMissense mutationCartilage oligomeric matrix proteinOsteochondrodysplasiaPhenotypeGeneGeneticsExtracellular matrixMutationDysplasiaBiologyVariable ExpressionCartilageMedicinePathologyAnatomyOsteoarthritis

Abstract

fetched live from OpenAlex

Key pointsN Four novel mutations (A219D, I192N, T120M, and E134K) and one recurrent mutation (R121W) were identified in MATN3 in seven families with multiple epiphyseal dysplasia.N All of the disease causing mutations are located within the b sheet of the single A-domain of matrilin-3, which strongly suggests that they have a deleterious effect on the folding and/or function of matrilin-3. NThe R121W mutation is associated with a marked interfamilial variability in the radiographic phenotype of patients, suggesting that other genetic factors are acting to modify the severity of the multiple epiphyseal dysplasia phenotype in these patients.52 www.jmedgenet.com

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.005
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: none
Teacher disagreement score0.010
Threshold uncertainty score0.018

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.005
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0010.000
Science and technology studies0.0010.001
Scholarly communication0.0010.001
Open science0.0010.000
Research integrity0.0100.005
Insufficient payload (model declined to judge)0.0050.003

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 head0.027
GPT teacher head0.281
Teacher spread0.253 · 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 source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designCase report
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

Citations67
Published2004
Admission routes1
Has abstractyes

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