MétaCan
Menu
Back to cohort
Record W1996832702 · doi:10.1002/mus.21222

Homozygous contiguous gene deletion of 13q12 causing LGMD2C and ARSACS in the same patient

2009· article· en· W1996832702 on OpenAlexaffabout
Hugh J. McMillan, Melissa T. Carter, Pierre Jacob, Eoghan Laffan, Michael D. O’Connor, Kym M. Boycott

Bibliographic record

VenueMuscle & Nerve · 2009
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsOttawa HospitalChildren's Hospital of Eastern Ontario
Fundersnot available
KeywordsAtaxiaSpasticityMuscular dystrophyGeneticsGeneMicroarray analysis techniquesMedicineSpasticBiologyGene expressionNeurosciencePhysical medicine and rehabilitation

Abstract

fetched live from OpenAlex

We describe a 10-year-old girl with limb-girdle muscular dystrophy type 2C (LGMD2C, gamma-sarcoglycan deficiency) with additional features that include progressive lower limb spasticity, peripheral neuropathy, and ataxia. The gene for LGMD2C lies in close approximation to the gene for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS) on chromosome 13q12. The clinical presentation was suspicious for a genomic rearrangement affecting the expression of both genes. Using chromosomal microarray analysis, a homozygous deletion that encompassed the genes for both disorders was identified. This is the first report of a patient with both neurological diseases, and this case illustrates the clinical utility of microarray technology in the investigation of patients with unusual presentations.

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)0.000
metaresearch head score (Gemma)0.001
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: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0010.001
Science and technology studies0.0010.001
Scholarly communication0.0010.001
Open science0.0010.001
Research integrity0.0020.001
Insufficient payload (model declined to judge)0.0020.001

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 head0.249
Teacher spread0.222 · 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

Citations20
Published2009
Admission routes2
Has abstractyes

Explore more

Same venueMuscle & NerveSame topicGenetic Neurodegenerative DiseasesFrench-language works237,207