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Record W2985482928

Oculopharyngeal muscular dystrophy

2004· article· en· W2985482928 on OpenAlexaboutno aff
J. Andoni Urtizberea

Bibliographic record

Venuenot available
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicMuscle Physiology and Disorders
Canadian institutionsnot available
Fundersnot available
KeywordsOculopharyngeal muscular dystrophyMedicinePtosisDysphagiaMyopathyPharyngeal musclesMuscular dystrophyPopulationFacioscapulohumeral muscular dystrophyPathologyPediatricsAnatomySurgeryInternal medicine
DOInot available

Abstract

fetched live from OpenAlex

Keywords Disease name and synonyms Definition/Diagnosis criteria Excluded diseases Prevalence Clinical description Management including treatment Etiology / Heredity Diagnostic methods Genetic counseling Antenatal diagnosis Unresolved questions References Abstract Oculopharyngeal muscular dystrophy (OPMD) is an autosomal dominant form of late-onset slowly progressive myopathy characterized by eyelid ptosis, dysphagia and, sometimes by other cranial and limb-muscle involvement. Most early cases were reported in the French Canadian population but the disease was subsequently found to be ubiquitous. From a clinical perspective, this disorder presents in the fourth to the sixth decade with progressive often asymmetrical ptosis resulting in a compensating contraction of the frontalis muscle and therefore a suggestive posture with retroflexion of the neck. Dysphagia is also an early symptom and can lead to nasal regurgitation and severe episodes of aspiration if overlooked. Limb-girdle muscle weakness, especially in the pelvic girdle, is often noted but varies widely among individuals without any correlation with the severity of ptosis or dysphagia. Creatine kinase (CK) levels are slightly elevated and electromyogram (EMG) studies suggest a mild myopathic process. The best clue to the diagnosis, after the clinical distribution of muscle weakness, is the presence of intranuclear inclusions in the muscle fibers. Rimmed vacuoles are often seen but are less specific. Genetic studies are now available and can establish the molecular signature of the disease. A short GCG- triplet repeat expansion in the gene encoding the PABPN1 protein (on chromosome 14q) is pathognomonic of OPMD. However, the disease pathogenesis remains unclear. Supportive treatment is available and consists of eyelid surgery and myotomy of the cricopharyngeal muscle in carefully selected cases.

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)0.002
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: none
GenreCandidate signal: Other · Consensus signal: Other
Teacher disagreement score0.028
Threshold uncertainty score0.093

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0040.004
Science and technology studies0.0000.000
Scholarly communication0.0010.001
Open science0.0000.001
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0280.010

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 head0.004
GPT teacher head0.217
Teacher spread0.213 · 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 designNot applicable
Domainnot available
GenreOther

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

Citations3
Published2004
Admission routes1
Has abstractyes

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