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Record W4412814844 · doi:10.14802/jmd.25163

Cerebellar Ataxia Due to FGF14 GAA Repeat Expansion: First Southeast Asian Case and Novel Neuroimaging Features

2025· article· en· W4412814844 on OpenAlex

Why this work is in the frame

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

affAt least one author lists a Canadian institution in the pinned OpenAlex snapshot.

Bibliographic record

VenueJournal of Movement Disorders · 2025
Typearticle
Languageen
FieldMedicine
TopicGlycogen Storage Diseases and Myoclonus
Canadian institutionsMcGill UniversityMontreal Neurological Institute and Hospital
Fundersnot available
KeywordsMedicineNeuroimagingAtaxiaNeuroscienceCerebellar ataxiaPsychiatry

Abstract

fetched live from OpenAlex

Trinucleotide (GAA) repeat expansion in FGF14 was recently discovered to explain 10-60% of undiagnosed cases of late-onset cerebellar ataxia (LOCA), in some cases displaying autosomal dominant inheritance. 1 To our knowledge, no cases have been reported in Southeast Asia (SEA), a region with diverse populations totalling >600 million people.Here, we report a Malaysian of Chinese ancestry who had LOCA and a large FGF14 repeat expansion of 336 GAA repeats.She had pronounced and selective bi-parietal lobe atrophy on brain imaging, which has not previously been reported.This report thus potentially expands the phenotype of GAA-FGF14-related neurodegeneration and its geographic distribution. Case ReportAn 82-year-old Chinese woman with no family history of neurological disorders presented to us in August 2020, aged 78 years, with the chief complaint of unsteady gait of several months duration.Other symptoms were clumsiness of the hands, intermittent slurring of speech, and "shakes" in the legs.She had been on betahistine for several years for vertigo.There were no REM sleep behaviour symptoms, urinary incontinence, or foot numbness.Her other medical issues were hypertension, hyperlipidaemia, and knee osteoarthritis.There was no history of significant alcohol intake.Examination revealed a moderately wide-based and shuffling, unsteady gait, with inability to perform tandem walk.Eye movements were normal, with no nystagmus.Speech was not dysarthric.There was a mild action tremor of the upper limbs.A mild resting tremor was observed in the legs intermittently, with slight cogwheel rigidity at the wrists and mild bradykinesia of upper limb movements.Halmagyi head thrust test was negative.

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.

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)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.255
Threshold uncertainty score0.691

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
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 head0.008
GPT teacher head0.252
Teacher spread0.244 · 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