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Record W4311448329 · doi:10.1056/nejmoa2207406

Deep Intronic <i>FGF14</i> GAA Repeat Expansion in Late-Onset Cerebellar Ataxia

2022· article· en· W4311448329 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.
fundA Canadian funder is recorded on the work.
aboutThe title or abstract carries a Canadian signal from the geographic lexicon.

Bibliographic record

VenueNew England Journal of Medicine · 2022
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsUniversity of OttawaUniversité de SherbrookeMcGill Genome CentreUniversité de MontréalChildren's Hospital of Eastern OntarioCentre Hospitalier de l’Université de MontréalMcGill UniversityMontreal Neurological Institute and HospitalMontreal Heart Institute
FundersFondation Groupe MonacoNational Institute of Neurological Disorders and StrokeNational Health and Medical Research CouncilMedical Research CouncilMedizinischen Fakultät, Eberhard Karls Universität TübingenLeonard M. Miller School of MedicineNational Institutes of HealthFondation de l'Hôpital Général de MontréalUniversitat de BarcelonaElse Kröner-Fresenius-StiftungUniversity College London Hospitals NHS Foundation TrustEberhard Karls Universität TübingenInstitut National de la Santé et de la Recherche MédicaleEuropean CommissionNational Institute of Mental Health and NeurosciencesWellcome TrustUniversity College LondonCanadian Institutes of Health ResearchRoyal College of Physicians and Surgeons of CanadaMcGill UniversityDeutsche ForschungsgemeinschaftGenome CanadaBrain Research UKBundesministerium für Bildung und ForschungNational Institute for Health and Care ResearchChildren's Hospital of Eastern Ontario FoundationOntario GenomicsCanada Foundation for InnovationDeutsches Zentrum für Neurodegenerative ErkrankungenOntario Genomics InstituteGénome QuébecNational Institute of Mental HealthUniversität BaselAustralian GovernmentFondation du Grand défi Pierre LavoieUniversité de LorraineUniversity of Miami
KeywordsTrinucleotide repeat expansionOdds ratioAtaxiaBiologyInternal medicineGeneticsMedicineNeuroscienceGeneAllele

Abstract

fetched live from OpenAlex

BACKGROUND: The late-onset cerebellar ataxias (LOCAs) have largely resisted molecular diagnosis. METHODS: We sequenced the genomes of six persons with autosomal dominant LOCA who were members of three French Canadian families and identified a candidate pathogenic repeat expansion. We then tested for association between the repeat expansion and disease in two independent case-control series - one French Canadian (66 patients and 209 controls) and the other German (228 patients and 199 controls). We also genotyped the repeat in 20 Australian and 31 Indian index patients. We assayed gene and protein expression in two postmortem cerebellum specimens and two induced pluripotent stem-cell (iPSC)-derived motor-neuron cell lines. RESULTS: RNA and protein. CONCLUSIONS: was found to be associated with LOCA. (Funded by Fondation Groupe Monaco and others.).

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)0.001
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesInsufficient payload (model declined to judge)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.276
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
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 integrity0.0000.001
Insufficient payload (model declined to judge)0.0010.000

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 head0.020
GPT teacher head0.259
Teacher spread0.239 · 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