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Record W4389150626 · doi:10.1136/jnnp-2023-abn.248

Assessment of FTLD genes and C9orf72 repeat length in atypical Parkinsonism

2023· article· en· W4389150626 on OpenAlexaff
Vaughan David, Real Raquel, Jabbari Edwin, Wu Lesley, Jaunmuktane Zane, Warner Tom, Tucci Arianna, Rowe James, Rohrer Jonathon, Morris Huw

Bibliographic record

Venuenot available
Typearticle
Languageen
FieldMedicine
TopicParkinson's Disease Mechanisms and Treatments
Canadian institutionsCentre for Movement Disorders
Fundersnot available
KeywordsC9orf72Frontotemporal lobar degenerationParkinsonismCorticobasal degenerationGeneticsFamily historyBiologyGenePhenotypeMutationTrinucleotide repeat expansionAlleleMedicineDementiaFrontotemporal dementiaPathologyDiseaseProgressive supranuclear palsyInternal medicineAtrophy

Abstract

fetched live from OpenAlex

There is clinical, genetic and pathological overlap between atypical parkinsonism (APS) and frontotem- poral lobar degeneration (FTLD). Some patients diagnosed with APS carry pathogenic mutations in FTLD genes, including C9orf72. Unaffected individuals usually have less than 8 hexanucleotide repeats (HR) in C9orf72 and >60 HRs is defined as pathogenic for FTD/ALS. Intermediate expansions are reported to be a risk factor for corticobasal degeneration (CBD). We are evaluating variation in FTLD genes in a large multicentre UK APS study (PROSPECT) and Brain Bank (BB) patients with APS, and controls. We have recruited 508 PROSPECT and 392 BB participants. We are screening these samples using repeat-primed PCR for C9orf72 repeat expansions and analysing whole genome sequence data. To date, we have identified four cases with pathogenic mutations in FTLD genes (MAPT, TBK1 and GRN) and one atypical corticobasal syndrome case with an intermediate expansion in C9orf72. Mutation carriers have similar phenotypes to sporadic APS, but usually with a younger age at onset and a positive family history. Analysis of FTLD genes is important in APS, especially in younger patients with a family history.

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)0.002
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.007

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.002
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0020.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.000
Insufficient payload (model declined to judge)0.0020.000

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 head0.028
GPT teacher head0.323
Teacher spread0.295 · 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 designObservational
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

Citations0
Published2023
Admission routes1
Has abstractyes

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