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Record W7117325868 · doi:10.1177/22143602251399244

A toolkit for new facioscapulohumeral muscular dystrophy trial sites

2025· article· en· W7117325868 on OpenAlexaff
Joost Kools, Lawrence Korngut, Janet Petrillo Ballantyne, Irene J. Roozen, Ria de Haas, Amanda Baracho Trindade Hill, Teresinha Evangelista, Valeria Sansone, Richard Roxburgh, Hanns Lochmüller, J. Statland, N. Johnson, Nicol C. Voermans

Bibliographic record

VenueJournal of Neuromuscular Diseases · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicMuscle Physiology and Disorders
Canadian institutionsChildren's Hospital of Eastern OntarioHotchkiss Brain InstituteUniversity of Calgary
Fundersnot available
KeywordsFacioscapulohumeral muscular dystrophyClinical trialReimbursementMuscular dystrophyDiseaseDrug developmentDrug trialMEDLINE

Abstract

fetched live from OpenAlex

Numerous potential treatments are being developed for facioscapulohumeral muscular dystrophy (FSHD). Project Mercury was initiated to overcome challenges that could slow or prevent effective therapies from widespread availability to patients. It is important that upcoming trials include trial sites from different countries. We share our lessons learnt in clinical trials to assist inexperienced sites to become eligible for upcoming clinical trials. To become an eligible site, several key elements need to be in place such as personnel, facilities, and accessible patient populations. Clinical trial networks, patient advocacy groups and patient registries can support new sites in establishing these elements. As the preparation, execution and close-out of clinical trials generally involve the same steps every time, it is recommended to create and follow a trial roadmap. Most clinical trials are sponsor-initiated and involve working closely with the sponsor and vendors. It is therefore important to understand each other perspectives and goals for each trial. Once a drug receives regulatory approval and becomes available for market use new challenges arise such as patient reimbursement and phase 4 surveillance of the patients. In summary, we are at a pivotal time for FSHD and other rare neuromuscular disorders with the development of new disease modifying therapies. It is vital that as many sites as possible can participate in upcoming trials.

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 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)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.726
Threshold uncertainty score0.659

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.001
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 head0.011
GPT teacher head0.278
Teacher spread0.268 · 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 teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designBench or experimental
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

Citations1
Published2025
Admission routes1
Has abstractyes

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