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Record W4405151222 · doi:10.1177/22143602241290353

International collaboration to improve knowledge on myotonic dystrophy type 2

2024· article· en· W4405151222 on OpenAlexaff
Stojan Perić, Vukan Ivanović, E. Ashley, Belen Esparis, Craig Campbell, Stephan Wenninger, Darren G. Monckton, Chiara Marini‐Bettolo, Helen Walker, Stanislav Voháňka, Kleed Cumming, Anna Łusakowska, Victoria Hodgkinson, Marjan Cosyns, Miriam Rodrigues, Eppie M. Yiu, Radim Mazanec, Tanya Stevenson, Anna Kostera‐Pruszczyk, Lawrence Korngut, Marlène Jagut, Benedikt Schoser, Robin Forbes, A. Poll, Richard Roxburgh

Bibliographic record

VenueJournal of Neuromuscular Diseases · 2024
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsHotchkiss Brain InstituteUniversity of CalgaryWestern University
Fundersnot available
KeywordsMyotonic dystrophyMedicineType (biology)PediatricsInternal medicineBiology

Abstract

fetched live from OpenAlex

BACKGROUND: The TREAT-NMD Global Registry Network is a global collaboration of neuromuscular disease registries, including myotonic dystrophy type 2 (DM2), which aims to facilitate collaborative research and clinical trials. OBJECTIVES: This study aimed to assess DM2 patients included in the network, and to analyse their socio-demographic and clinical features. METHODS: Data were collected through email surveys sent to 16 TREAT-NMD myotonic dystrophy core member registries. 10 registries enrolled DM2 patients. RESULTS: The total number of DM2 cases was 1,720, with the Czech, German, and USA registries enrolling the most patients (445, 430, and 339 cases, respectively). The highest rates were seen in Czechia and Serbia (4.2 and 2.0 registered per 100,000 population, respectively). High DM2:DM1 ratios were seen in Central Europe. The median age at registry entry was 51 years. Symptom onset occurred before age 20 in 14% of cases. One fifth of patients used an assistive device to walk, and 4% were non-ambulatory. Insertion of a pacemaker or implantable cardioverter-defibrillator was reported in 4% of subjects, while 7% used non-invasive ventilation. CONCLUSIONS: This represents the largest DM2 cohort assembled to date, providing demographic and clinical data for future research and trial recruitment, illustrating TREAT-NMD's international reach and the importance of capturing DM2 data.

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)0.039
metaresearch head score (Gemma)0.055
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: Empirical · Consensus signal: none
Teacher disagreement score0.039
Threshold uncertainty score0.207

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0390.055
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0050.006
Science and technology studies0.0010.001
Scholarly communication0.0030.003
Open science0.0010.007
Research integrity0.0020.003
Insufficient payload (model declined to judge)0.0170.003

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 head0.018
GPT teacher head0.309
Teacher spread0.291 · 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
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

Citations2
Published2024
Admission routes1
Has abstractyes

Explore more

Same venueJournal of Neuromuscular DiseasesSame topicGenetic Neurodegenerative DiseasesFrench-language works237,207