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Record W4317861410 · doi:10.1212/wnl.0000000000201612

Natural History Study of <i>STXBP1</i> -Developmental and Epileptic Encephalopathy Into Adulthood

2023· erratum· en· W4317861410 on OpenAlexfundno aff

Bibliographic record

VenueNeurology · 2023
Typeerratum
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetics and Neurodevelopmental Disorders
Canadian institutionsnot available
FundersUCB PharmaEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentNational Health and Medical Research CouncilNational Center for Advancing Translational SciencesMedical Research CouncilNovartis PharmaPerelman School of Medicine, University of PennsylvaniaIntellectual and Developmental Disabilities Research CenterNational Institutes of HealthH. Lundbeck A/SVlaamse regeringEisaiBundesministerium für Bildung und ForschungZynerba PharmaceuticalsNational Institute of Child Health and Human DevelopmentSanofiGW PharmaceuticalsArvelle TherapeuticsGlaxoSmithKlineLivaNovaDeutsche ForschungsgemeinschaftUltragenyx PharmaceuticalWellcome TrustZogenixAngelini PharmaMinistero dell’Istruzione, dell’Università e della RicercaCanadian Institutes of Health ResearchFonds Wetenschappelijk OnderzoekInstitute for Translational Medicine and TherapeuticsChildren's Hospital of PhiladelphiaUniversity of Pennsylvania
KeywordsNatural historyEpilepsyEncephalopathyYoung adultMedicineNeurosciencePsychologyPsychiatryGerontologyInternal medicine

Abstract

fetched live from OpenAlex

Background and ObjectivesPathogenic STXBP1 variants cause a severe early-onset developmental and epileptic encephalopathy (STXBP1-DEE).We aimed to investigate the natural history of STXBP1-DEE in adults focusing on seizure evolution, the presence of movement disorders, and the level of functional (in)dependence. MethodsIn this observational study, patients with a minimum age of 18 years carrying a (likely) pathogenic STXBP1 variant were recruited through medical genetics departments and epilepsy centers.Treating clinicians completed clinical questionnaires and performed semistructured video examinations while performing tasks from the (modified) Unified Parkinson Disease Rating Scale when possible.

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)0.000
metaresearch head score (Gemma)0.001
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: Other · Consensus signal: none
Teacher disagreement score0.002
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
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 integrity0.0000.000
Insufficient payload (model declined to judge)0.0020.001

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 head0.214
Teacher spread0.207 · 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
GenreOther

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
Published2023
Admission routes1
Has abstractyes

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