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Record W4414457230 · doi:10.1177/08830738251377579

Epilepsy due to a <i>MED25</i> Homozygous Pathogenic Founder Variant

2025· article· en· W4414457230 on OpenAlexafffundabout
Andy Cheuk‐Him Ng, Sabrina D’Alfonso, A. Micheil Innes, Morris H. Scantlebury

Bibliographic record

VenueJournal of Child Neurology · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomics and Rare Diseases
Canadian institutionsStollery Children's HospitalAlberta Children's HospitalUniversity of CalgaryUniversity of Alberta
FundersAlberta Children's Hospital Research Institute
KeywordsEpilepsyPhenotypeContext (archaeology)Intellectual disabilityFounder effectSeizure DisordersClinical significance

Abstract

fetched live from OpenAlex

Previous reports have described a spectrum of clinical phenotypes in patients with MED25 pathogenic variants. One specific phenotype is the Basel-Vanagaite-Smirin-Yosef syndrome, which is a rare, autosomal recessive disorder characterized by a wide range of symptoms including eye abnormalities, cardiac abnormalities, palatal abnormalities, intellectual disability, and epilepsy. Although epilepsy appears to be common, the electroclinical phenotypes of patients with Basel-Vanagaite-Smirin-Yosef syndrome have not been well described. Here, we report 3 Basel-Vanagaite-Smirin-Yosef syndrome patients from 2 families of Lebanese descent with a homozygous MED25 founder variant from Alberta Children's Hospital, Calgary, Alberta. We describe in detail electroclinical phenotypes in each of these 3 patients. These patients developed seizures with onset between 2 and 3 years of age, multifocal and generalized discharges, as well as photoparoxysmal responses on electroencephalogram. We review the relevance of these epilepsy findings in the context of other published reports of Basel-Vanagaite-Smirin-Yosef syndrome.

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)0.000
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.014
Threshold uncertainty score0.028

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.000
Science and technology studies0.0010.001
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 head0.003
GPT teacher head0.217
Teacher spread0.214 · 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 designCase report
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
Published2025
Admission routes3
Has abstractyes

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