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Record W4409951238 · doi:10.1186/s13023-025-03725-0

European experience of steroid therapy in children with developmental and epileptic encephalopathy with spike wave activation in sleep ((D)EE-SWAS)

2025· article· en· W4409951238 on OpenAlexaff
Dilan Canbay, Floor E. Jansen, Jan Schönberger, Victoria San Antonio‐Arce, Julia Jacobs, Kerstin Alexandra Klotz

Bibliographic record

VenueOrphanet Journal of Rare Diseases · 2025
Typearticle
Languageen
FieldMedicine
TopicEpilepsy research and treatment
Canadian institutionsAlberta Children's Hospital
Fundersnot available
KeywordsClobazamEpilepsyMedicinePsychiatryPediatricsPsychology

Abstract

fetched live from OpenAlex

BACKGROUND: Developmental and epileptic encephalopathy with spike wave activation in sleep (DEE-SWAS) and epileptic encephalopathy with spike wave activation in sleep (EE-SWAS) are rare but well-known childhood epileptic disorders. Steroids are one of the first line treatment options, but a variety of steroid regimens exists. The aim of this survey was to evaluate the practices of steroid therapy in the treatment of (D)EE-SWAS across European centers. METHODS: An online survey was conducted (via 'SurveyMonkey' Europe) among European epilepsy centers. Questions asked included: characteristics of replying center, applied definition of DEE-SWAS, existence of regional/national guidelines regarding diagnostic and therapeutic management. Particular attention was paid to the indication/contraindication of steroids and treatment regimens used. RESULTS: Responses were obtained from 60 centers across 18 countries. Only 15% of centers reported the availability of national guidelines for the management of (D)EE-SWAS. There were variations in definition of (D)EE-SWAS, with Spike-Wave- Index (SWI) > 85 (irrespective of cognitive status) and SWI > 50% with concurrent neurodevelopmental regression being the most prevalent, reported in 36% and 50%, respectively. Steroids and clobazam were considered the predominant treatment options, with the primary indication for steroids being neurodevelopmental arrest (52%) and failure of clobazam treatment (51%). Treatment goals of steroid treatment primarily focused on neurodevelopmental improvement (95%), and reduction of SWI (66%). Methylprednisolone and prednisone were the most frequently used steroids, although other steroid types were also reported. Pulse therapy was utilized exclusively in 47% of centers. The most commonly used steroid regimen was intravenous/oral methylprednisolone pulse therapy (20 mg/kg/day for 3 days, either monthly or weekly), although a broad variety of different regimens were reported. Criteria influencing decisions about steroid treatment were largely based on personal experience, with scientific publications playing a role in decision-making in only 14% of centers. CONCLUSION: Steroids are part of the first line therapy of (D)EE-SWAS across Europe, but heterogeneity in formulations, dosages, and regimens persists due to limited guideline availability. The absence of comparative studies and the discordant definitions of (D)EE-SWAS further hinder comparisons of treatment efficacy. We recommend that harmonizing steroid treatment strategies is imperative for optimizing (D)EE-SWAS management.

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)0.002
metaresearch head score (Gemma)0.007
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 score0.005
Threshold uncertainty score0.009

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0020.007
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.001
Science and technology studies0.0000.001
Scholarly communication0.0010.001
Open science0.0000.001
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0010.000

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 head0.254
Teacher spread0.243 · 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

Citations5
Published2025
Admission routes1
Has abstractyes

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