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Antecollis and Levodopa-Responsive Parkinsonism Are Late Features of Dravet Syndrome (P3.241)

2016· article· en· W4389442806 on OpenAlexaff
Fasano Alfonso, Felippe Borlot, Anthony E. Lang, Danielle M. Andrade

Bibliographic record

VenueNeurology · 2016
Typearticle
Languageen
FieldMedicine
TopicEpilepsy research and treatment
Canadian institutionsHospital for Sick ChildrenToronto Western HospitalUniversity of Toronto
Fundersnot available
KeywordsParkinsonismDravet syndromeLevodopaMedicinePsychiatryEpilepsyInternal medicineParkinson's diseaseDisease

Abstract

fetched live from OpenAlex

Objective: This study described the findings of motor assessments in an adult group of patients with Dravet Syndrome. Background: Dravet syndrome is one of the most severe epileptic encephalopathies. In addition to seizures, cognitive disabilities and gait disturbances are common features. Methods: Adults with clinical and genetic diagnosis of Dravet Syndrome were included. Previous exposure to antipsychotics was an exclusion criterion. A modified Unified Parkinson’s Disease Rating Scale was used. Results: Out of 14 patients, 12 entered the study (mean age: 27.9 ± 7.9). Eight patients (66[percnt]) fulfilled the diagnostic criteria for antecollis, whose severity significantly correlated with patients’ age (ρ = 0.59, p = 0.04). Three patients presented camptocormia, and one of them also had Pisa syndrome. Eleven out of the 12 (91[percnt]) patients presented with parkinsonism, which significantly correlated with patients’ age (ρ = 0.61, p = 0.03). Nine out of 12 (75 [percnt]) cases had gait abnormalities. Postural reflexes were impaired in all the patients with parkinsonism. Levodopa was offered to the four most affected cases; only two have received the medication, and both had significant improvement of slowness and rigidity. Conclusions: We describe for the first time that the majority of adult patients with Dravet Syndrome have features of parkinsonism and antecollis. Meaningful improvement after levodopa trial was seen. These findings expand the phenotype of a disease mainly known by its severe epilepsy and cognitive delay to include drug-responsive movement abnormalities. Knowledge of levodopa responsiveness may greatly diminish the burden of care of such complex patients.

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: Case report · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.001
Threshold uncertainty score0.004

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)0.0010.000

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 head0.014
GPT teacher head0.271
Teacher spread0.257 · 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
Published2016
Admission routes1
Has abstractyes

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