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Record W2345476017 · doi:10.1055/s-0036-1571792

Epilepsy in Neurological Phenotypes of Epidermal Nevus Syndrome

2016· article· en· W2345476017 on OpenAlexaff
Laura Flores‐Sarnat

Bibliographic record

VenueJournal of Pediatric Epilepsy · 2016
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetic and rare skin diseases.
Canadian institutionsAlberta Children's HospitalUniversity of Calgary
Fundersnot available
KeywordsEpilepsyDermatologyNevusMedicinePathologyBiologyNeuroscienceGenetics

Abstract

fetched live from OpenAlex

Epidermal nevus syndrome (ENS) encompasses a group of congenital genetic disorders characterized by the association of an epidermal nevus with systemic anomalies including the brain. The most frequent subtype with neurological features is <i>linear sebaceous nevus syndrome</i> (LSNS), followed by keratinocytic nevus syndrome (ENS). Both are caused by postzygotic <i>RAS</i> mutations. All forms of ENS are mosaic disorders that share a common embryological basis and pathogenesis as <i>neurocristopathies</i>. The lines of Blaschko indicate the cutaneous migratory pathways of neural crest. Neural crest defects explain the development of nevi and associated multisystemic anomalies: brain, ocular, osseous, muscular, orodental, cardiovascular, endocrine, renal/urinary, and adipose tissue. Epilepsy and cognitive and motor deficits are caused mainly by hemimegalencephaly (HME), a closely related mosaic anomaly caused by somatic mutations in PI3K–AKT3–mTOR pathway that determines overgrowth. The usual onset of epilepsy is in the neonatal period or early infancy, often as infantile spasms. The pioneer description of nevus sebaceous in 1895 by Jadassohn is the basis for delineating the LSNS. The eponym “Schimmelpenning syndrome” as a substitute for that long-established term makes no sense in the context of the large number of authors who contributed to defining this syndrome. A review of 111 cases of LSNS and other subtypes enabled a classification of neurological phenotypes; epilepsy occurred in 77% and HME in 38%. Three distinctive additional syndromes also are within the spectrum of ENS: CLOVES, SCALP, and Heide's syndrome. The impact of epilepsy in patients affected by ENS is related to time of onset and extent of brain anomalies. Timing of the mutation determines the phenotype and severity of the systemic lesions.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation 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 score0.014
Threshold uncertainty score0.405

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
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)0.0000.000

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 head0.009
GPT teacher head0.234
Teacher spread0.225 · 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 teacher head, 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

Citations11
Published2016
Admission routes1
Has abstractyes

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