Literature DB >> 32853054

Neuropsychological profiles of two patients with differing SCN8A-pathogenic variants.

L Collins Medlin1, Luis Bello-Espinosa1,2,3, William S MacAllister1,2,3.   

Abstract

The impact of gene-related early infancy onset epilepsies in cognitive development can be potentially devastating. Here we report two cases of SCN8A-related epilepsy that highlight the neuropsychological heterogeneity seen with differing de-novo pathogenic variants. Case 1 is a 6-year-old right-handed girl who presented with SCN8A-developmental and epileptic encephalopathy (SCN8A-DEE) and a missense pathogenic variant (c.802A > C), not previously documented in the literature. Her history includes speech and motor delay, with focal motor seizures starting at 4-months. Early EEG showed bilateral centroparietal epileptiform discharges. She shows motor and language delays and prominent motor tics. Testing documented Intellectual Disability (ID) (Mild) with widespread neuropsychological deficits (i.e., academics, attention/executive functions, memory, visual-spatial skills, fine motor, language). Case 2 is an 8-year-old right-handed girl who presented with SCN8A-related epilepsy with c.5630A > G pathogenic variant with seizure onset at 5-months. Her initial EEG showed right occipital spikes. She shows low average intellect and average academics, but evaluation documented attention deficits, fine motor delays, and behavioral issues in addition to tics; she was diagnosed with Attention-Deficit/Hyperactivity Disorder, Oppositional Defiant Disorder, Obsessive Compulsive Disorder, and Tourette's. These cases expand limited knowledge regarding neuropsychological functioning of children with SCN8A-related epilepsy with unique de-novo pathogenic variants. While SCN8A-DEE is clearly associated with ID, other pathogenic variants may show better preserved intellect, despite other neuropsychological and behavioral concerns.

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Keywords:  Children; SCN8A; epilepsy; neuropsychology

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Year:  2020        PMID: 32853054     DOI: 10.1080/21622965.2020.1807983

Source DB:  PubMed          Journal:  Appl Neuropsychol Child        ISSN: 2162-2965            Impact factor:   1.493


  1 in total

1.  Effect of fenfluramine on seizures and comorbidities in SCN8A-developmental and epileptic encephalopathy: A case series.

Authors:  Ángel Aledo-Serrano; Borja Cabal-Paz; Elena Gardella; Pablo Gómez-Porro; Otilia Martínez-Múgica; Alvaro Beltrán-Corbellini; Rafael Toledano; Irene García-Morales; Antonio Gil-Nagel
Journal:  Epilepsia Open       Date:  2022-07-20
  1 in total

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