Life-span characterization of epilepsy and comorbidities in Dravet syndrome mice carrying a targeted deletion of exon 1 of the Scn1a gene.


Journal

Experimental neurology
ISSN: 1090-2430
Titre abrégé: Exp Neurol
Pays: United States
ID NLM: 0370712

Informations de publication

Date de publication:
08 2022
Historique:
received: 29 10 2021
revised: 06 04 2022
accepted: 21 04 2022
pubmed: 30 4 2022
medline: 9 6 2022
entrez: 29 4 2022
Statut: ppublish

Résumé

Dravet Syndrome (DS) is a catastrophic form of paediatric epilepsy associated with multiple comorbidities mainly caused by mutations in the SCN1A gene. DS progresses in three different phases termed febrile, worsening and stabilization stage. Mice that are haploinsufficient for Scn1a faithfully model each stage of DS, although various aspects have not been fully described, including the temporal appearance and sex differences of the epilepsy and comorbidities. The aim of the present study was to investigate the epilepsy landscape according to the progression of DS and the long-term co-morbidities in the Scn1a(+/-) Male and female F1.Scn1a(+/+) and F1.Scn1a(+/-) At P18, F1.Scn1a(+/-) These results reveal new features of this model that will optimize use and selection of phenotype assays for future studies on the mechanisms, diagnosis, and treatment of DS.

Identifiants

pubmed: 35487274
pii: S0014-4886(22)00115-7
doi: 10.1016/j.expneurol.2022.114090
pii:
doi:

Substances chimiques

NAV1.1 Voltage-Gated Sodium Channel 0
Scn1a protein, mouse 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

114090

Informations de copyright

Copyright © 2022 The Authors. Published by Elsevier Inc. All rights reserved.

Auteurs

Rogério R Gerbatin (RR)

Department of Physiology and Medical Physics, RCSI University of Medicine and Health Sciences, Dublin, Ireland; FutureNeuro SFI Research Centre, RCSI University of Medicine and Health Sciences, Dublin, Ireland.

Joana Augusto (J)

Department of Physiology and Medical Physics, RCSI University of Medicine and Health Sciences, Dublin, Ireland; FutureNeuro SFI Research Centre, RCSI University of Medicine and Health Sciences, Dublin, Ireland; Department of Physiology, Faculty of Medicine, Trinity College Dublin, Dublin, Ireland.

Halima Boutouil (H)

Department of Physiology and Medical Physics, RCSI University of Medicine and Health Sciences, Dublin, Ireland; FutureNeuro SFI Research Centre, RCSI University of Medicine and Health Sciences, Dublin, Ireland; School of Mechanical and Manufacturing Engineering, Dublin City University, Dublin, Ireland.

Cristina R Reschke (CR)

Department of Physiology and Medical Physics, RCSI University of Medicine and Health Sciences, Dublin, Ireland; FutureNeuro SFI Research Centre, RCSI University of Medicine and Health Sciences, Dublin, Ireland; School of Pharmacy and Biomolecular Sciences, RCSI University of Medicine and Health Sciences, Dublin, Ireland.

David C Henshall (DC)

Department of Physiology and Medical Physics, RCSI University of Medicine and Health Sciences, Dublin, Ireland; FutureNeuro SFI Research Centre, RCSI University of Medicine and Health Sciences, Dublin, Ireland. Electronic address: dhenshall@rcsi.ie.

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Classifications MeSH