نتایج جستجو برای: scn1a mutations

تعداد نتایج: 173129  

Journal: :The Journal of neuroscience : the official journal of the Society for Neuroscience 2007
Ikuo Ogiwara Hiroyuki Miyamoto Noriyuki Morita Nafiseh Atapour Emi Mazaki Ikuyo Inoue Tamaki Takeuchi Shigeyoshi Itohara Yuchio Yanagawa Kunihiko Obata Teiichi Furuichi Takao K Hensch Kazuhiro Yamakawa

Loss-of-function mutations in human SCN1A gene encoding Nav1.1 are associated with a severe epileptic disorder known as severe myoclonic epilepsy in infancy. Here, we generated and characterized a knock-in mouse line with a loss-of-function nonsense mutation in the Scn1a gene. Both homozygous and heterozygous knock-in mice developed epileptic seizures within the first postnatal month. Immunohis...

Journal: :The Journal of neuroscience : the official journal of the Society for Neuroscience 2012
Lei Sun Jeff Gilligan Cynthia Staber Ryan J Schutte Vivian Nguyen Diane K O'Dowd Robert Reenan

Over 40 missense mutations in the human SCN1A sodium channel gene are linked to an epilepsy syndrome termed genetic epilepsy with febrile seizures plus (GEFS+). Inheritance of GEFS+ is dominant, but the underlying cellular mechanisms remain poorly understood. Here we report that knock-in of a GEFS+ SCN1A mutation (K1270T) into the Drosophila sodium channel gene, para, causes a semidominant temp...

Journal: :Seizure 2011
Marilena Vecchi Matteo Cassina Alberto Casarin Chiara Rigon Paola Drigo Luca De Palma Maurizio Clementi

Epilepsies can be caused by specific genetic anomalies or by non-genetic factors, but in many cases the underlying cause is unknown. Mutations in the SCN1A and SCN2A genes are reported in childhood epilepsies; in particular SCN1A was found mutated in patients with Dravet syndrome and with generalized epilepsy with febrile seizures plus (GEFS+). In this paper we report a patient presenting with ...

2016
Chunxiang Fan Stefan Wolking Frank Lehmann-Horn Ulrike BS Hedrich Tobias Freilinger Holger Lerche Guntram Borck Christian Kubisch Karin Jurkat-Rott

Introduction Familial hemiplegic migraine (FHM) is a rare autosomal dominant subtype of migraine with aura. The FHM3 subtype is caused by mutations in SCN1A, which is also the most frequent epilepsy gene encoding the voltage-gated Na+ channel NaV1.1. The aim of this study was to explore the clinical, genetic and pathogenetic features of a pure FHM3 family. Methods A three-generation family was ...

Journal: :Seizure-european Journal of Epilepsy 2021

PurposeThis study aimed to investigate the genetic aetiology in Chinese children diagnosed with status epilepticus (SE).MethodsNext-generation sequencing, copy number variation (CNV) analysis, and other testing methods were conducted for SE lacking an identifiable non-genetic aetiology. Furthermore, phenotype molecular data of patients retrospectively analysed.ResultsAmong aetiology, 73 out 163...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2012
Christine S Cheah Frank H Yu Ruth E Westenbroek Franck K Kalume John C Oakley Gregory B Potter John L Rubenstein William A Catterall

Heterozygous loss-of-function mutations in the brain sodium channel Na(V)1.1 cause Dravet syndrome (DS), a pharmacoresistant infantile-onset epilepsy syndrome with comorbidities of cognitive impairment and premature death. Previous studies using a mouse model of DS revealed reduced sodium currents and impaired excitability in GABAergic interneurons in the hippocampus, leading to the hypothesis ...

Journal: :Current pain and headache reports 2010
Marino Muxfeldt Bianchin Renata Gomes Londero José Eduardo Lima Marcelo Eduardo Bigal

The association of epilepsy and migraine has been long recognized. Migraine and epilepsy are both chronic disorders with episodic attacks. Furthermore, headache may be a premonitory or postdromic symptom of seizures, and migraine headaches may cause seizures per se (migralepsy). Migraine and epilepsy are comorbid, sharing pathophysiological mechanisms and common clinical features. Several recen...

Journal: :The Journal of neuroscience : the official journal of the Society for Neuroscience 2010
Tomoji Mashimo Iori Ohmori Mamoru Ouchida Yukihiro Ohno Toshiko Tsurumi Takafumi Miki Minoru Wakamori Shizuka Ishihara Takashi Yoshida Akiko Takizawa Megumi Kato Masumi Hirabayashi Masashi Sasa Yasuo Mori Tadao Serikawa

Although febrile seizures (FSs) are the most common convulsive syndrome in infants and childhood, the etiology of FSs has remained unclarified. Several missense mutations of the Na(v)1.1 channel (SCN1A), which alter channel properties, have been reported in a familial syndrome of GEFS+ (generalized epilepsy with febrile seizures plus). Here, we generated Scn1a-targeted rats carrying a missense ...

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