Temporal lobe epilepsy and GEFS+ phenotypes associated with SCN1B mutations.

نویسندگان

  • Ingrid E Scheffer
  • Louise A Harkin
  • Bronwyn E Grinton
  • Leanne M Dibbens
  • Samantha J Turner
  • Marta A Zielinski
  • Ruwei Xu
  • Graeme Jackson
  • Judith Adams
  • Mary Connellan
  • Steven Petrou
  • R Mark Wellard
  • Regula S Briellmann
  • Robyn H Wallace
  • John C Mulley
  • Samuel F Berkovic
چکیده

SCN1B, the gene encoding the sodium channel beta 1 subunit, was the first gene identified for generalized epilepsy with febrile seizures plus (GEFS+). Only three families have been published with SCN1B mutations. Here, we present four new families with SCN1B mutations and characterize the associated phenotypes. Analysis of SCN1B was performed on 402 individuals with various epilepsy syndromes. Four probands with missense mutations were identified. Detailed electroclinical phenotyping was performed on all available affected family members including quantitative MR imaging in those with temporal lobe epilepsy (TLE). Two new families with the original C121W SCN1B mutation were identified; novel mutations R85C and R85H were each found in one family. The following phenotypes occurred in the six families with SCN1B missense mutations: 22 febrile seizures, 20 febrile seizures plus, five TLE, three other GEFS+ phenotypes, two unclassified and ten unaffected individuals. All individuals with confirmed TLE had the C121W mutation; two underwent temporal lobectomy (one with hippocampal sclerosis and one without) and both are seizure free. We confirm the role of SCN1B in GEFS+ and show that the GEFS+ spectrum may include TLE alone. TLE with an SCN1B mutation is not a contraindication to epilepsy surgery.

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SHORT COMMUNICATION Enhanced inactivation and acceleration of activation of the sodium channel associated with epilepsy in man

Generalized epilepsy with febrile seizures-plus (GEFS) is a benign Mendelian syndrome characterized by childhood-onset febrile and afebrile seizures. Three point mutations within two voltage-gated sodium channel genes have been identi®ed so far: in GEFS type 1 a mutation in the b1-subunit gene SCN1B, and in GEFS type 2 two mutations within the neuronal a-subunit gene SCN1A. Functional expressio...

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Enhanced inactivation and acceleration of activation of the sodium channel associated with epilepsy in man.

Generalized epilepsy with febrile seizures-plus (GEFS+) is a benign Mendelian syndrome characterized by childhood-onset febrile and afebrile seizures. Three point mutations within two voltage-gated sodium channel genes have been identified so far: in GEFS+ type 1 a mutation in the beta1-subunit gene SCN1B, and in GEFS+ type 2 two mutations within the neuronal alpha-subunit gene SCN1A. Functiona...

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عنوان ژورنال:
  • Brain : a journal of neurology

دوره 130 Pt 1  شماره 

صفحات  -

تاریخ انتشار 2007