Literature DB >> 23916860

Ring chromosome 20: a pediatric potassium channelopathy responsive to treatment with ezogabine.

Diana J Walleigh1, Agustin Legido, Ignacio Valencia.   

Abstract

BACKGROUND: Ring chromosome 20 is a genetic disorder characterized by intractable epilepsy, behavioral problems, and cognitive deficit. The potassium channel-coding gene KCNQ2 is localized at the locus q13.3 on the chromosome 20, the most common site where the ring occurs. Ezogabine is the first potassium channel opener marketed in the United States. PATIENTS: We describe an 8-year-old girl with mosaic ring chromosome 20 and refractory epilepsy who had a remarkable improvement in seizure control with ezogabine.
CONCLUSIONS: This is the first report using the new antiepileptic drug ezogabine to treat pediatric epilepsy. We hypothesize that ring chromosome 20 patients have epilepsy related to abnormalities in the potassium channels, making it susceptible for treatment with potassium channel openers.
Copyright © 2013 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  epilepsy; ezogabine; potassium channel openers; ring chromosome 20

Mesh:

Substances:

Year:  2013        PMID: 23916860     DOI: 10.1016/j.pediatrneurol.2013.06.005

Source DB:  PubMed          Journal:  Pediatr Neurol        ISSN: 0887-8994            Impact factor:   3.372


  6 in total

Review 1.  The genetics of the epilepsies.

Authors:  Christelle M El Achkar; Heather E Olson; Annapurna Poduri; Phillip L Pearl
Journal:  Curr Neurol Neurosci Rep       Date:  2015-07       Impact factor: 5.081

2.  Autism Spectrum Disorder and a De Novo Kcnq2 Gene Mutation: A Case Report.

Authors:  Martina Siracusano; Claudia Marcovecchio; Assia Riccioni; Caterina Dante; Luigi Mazzone
Journal:  Pediatr Rep       Date:  2022-04-24

Review 3.  Genetic forms of epilepsies and other paroxysmal disorders.

Authors:  Heather E Olson; Annapurna Poduri; Phillip L Pearl
Journal:  Semin Neurol       Date:  2014-09-05       Impact factor: 3.420

Review 4.  Homeostasis or channelopathy? Acquired cell type-specific ion channel changes in temporal lobe epilepsy and their antiepileptic potential.

Authors:  Jakob Wolfart; Debora Laker
Journal:  Front Physiol       Date:  2015-06-15       Impact factor: 4.566

5.  Pharmacokinetics of XEN496, a Novel Pediatric Formulation of Ezogabine, Under Fed and Fasted Conditions: A Phase 1 Trial.

Authors:  Rostam Namdari; Constanza Luzon; Jay A Cadieux; Jennifer Leung; Gregory N Beatch
Journal:  Neurol Ther       Date:  2022-04-05

6.  Lithium improved behavioral and epileptic symptoms in an adolescent with ring chromosome 20 and bipolar disorder not otherwise specified.

Authors:  Adlane Inal; Boris Chaumette; Maryam Soleimani; Anne-Marie Guerrot; Alice Goldenberg; Axel Lebas; Priscille Gerardin; Vladimir Ferrafiat
Journal:  Clin Case Rep       Date:  2018-10-12
  6 in total

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