Literature DB >> 23774309

Video/EEG findings in a KCNQ2 epileptic encephalopathy: a case report and revision of literature data.

Domenico Serino1, Nicola Specchio, Giuseppe Pontrelli, Federico Vigevano, Lucia Fusco.   

Abstract

We describe the EEG findings of an infant with early-onset epileptic encephalopathy with mutation of the KCNQ2 gene and a family history of neonatal seizures. The infant presented with multifocal drug-resistant seizures with onset during the third day of life. Family history was positive for early-onset neonatal seizures. Metabolic screening and neuroimaging were negative. Direct sequencing of KCQN2 from both the mother and child revealed a heterozygous cytosine-to-guanine mutation (Dedek et al., 2003). Interictal EEG showed a very discontinuous pattern which evolved towards a defined burst-suppression pattern during sleep and a multifocal, random, attenuation pattern during wakefulness. Focal, tonic seizures with head deviation, sometimes followed by asynchronous and asymmetrical clonic jerks, eyelid myoclonias, and polypnoea, were recorded. Ictal EEG was characterised by focal, low-voltage, fast activity, followed by recruiting theta rhythms and bilateral, focal, spike-wave complexes, alternatively localised to one hemisphere and subsequently diffusing to the other. ACTH therapy was introduced, resulting in a significant improvement in EEG activity and gradual reduction in seizure frequency, with cessation at age 13 weeks. [Published with video sequences].

Entities:  

Keywords:  KCNQ2; burst-suppression; encephalopathy; epilepsy; infant

Mesh:

Substances:

Year:  2013        PMID: 23774309     DOI: 10.1684/epd.2013.0578

Source DB:  PubMed          Journal:  Epileptic Disord        ISSN: 1294-9361            Impact factor:   1.819


  6 in total

1.  Clinical and genetic features of 13 Spanish patients with KCNQ2 mutations.

Authors:  Montesclaros Hortigüela; Ana Fernández-Marmiesse; Verónica Cantarín; Sofía Gouveia; Juan J García-Peñas; Carmen Fons; Judith Armstrong; Desirée Barrios; Felícitas Díaz-Flores; Pilar Tirado; María L Couce; Luis G Gutiérrez-Solana
Journal:  J Hum Genet       Date:  2016-08-18       Impact factor: 3.172

2.  Evolutionary analysis of voltage-gated potassium channels by Bayes method.

Authors:  Qi Huang; Yuan Wu; Xing Wei; Wenwu He; Xixia Liu; Jiemei Ye
Journal:  J Mol Neurosci       Date:  2013-12-07       Impact factor: 3.444

3.  Severe Neonatal Epileptic Encephalopathy and KCNQ2 Mutation: Neuropathological Substrate?

Authors:  Charlotte Dalen Meurs-van der Schoor; Mirjam van Weissenbruch; Marjan van Kempen; Marianna Bugiani; Eleonora Aronica; Hanneke Ronner; R Jeroen Vermeulen
Journal:  Front Pediatr       Date:  2014-12-19       Impact factor: 3.418

4.  Extending the KCNQ2 encephalopathy spectrum: clinical and neuroimaging findings in 17 patients.

Authors:  Sarah Weckhuysen; Vanja Ivanovic; Rik Hendrickx; Rudy Van Coster; Helle Hjalgrim; Rikke S Møller; Sabine Grønborg; An-Sofie Schoonjans; Berten Ceulemans; Sinead B Heavin; Christin Eltze; Rita Horvath; Gianluca Casara; Tiziana Pisano; Lucio Giordano; Kevin Rostasy; Edda Haberlandt; Beate Albrecht; Andrea Bevot; Ira Benkel; Steffan Syrbe; Beth Sheidley; Renzo Guerrini; Annapurna Poduri; Johannes R Lemke; Simone Mandelstam; Ingrid Scheffer; Marco Angriman; Pasquale Striano; Carla Marini; Arvid Suls; Peter De Jonghe
Journal:  Neurology       Date:  2013-10-09       Impact factor: 9.910

Review 5.  Prioritization of Variants Detected by Next Generation Sequencing According to the Mutation Tolerance and Mutational Architecture of the Corresponding Genes.

Authors:  Iria Roca; Ana Fernández-Marmiesse; Sofía Gouveia; Marta Segovia; María L Couce
Journal:  Int J Mol Sci       Date:  2018-05-27       Impact factor: 5.923

Review 6.  Capturing seizures in clinical trials of antiseizure medications for KCNQ2-DEE.

Authors:  John J Millichap; Cynthia L Harden; Dennis J Dlugos; Jacqueline A French; Noam N Butterfield; Celene Grayson; Ernesto Aycardi; Simon N Pimstone
Journal:  Epilepsia Open       Date:  2021-01-29
  6 in total

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