Literature DB >> 24352161

De novo SCN8A mutation identified by whole-exome sequencing in a boy with neonatal epileptic encephalopathy, multiple congenital anomalies, and movement disorders.

Ulvi Vaher1, Margit Nõukas2, Tiit Nikopensius2, Mart Kals3, Tarmo Annilo2, Mari Nelis3, Katrin Ounap4, Tiia Reimand5, Inga Talvik6, Pilvi Ilves7, Andres Piirsoo8, Enn Seppet9, Andres Metspalu2, Tiina Talvik6.   

Abstract

Epileptic encephalopathies represent a clinically and genetically heterogeneous group of disorders, majority of which are of unknown etiology. We used whole-exome sequencing of a parent-offspring trio to identify the cause of early infantile epileptic encephalopathy in a boy with neonatal seizures, movement disorders, and multiple congenital anomalies who died at the age of 17 months because of respiratory illness and identified a de novo heterozygous missense mutation (c.3979A>G; p.Ile1327Val) in SCN8A (voltage-gated sodium-channel type VIII alpha subunit) gene. The variant was confirmed in the proband with Sanger sequencing. Because the clinical phenotype associated with SCN8A mutations has previously been identified only in a few patients with or without epileptic seizures, these data together with our results suggest that mutations in SCN8A can lead to early infantile epileptic encephalopathy with a broad phenotypic spectrum. Additional investigations will be worthwhile to determine the prevalence and contribution of SCN8A mutations to epileptic encephalopathies.
© The Author(s) 2013.

Entities:  

Keywords:  SCN8A; epileptic encephalopathy; exome sequencing

Mesh:

Substances:

Year:  2013        PMID: 24352161     DOI: 10.1177/0883073813511300

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  27 in total

1.  SCN8A encephalopathy: Research progress and prospects.

Authors:  Miriam H Meisler; Guy Helman; Michael F Hammer; Brandy E Fureman; William D Gaillard; Alan L Goldin; Shinichi Hirose; Atsushi Ishii; Barbara L Kroner; Christoph Lossin; Heather C Mefford; Jack M Parent; Manoj Patel; John Schreiber; Randall Stewart; Vicky Whittemore; Karen Wilcox; Jacy L Wagnon; Phillip L Pearl; Adeline Vanderver; Ingrid E Scheffer
Journal:  Epilepsia       Date:  2016-06-08       Impact factor: 5.864

Review 2.  Epileptic encephalopathies: new genes and new pathways.

Authors:  Sahar Esmaeeli Nieh; Elliott H Sherr
Journal:  Neurotherapeutics       Date:  2014-10       Impact factor: 7.620

3.  Coffin-Siris Syndrome with obesity, macrocephaly, hepatomegaly and hyperinsulinism caused by a mutation in the ARID1B gene.

Authors:  Mari-Anne Vals; Eve Õiglane-Shlik; Margit Nõukas; Riina Shor; Aleksandr Peet; Mart Kals; Paula Ann Kivistik; Andres Metspalu; Katrin Õunap
Journal:  Eur J Hum Genet       Date:  2014-02-26       Impact factor: 4.246

4.  The phenotypic spectrum of SCN8A encephalopathy.

Authors:  Jan Larsen; Gemma L Carvill; Elena Gardella; Gerhard Kluger; Gudrun Schmiedel; Nina Barisic; Christel Depienne; Eva Brilstra; Yuan Mang; Jens Erik Klint Nielsen; Martin Kirkpatrick; David Goudie; Rebecca Goldman; Johanna A Jähn; Birgit Jepsen; Deepak Gill; Miriam Döcker; Saskia Biskup; Jacinta M McMahon; Bobby Koeleman; Mandy Harris; Kees Braun; Carolien G F de Kovel; Carla Marini; Nicola Specchio; Tania Djémié; Sarah Weckhuysen; Niels Tommerup; Monica Troncoso; Ledia Troncoso; Andrea Bevot; Markus Wolff; Helle Hjalgrim; Renzo Guerrini; Ingrid E Scheffer; Heather C Mefford; Rikke S Møller
Journal:  Neurology       Date:  2015-01-07       Impact factor: 9.910

5.  Toward routine genetics-based diagnoses for the epileptic encephalopathies.

Authors:  Andrew Escayg; Jennifer C Wong
Journal:  Epilepsy Curr       Date:  2014-05       Impact factor: 7.500

6.  Characterization of a de novo SCN8A mutation in a patient with epileptic encephalopathy.

Authors:  Carolien G F de Kovel; Miriam H Meisler; Eva H Brilstra; Frederique M C van Berkestijn; Ruben van 't Slot; Stef van Lieshout; Isaac J Nijman; Janelle E O'Brien; Michael F Hammer; Mark Estacion; Stephen G Waxman; Sulayman D Dib-Hajj; Bobby P C Koeleman
Journal:  Epilepsy Res       Date:  2014-09-04       Impact factor: 3.045

Review 7.  Ion Channels in Genetic Epilepsy: From Genes and Mechanisms to Disease-Targeted Therapies.

Authors:  Julia Oyrer; Snezana Maljevic; Ingrid E Scheffer; Samuel F Berkovic; Steven Petrou; Christopher A Reid
Journal:  Pharmacol Rev       Date:  2018-01       Impact factor: 25.468

8.  Mutations in epilepsy and intellectual disability genes in patients with features of Rett syndrome.

Authors:  Heather E Olson; Dimira Tambunan; Christopher LaCoursiere; Marti Goldenberg; Rebecca Pinsky; Emilie Martin; Eugenia Ho; Omar Khwaja; Walter E Kaufmann; Annapurna Poduri
Journal:  Am J Med Genet A       Date:  2015-04-25       Impact factor: 2.802

9.  A novel de novo mutation of SCN8A (Nav1.6) with enhanced channel activation in a child with epileptic encephalopathy.

Authors:  Mark Estacion; Janelle E O'Brien; Allison Conravey; Michael F Hammer; Stephen G Waxman; Sulayman D Dib-Hajj; Miriam H Meisler
Journal:  Neurobiol Dis       Date:  2014-05-27       Impact factor: 5.996

10.  An Scn1a epilepsy mutation in Scn8a alters seizure susceptibility and behavior.

Authors:  Christopher D Makinson; Karoni Dutt; Frank Lin; Ligia A Papale; Anupama Shankar; Arthur J Barela; Robert Liu; Alan L Goldin; Andrew Escayg
Journal:  Exp Neurol       Date:  2015-09-26       Impact factor: 5.330

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