Literature DB >> 30094525

WWOX-associated encephalopathies: identification of the phenotypic spectrum and the resulting genotype-phenotype correlation.

Hepsen Mine Serin1, Erdem Simsek2, Esra Isik3, Sarenur Gokben2.   

Abstract

Epileptic encephalopathies are a group of disorders in which epileptiform abnormalities cause progressive deterioration in cerebral function. Genetic causes have been described in several of the epileptic encephalopathies, and many previously unknown genes have been identified. WW domain-containing oxidoreductase (WWOX) has recently been implicated in autosomal recessive spinocerebellar ataxia type 12 (SCAR12) and severe early-onset epileptic encephalopathy. With whole-exome sequencing, we identified a homozygous WWOX missense mutation, p.Leu239Arg, in a girl from a consanguineous family with psychomotor developmental delay, acquired microcephaly, and epileptic seizures. WWOX-related epileptic encephalopathy is a rare condition but it should be considered in cases having early epileptic spasms and parental consanguinity.

Entities:  

Keywords:  Epileptic encephalopathy; Genetic; Microcephaly; WWOX

Mesh:

Substances:

Year:  2018        PMID: 30094525     DOI: 10.1007/s10072-018-3528-6

Source DB:  PubMed          Journal:  Neurol Sci        ISSN: 1590-1874            Impact factor:   3.307


  10 in total

1.  A novel whole exon deletion in WWOX gene causes early epilepsy, intellectual disability and optic atrophy.

Authors:  Salma Ben-Salem; Aisha M Al-Shamsi; Anne John; Bassam R Ali; Lihadh Al-Gazali
Journal:  J Mol Neurosci       Date:  2014-11-18       Impact factor: 3.444

2.  A novel missense variant in the SDR domain of the WWOX gene leads to complete loss of WWOX protein with early-onset epileptic encephalopathy and severe developmental delay.

Authors:  Jessika Johannsen; Fanny Kortüm; Georg Rosenberger; Kristin Bokelmann; Markus A Schirmer; Jonas Denecke; René Santer
Journal:  Neurogenetics       Date:  2018-05-28       Impact factor: 2.660

Review 3.  Understanding Genotypes and Phenotypes in Epileptic Encephalopathies.

Authors:  Ingo Helbig; Abou Ahmad N Tayoun
Journal:  Mol Syndromol       Date:  2016-08-20

4.  WWOX-related encephalopathies: delineation of the phenotypical spectrum and emerging genotype-phenotype correlation.

Authors:  Cyril Mignot; Laetitia Lambert; Laurent Pasquier; Thierry Bienvenu; Andrée Delahaye-Duriez; Boris Keren; Jérémie Lefranc; Aline Saunier; Lila Allou; Virginie Roth; Mylène Valduga; Aissa Moustaïne; Stéphane Auvin; Catherine Barrey; Sandra Chantot-Bastaraud; Nicolas Lebrun; Marie-Laure Moutard; Marie-Christine Nougues; Anne-Isabelle Vermersch; Bénédicte Héron; Eva Pipiras; Delphine Héron; Laurence Olivier-Faivre; Jean-Louis Guéant; Philippe Jonveaux; Christophe Philippe
Journal:  J Med Genet       Date:  2014-11-19       Impact factor: 6.318

5.  WWOX and severe autosomal recessive epileptic encephalopathy: first case in the prenatal period.

Authors:  Mylène Valduga; Christophe Philippe; Laetitia Lambert; Pascale Bach-Segura; Emmanuelle Schmitt; Jean Pierre Masutti; Bénédicte François; Patrick Pinaud; Mireille Vibert; Philippe Jonveaux
Journal:  J Hum Genet       Date:  2015-02-26       Impact factor: 3.172

6.  A new form of childhood onset, autosomal recessive spinocerebellar ataxia and epilepsy is localized at 16q21-q23.

Authors:  M Gribaa; M Salih; M Anheim; C Lagier-Tourenne; D H'mida; N Drouot; A Mohamed; S Elmalik; M Kabiraj; M Al-Rayess; M Almubarak; C Bétard; H Goebel; M Koenig
Journal:  Brain       Date:  2007-04-30       Impact factor: 13.501

7.  The tumour suppressor gene WWOX is mutated in autosomal recessive cerebellar ataxia with epilepsy and mental retardation.

Authors:  Martial Mallaret; Matthis Synofzik; Jaeho Lee; Cari A Sagum; Muhammad Mahajnah; Rajech Sharkia; Nathalie Drouot; Mathilde Renaud; Fabrice A C Klein; Mathieu Anheim; Christine Tranchant; Cyril Mignot; Jean-Louis Mandel; Mark Bedford; Peter Bauer; Mustafa A Salih; Rebecca Schüle; Ludger Schöls; C Marcelo Aldaz; Michel Koenig
Journal:  Brain       Date:  2013-12-24       Impact factor: 13.501

8.  A spontaneous mutation of the Wwox gene and audiogenic seizures in rats with lethal dwarfism and epilepsy.

Authors:  H Suzuki; K Katayama; M Takenaka; K Amakasu; K Saito; K Suzuki
Journal:  Genes Brain Behav       Date:  2009-05-20       Impact factor: 3.449

9.  The supposed tumor suppressor gene WWOX is mutated in an early lethal microcephaly syndrome with epilepsy, growth retardation and retinal degeneration.

Authors:  Ghada Abdel-Salam; Michaela Thoenes; Hanan H Afifi; Friederike Körber; Daniel Swan; Hanno Jörn Bolz
Journal:  Orphanet J Rare Dis       Date:  2014-01-23       Impact factor: 4.123

Review 10.  The genetic landscape of the epileptic encephalopathies of infancy and childhood.

Authors:  Amy McTague; Katherine B Howell; J Helen Cross; Manju A Kurian; Ingrid E Scheffer
Journal:  Lancet Neurol       Date:  2015-11-17       Impact factor: 44.182

  10 in total
  2 in total

Review 1.  The WWOX gene in brain development and pathology.

Authors:  K Kośla; Ż Kałuzińska; A K Bednarek
Journal:  Exp Biol Med (Maywood)       Date:  2020-05-09

Review 2.  WWOX Loss of Function in Neurodevelopmental and Neurodegenerative Disorders.

Authors:  C Marcelo Aldaz; Tabish Hussain
Journal:  Int J Mol Sci       Date:  2020-11-24       Impact factor: 5.923

  2 in total

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