Literature DB >> 26821542

Epileptic Encephalopathy in Childhood: A Stepwise Approach for Identification of Underlying Genetic Causes.

Jaina Patel1, Saadet Mercimek-Mahmutoglu2,3.   

Abstract

Epilepsy is one of the most common neurological disorders in childhood. Epilepsy associated with global developmental delay and cognitive dysfunction is defined as epileptic encephalopathy. Certain inherited metabolic disorders presenting with epileptic encephalopathy can be treated with disease specific diet, vitamin, amino acid or cofactor supplementations. In those disorders, disease specific therapy is successful to achieve good seizure control and improve long-term neurodevelopmental outcome. For this reason, intractable epilepsy with global developmental delay or history of developmental regression warrants detailed metabolic investigations for the possibility of an underlying treatable inherited metabolic disorder, which should be undertaken as first line investigations. An underlying genetic etiology in epileptic encephalopathy has been supported by recent studies such as array comparative genomic hybridization, targeted next generation sequencing panels, whole exome and whole genome sequencing. These studies report a diagnostic yield up to 70%, depending on the applied genetic testing as well as number of patients enrolled. In patients with epileptic encephalopathy, a stepwise approach for diagnostic work-up will help to diagnose treatable inherited metabolic disorders quickly. Application of detailed genetic investigations such as targeted next generation sequencing as second line and whole exome sequencing as third line testing will diagnose underlying genetic disease which will help for genetic counseling as well as guide for prenatal diagnosis. Knowledge of underlying genetic cause will provide novel insights into the pathogenesis of epileptic encephalopathy and pave the ground towards the development of targeted neuroprotective treatment strategies to improve the health outcome of children with epileptic encephalopathy.

Entities:  

Keywords:  Copy number variants; Epilepsy; Genetics; Inherited metabolic disorders; Molecular genetics

Mesh:

Year:  2016        PMID: 26821542     DOI: 10.1007/s12098-015-1979-9

Source DB:  PubMed          Journal:  Indian J Pediatr        ISSN: 0019-5456            Impact factor:   1.967


  89 in total

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Authors:  Gemma L Carvill; Sarah Weckhuysen; Jacinta M McMahon; Corinna Hartmann; Rikke S Møller; Helle Hjalgrim; Joseph Cook; Eileen Geraghty; Brian J O'Roak; Steve Petrou; Alison Clarke; Deepak Gill; Lynette G Sadleir; Hiltrud Muhle; Sarah von Spiczak; Marina Nikanorova; Bree L Hodgson; Elena V Gazina; Arvid Suls; Jay Shendure; Leanne M Dibbens; Peter De Jonghe; Ingo Helbig; Samuel F Berkovic; Ingrid E Scheffer; Heather C Mefford
Journal:  Neurology       Date:  2014-03-12       Impact factor: 9.910

2.  Lysine restricted diet for pyridoxine-dependent epilepsy: first evidence and future trials.

Authors:  Clara D M van Karnebeek; Hans Hartmann; Sravan Jaggumantri; Levinus A Bok; Barb Cheng; Mary Connolly; Curtis R Coughlin; Anibh M Das; Sidney M Gospe; Cornelis Jakobs; Johanna H van der Lee; Saadet Mercimek-Mahmutoglu; Uta Meyer; Eduard Struys; Graham Sinclair; Johan Van Hove; Jean-Paul Collet; Barbara R Plecko; Sylvia Stockler
Journal:  Mol Genet Metab       Date:  2012-09-10       Impact factor: 4.797

3.  Chromosomal microarray in unexplained severe early onset epilepsy - A single centre cohort.

Authors:  Nicholas M Allen; Judith Conroy; Amre Shahwan; Sean Ennis; Bryan Lynch; Sally A Lynch; Mary D King
Journal:  Eur J Paediatr Neurol       Date:  2015-04-10       Impact factor: 3.140

4.  STXBP1-related encephalopathy presenting as infantile spasms and generalized tremor in three patients.

Authors:  Cyril Mignot; Marie-Laure Moutard; Oriane Trouillard; Isabelle Gourfinkel-An; Aurélia Jacquette; Benoit Arveiler; Fanny Morice-Picard; Didier Lacombe; Catherine Chiron; Dorothée Ville; Perrine Charles; Eric LeGuern; Christel Depienne; Delphine Héron
Journal:  Epilepsia       Date:  2011-07-18       Impact factor: 5.864

5.  Targeted next generation sequencing as a diagnostic tool in epileptic disorders.

Authors:  Johannes R Lemke; Erik Riesch; Tim Scheurenbrand; Max Schubach; Christian Wilhelm; Isabelle Steiner; Jörg Hansen; Carolina Courage; Sabina Gallati; Sarah Bürki; Susi Strozzi; Barbara Goeggel Simonetti; Sebastian Grunt; Maja Steinlin; Michael Alber; Markus Wolff; Thomas Klopstock; Eva C Prott; Rüdiger Lorenz; Christiane Spaich; Sabine Rona; Maya Lakshminarasimhan; Judith Kröll; Thomas Dorn; Günter Krämer; Matthis Synofzik; Felicitas Becker; Yvonne G Weber; Holger Lerche; Detlef Böhm; Saskia Biskup
Journal:  Epilepsia       Date:  2012-05-21       Impact factor: 5.864

6.  Evidence-based treatment of guanidinoacetate methyltransferase (GAMT) deficiency.

Authors:  Krista S Viau; Sharon L Ernst; Marzia Pasquali; Lorenzo D Botto; Gary Hedlund; Nicola Longo
Journal:  Mol Genet Metab       Date:  2013-09-08       Impact factor: 4.797

7.  Epileptic and nonepileptic features in patients with early onset epileptic encephalopathy and STXBP1 mutations.

Authors:  Mathieu Milh; Nathalie Villeneuve; Mondher Chouchane; Anna Kaminska; Cécile Laroche; Marie Anne Barthez; Cyril Gitiaux; Céline Bartoli; Ana Borges-Correia; Pierre Cacciagli; Cécile Mignon-Ravix; Hélène Cuberos; Brigitte Chabrol; Laurent Villard
Journal:  Epilepsia       Date:  2011-07-19       Impact factor: 5.864

8.  Favorable outcome in a newborn with molybdenum cofactor type A deficiency treated with cPMP.

Authors:  Marrit M Hitzert; Arend F Bos; Klasien A Bergman; Alex Veldman; Guenter Schwarz; José Angel Santamaria-Araujo; Rebecca Heiner-Fokkema; Deborah A Sival; Roelineke J Lunsing; Sita Arjune; Jos G W Kosterink; Francjan J van Spronsen
Journal:  Pediatrics       Date:  2012-09-17       Impact factor: 7.124

9.  An update on serine deficiency disorders.

Authors:  S N van der Crabben; N M Verhoeven-Duif; E H Brilstra; L Van Maldergem; T Coskun; E Rubio-Gozalbo; R Berger; T J de Koning
Journal:  J Inherit Metab Dis       Date:  2013-03-06       Impact factor: 4.982

10.  De novo mutations in epileptic encephalopathies.

Authors:  Andrew S Allen; Samuel F Berkovic; Patrick Cossette; Norman Delanty; Dennis Dlugos; Evan E Eichler; Michael P Epstein; Tracy Glauser; David B Goldstein; Yujun Han; Erin L Heinzen; Yuki Hitomi; Katherine B Howell; Michael R Johnson; Ruben Kuzniecky; Daniel H Lowenstein; Yi-Fan Lu; Maura R Z Madou; Anthony G Marson; Heather C Mefford; Sahar Esmaeeli Nieh; Terence J O'Brien; Ruth Ottman; Slavé Petrovski; Annapurna Poduri; Elizabeth K Ruzzo; Ingrid E Scheffer; Elliott H Sherr; Christopher J Yuskaitis; Bassel Abou-Khalil; Brian K Alldredge; Jocelyn F Bautista; Samuel F Berkovic; Alex Boro; Gregory D Cascino; Damian Consalvo; Patricia Crumrine; Orrin Devinsky; Dennis Dlugos; Michael P Epstein; Miguel Fiol; Nathan B Fountain; Jacqueline French; Daniel Friedman; Eric B Geller; Tracy Glauser; Simon Glynn; Sheryl R Haut; Jean Hayward; Sandra L Helmers; Sucheta Joshi; Andres Kanner; Heidi E Kirsch; Robert C Knowlton; Eric H Kossoff; Rachel Kuperman; Ruben Kuzniecky; Daniel H Lowenstein; Shannon M McGuire; Paul V Motika; Edward J Novotny; Ruth Ottman; Juliann M Paolicchi; Jack M Parent; Kristen Park; Annapurna Poduri; Ingrid E Scheffer; Renée A Shellhaas; Elliott H Sherr; Jerry J Shih; Rani Singh; Joseph Sirven; Michael C Smith; Joseph Sullivan; Liu Lin Thio; Anu Venkat; Eileen P G Vining; Gretchen K Von Allmen; Judith L Weisenberg; Peter Widdess-Walsh; Melodie R Winawer
Journal:  Nature       Date:  2013-08-11       Impact factor: 49.962

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  4 in total

1.  Editorial: New Horizons in Genetic Diagnosis in Pediatric Practice: The Excitement and Challenges!

Authors:  Ratna D Puri; Madhulika Kabra
Journal:  Indian J Pediatr       Date:  2016-08-11       Impact factor: 1.967

2.  Novel in-frame deletion in MFSD8 gene revealed by trio whole exome sequencing in an Iranian affected with neuronal ceroid lipofuscinosis type 7: a case report.

Authors:  Ali Hosseini Bereshneh; Masoud Garshasbi
Journal:  J Med Case Rep       Date:  2018-09-25

3.  Case report: Clinical and magnetic resonance spectroscopy presentation of a female severely affected with X-linked creatine transporter deficiency.

Authors:  Katherine Morey; Barbara Hallinan; Kim M Cecil
Journal:  Radiol Case Rep       Date:  2022-02-03

Review 4.  Inborn Errors of Metabolism and Epilepsy: Current Understanding, Diagnosis, and Treatment Approaches.

Authors:  Suvasini Sharma; Asuri N Prasad
Journal:  Int J Mol Sci       Date:  2017-07-02       Impact factor: 5.923

  4 in total

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