Literature DB >> 25914188

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

Heather E Olson1,2,3,4, Dimira Tambunan1, Christopher LaCoursiere1, Marti Goldenberg4, Rebecca Pinsky1, Emilie Martin1, Eugenia Ho4,5, Omar Khwaja3,4,5, Walter E Kaufmann2,3,4,5, Annapurna Poduri1,2,3,4,6.   

Abstract

Rett syndrome and neurodevelopmental disorders with features overlapping this syndrome frequently remain unexplained in patients without clinically identified MECP2 mutations. We recruited a cohort of 11 patients with features of Rett syndrome and negative initial clinical testing for mutations in MECP2. We analyzed their phenotypes to determine whether patients met formal criteria for Rett syndrome, reviewed repeat clinical genetic testing, and performed exome sequencing of the probands. Using 2010 diagnostic criteria, three patients had classical Rett syndrome, including two for whom repeat MECP2 gene testing had identified mutations. In a patient with neonatal onset epilepsy with atypical Rett syndrome, we identified a frameshift deletion in STXBP1. Among seven patients with features of Rett syndrome not fulfilling formal diagnostic criteria, four had suspected pathogenic mutations, one each in MECP2, FOXG1, SCN8A, and IQSEC2. MECP2 mutations are highly correlated with classical Rett syndrome. Genes associated with atypical Rett syndrome, epilepsy, or intellectual disability should be considered in patients with features overlapping with Rett syndrome and negative MECP2 testing. While most of the identified mutations were apparently de novo, the SCN8A variant was inherited from an unaffected parent mosaic for the mutation, which is important to note for counseling regarding recurrence risks.
© 2015 Wiley Periodicals, Inc.

Entities:  

Keywords:  CDKL5; FOXG1; MECP2; Rett syndrome; SCN8A; STXBP1; deletions; genetic; mutations; whole exome sequencing

Mesh:

Substances:

Year:  2015        PMID: 25914188      PMCID: PMC5722031          DOI: 10.1002/ajmg.a.37132

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  36 in total

1.  Mutations in MEF2C from the 5q14.3q15 microdeletion syndrome region are a frequent cause of severe mental retardation and diminish MECP2 and CDKL5 expression.

Authors:  Markus Zweier; Anne Gregor; Christiane Zweier; Hartmut Engels; Heinrich Sticht; Eva Wohlleber; Emilia K Bijlsma; Susan E Holder; Martin Zenker; Eva Rossier; Ute Grasshoff; Diana S Johnson; Lisa Robertson; Helen V Firth; Arif B Ekici; André Reis; Anita Rauch
Journal:  Hum Mutat       Date:  2010-06       Impact factor: 4.878

2.  Congenital variant of Rett syndrome due to an intragenic large deletion in MECP2.

Authors:  Yu Kobayashi; Tsukasa Ohashi; Noriyuki Akasaka; Jun Tohyama
Journal:  Brain Dev       Date:  2011-10-15       Impact factor: 1.961

3.  Next-generation sequencing in X-linked intellectual disability.

Authors:  Andreas Tzschach; Ute Grasshoff; Stefanie Beck-Woedl; Claudia Dufke; Claudia Bauer; Martin Kehrer; Christina Evers; Ute Moog; Barbara Oehl-Jaschkowitz; Nataliya Di Donato; Robert Maiwald; Christine Jung; Alma Kuechler; Solveig Schulz; Peter Meinecke; Stephanie Spranger; Jürgen Kohlhase; Jörg Seidel; Silke Reif; Manuela Rieger; Angelika Riess; Marc Sturm; Julia Bickmann; Christopher Schroeder; Andreas Dufke; Olaf Riess; Peter Bauer
Journal:  Eur J Hum Genet       Date:  2015-02-04       Impact factor: 4.246

4.  The CDKL5 disorder is an independent clinical entity associated with early-onset encephalopathy.

Authors:  Stephanie Fehr; Meredith Wilson; Jenny Downs; Simon Williams; Alessandra Murgia; Stefano Sartori; Marilena Vecchi; Gladys Ho; Roberta Polli; Stavroula Psoni; Xinhua Bao; Nick de Klerk; Helen Leonard; John Christodoulou
Journal:  Eur J Hum Genet       Date:  2012-08-08       Impact factor: 4.246

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

Authors:  Ulvi Vaher; Margit Nõukas; Tiit Nikopensius; Mart Kals; Tarmo Annilo; Mari Nelis; Katrin Ounap; Tiia Reimand; Inga Talvik; Pilvi Ilves; Andres Piirsoo; Enn Seppet; Andres Metspalu; Tiina Talvik
Journal:  J Child Neurol       Date:  2013-12-18       Impact factor: 1.987

6.  Netrin G1 mutations are an uncommon cause of atypical Rett syndrome with or without epilepsy.

Authors:  Juliette Nectoux; Benoit Girard; Nadia Bahi-Buisson; Fabienne Prieur; Alexandra Afenjar; Haydee Rosas-Vargas; Jamel Chelly; Thierry Bienvenu
Journal:  Pediatr Neurol       Date:  2007-10       Impact factor: 3.372

Review 7.  Genetic testing in the epilepsies-developments and dilemmas.

Authors:  Annapurna Poduri; Beth Rosen Sheidley; Sara Shostak; Ruth Ottman
Journal:  Nat Rev Neurol       Date:  2014-04-15       Impact factor: 42.937

8.  Diagnostic exome sequencing identifies two novel IQSEC2 mutations associated with X-linked intellectual disability with seizures: implications for genetic counseling and clinical diagnosis.

Authors:  Stephanie K Gandomi; K D Farwell Gonzalez; M Parra; L Shahmirzadi; J Mancuso; P Pichurin; R Temme; S Dugan; W Zeng; Sha Tang
Journal:  J Genet Couns       Date:  2013-12-04       Impact factor: 2.537

9.  Parental somatic mosaicism is underrecognized and influences recurrence risk of genomic disorders.

Authors:  Ian M Campbell; Bo Yuan; Caroline Robberecht; Rolph Pfundt; Przemyslaw Szafranski; Meriel E McEntagart; Sandesh C S Nagamani; Ayelet Erez; Magdalena Bartnik; Barbara Wiśniowiecka-Kowalnik; Katie S Plunkett; Amber N Pursley; Sung-Hae L Kang; Weimin Bi; Seema R Lalani; Carlos A Bacino; Mala Vast; Karen Marks; Michael Patton; Peter Olofsson; Ankita Patel; Joris A Veltman; Sau Wai Cheung; Chad A Shaw; Lisenka E L M Vissers; Joris R Vermeesch; James R Lupski; Paweł Stankiewicz
Journal:  Am J Hum Genet       Date:  2014-07-31       Impact factor: 11.025

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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  33 in total

1.  A novel splicing mutation in the IQSEC2 gene that modulates the phenotype severity in a family with intellectual disability.

Authors:  Irene Madrigal; Maria Isabel Alvarez-Mora; Jordi Rosell; Laia Rodríguez-Revenga; Olof Karlberg; Sascha Sauer; Ann-Christine Syvänen; Montserrat Mila
Journal:  Eur J Hum Genet       Date:  2016-01-06       Impact factor: 4.246

2.  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

3.  Regulation of dendrite growth and maintenance by exocytosis.

Authors:  Yun Peng; Jiae Lee; Kimberly Rowland; Yuhui Wen; Hope Hua; Nicole Carlson; Shweta Lavania; Jay Z Parrish; Michael D Kim
Journal:  J Cell Sci       Date:  2015-10-19       Impact factor: 5.285

4.  STXBP1 encephalopathies: Clinical spectrum, disease mechanisms, and therapeutic strategies.

Authors:  Debra Abramov; Noah Guy Lewis Guiberson; Jacqueline Burré
Journal:  J Neurochem       Date:  2020-08-04       Impact factor: 5.372

5.  Monogenic disorders that mimic the phenotype of Rett syndrome.

Authors:  Siddharth Srivastava; Sonal Desai; Julie Cohen; Constance Smith-Hicks; Kristin Barañano; Ali Fatemi; SakkuBai Naidu
Journal:  Neurogenetics       Date:  2018-01-10       Impact factor: 2.660

6.  Effects of Deltamethrin Acute Exposure on Nav1.6 Channels and Medium Spiny Neurons of the Nucleus Accumbens.

Authors:  Cynthia M Tapia; Oluwarotimi Folorunso; Aditya K Singh; Kathleen McDonough; Fernanda Laezza
Journal:  Toxicology       Date:  2020-05-06       Impact factor: 4.221

7.  MECP2 mutations in Czech patients with Rett syndrome and Rett-like phenotypes: novel mutations, genotype-phenotype correlations and validation of high-resolution melting analysis for mutation scanning.

Authors:  Daniela Zahorakova; Petra Lelkova; Vladimir Gregor; Martin Magner; Jiri Zeman; Pavel Martasek
Journal:  J Hum Genet       Date:  2016-03-17       Impact factor: 3.172

8.  Gonadal mosaicism of a novel IQSEC2 variant causing female limited intellectual disability and epilepsy.

Authors:  Lisa J Ewans; Michael Field; Ying Zhu; Gillian Turner; Melanie Leffler; Marcel E Dinger; Mark J Cowley; Michael F Buckley; Ingrid E Scheffer; Matilda R Jackson; Tony Roscioli; Cheryl Shoubridge
Journal:  Eur J Hum Genet       Date:  2017-03-15       Impact factor: 4.246

9.  A New Approach to Rare Diseases of Children: The Undiagnosed Diseases Network.

Authors:  Chloe M Reuter; Elise Brimble; Colette DeFilippo; Annika M Dries; Gregory M Enns; Euan A Ashley; Jonathan A Bernstein; Paul Graham Fisher; Matthew T Wheeler
Journal:  J Pediatr       Date:  2018-01-11       Impact factor: 4.406

10.  Altered excitatory transmission onto hippocampal interneurons in the IQSEC2 mouse model of X-linked neurodevelopmental disease.

Authors:  Megha Sah; Amy N Shore; Sabrina Petri; Ayla Kanber; Mu Yang; Matthew C Weston; Wayne N Frankel
Journal:  Neurobiol Dis       Date:  2020-01-21       Impact factor: 5.996

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