Literature DB >> 21124335

An isoform of the severe encephalopathy-related CDKL5 gene, including a novel exon with extremely high sequence conservation, is specifically expressed in brain.

Yann Fichou1, Juliette Nectoux, Nadia Bahi-Buisson, Jamel Chelly, Thierry Bienvenu.   

Abstract

We report the identification of a novel exon, which is referred to as exon 16b, within the cyclin-dependent kinase (CDK)-like 5 (CDKL5) gene that is implicated in the X-linked infantile spasm syndrome and the early-onset seizure variant of Rett syndrome. Interestingly, it is highly conserved in species through evolution, suggesting a potential functional role, but does not display any homology with other referenced sequences. Most importantly, the transcript including this exon is specifically expressed in brain. We suggest that CDKL5 exon 16b should now be considered in the genetic screening of patients presenting with a CDKL5-related disease profile.

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Year:  2010        PMID: 21124335     DOI: 10.1038/jhg.2010.143

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  14 in total

1.  Identification of a novel CDKL5 exon and pathogenic mutations in patients with severe mental retardation, early-onset seizures and Rett-like features.

Authors:  Nils Rademacher; Melanie Hambrock; Ute Fischer; Bettina Moser; Berten Ceulemans; Wolfgang Lieb; Rainer Boor; Irina Stefanova; Gabriele Gillessen-Kaesbach; Charlotte Runge; Georg Christoph Korenke; Stefanie Spranger; Franco Laccone; Andreas Tzschach; Vera M Kalscheuer
Journal:  Neurogenetics       Date:  2011-02-12       Impact factor: 2.660

2.  A novel transcript of cyclin-dependent kinase-like 5 (CDKL5) has an alternative C-terminus and is the predominant transcript in brain.

Authors:  Sarah L Williamson; Laura Giudici; Charlotte Kilstrup-Nielsen; Wendy Gold; Gregory J Pelka; Patrick P L Tam; Andrew Grimm; Dionigio Prodi; Nicoletta Landsberger; John Christodoulou
Journal:  Hum Genet       Date:  2011-07-12       Impact factor: 4.132

3.  CDKL5-Related Disorders: From Clinical Description to Molecular Genetics.

Authors:  N Bahi-Buisson; T Bienvenu
Journal:  Mol Syndromol       Date:  2011-09-13

4.  Soluble Recombinant Protein Production in Pseudoalteromonas haloplanktis TAC125: The Case Study of the Full-Length Human CDKL5 Protein.

Authors:  Marzia Calvanese; Andrea Colarusso; Concetta Lauro; Ermenegilda Parrilli; Maria Luisa Tutino
Journal:  Methods Mol Biol       Date:  2022

5.  International Consensus Recommendations for the Assessment and Management of Individuals With CDKL5 Deficiency Disorder.

Authors:  Sam Amin; Marie Monaghan; Angel Aledo-Serrano; Nadia Bahi-Buisson; Richard F Chin; Angus J Clarke; J Helen Cross; Scott Demarest; Orrin Devinsky; Jenny Downs; Elia M Pestana Knight; Heather Olson; Carol-Anne Partridge; Graham Stuart; Marina Trivisano; Sameer Zuberi; Tim A Benke
Journal:  Front Neurol       Date:  2022-06-20       Impact factor: 4.086

6.  Mutations in the C-terminus of CDKL5: proceed with caution.

Authors:  Bertrand Diebold; Chloé Delépine; Svetlana Gataullina; Andrée Delahaye; Juliette Nectoux; Thierry Bienvenu
Journal:  Eur J Hum Genet       Date:  2013-06-12       Impact factor: 4.246

Review 7.  What we know and would like to know about CDKL5 and its involvement in epileptic encephalopathy.

Authors:  Charlotte Kilstrup-Nielsen; Laura Rusconi; Paolo La Montanara; Dalila Ciceri; Anna Bergo; Francesco Bedogni; Nicoletta Landsberger
Journal:  Neural Plast       Date:  2012-06-17       Impact factor: 3.599

8.  CDKL5 variants: Improving our understanding of a rare neurologic disorder.

Authors:  Ralph D Hector; Vera M Kalscheuer; Friederike Hennig; Helen Leonard; Jenny Downs; Angus Clarke; Tim A Benke; Judith Armstrong; Mercedes Pineda; Mark E S Bailey; Stuart R Cobb
Journal:  Neurol Genet       Date:  2017-12-15

9.  Mutation in an alternative transcript of CDKL5 in a boy with early-onset seizures.

Authors:  Dale L Bodian; John M Schreiber; Thierry Vilboux; Alina Khromykh; Natalie S Hauser
Journal:  Cold Spring Harb Mol Case Stud       Date:  2018-06-01

10.  Characterisation of CDKL5 Transcript Isoforms in Human and Mouse.

Authors:  Ralph D Hector; Owen Dando; Nicoletta Landsberger; Charlotte Kilstrup-Nielsen; Peter C Kind; Mark E S Bailey; Stuart R Cobb
Journal:  PLoS One       Date:  2016-06-17       Impact factor: 3.240

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