Literature DB >> 24615633

Rett syndrome and MeCP2.

Vichithra R B Liyanage1, Mojgan Rastegar.   

Abstract

Rett syndrome (RTT) is a severe and progressive neurological disorder, which mainly affects young females. Mutations of the methyl-CpG binding protein 2 (MECP2) gene are the most prevalent cause of classical RTT cases. MECP2 mutations or altered expression are also associated with a spectrum of neurodevelopmental disorders such as autism spectrum disorders with recent links to fetal alcohol spectrum disorders. Collectively, MeCP2 relation to these neurodevelopmental disorders highlights the importance of understanding the molecular mechanisms by which MeCP2 impacts brain development, mental conditions, and compromised brain function. Since MECP2 mutations were discovered to be the primary cause of RTT, a significant progress has been made in the MeCP2 research, with respect to the expression, function and regulation of MeCP2 in the brain and its contribution in RTT pathogenesis. To date, there have been intensive efforts in designing effective therapeutic strategies for RTT benefiting from mouse models and cells collected from RTT patients. Despite significant progress in MeCP2 research over the last few decades, there is still a knowledge gap between the in vitro and in vivo research findings and translating these findings into effective therapeutic interventions in human RTT patients. In this review, we will provide a synopsis of Rett syndrome as a severe neurological disorder and will discuss the role of MeCP2 in RTT pathophysiology.

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Year:  2014        PMID: 24615633      PMCID: PMC5798978          DOI: 10.1007/s12017-014-8295-9

Source DB:  PubMed          Journal:  Neuromolecular Med        ISSN: 1535-1084            Impact factor:   3.843


  299 in total

1.  Clinical profiles of four patients with Rett syndrome carrying a novel exon 1 mutation or genomic rearrangement in the MECP2 gene.

Authors:  D Bartholdi; A Klein; M Weissert; N Koenig; A Baumer; E Boltshauser; A Schinzel; W Berger; G Mátyás
Journal:  Clin Genet       Date:  2006-04       Impact factor: 4.438

Review 2.  Cardiac disease and Rett syndrome.

Authors:  M Acampa; F Guideri
Journal:  Arch Dis Child       Date:  2006-05       Impact factor: 3.791

3.  Dendritic spine pathologies in hippocampal pyramidal neurons from Rett syndrome brain and after expression of Rett-associated MECP2 mutations.

Authors:  Christopher A Chapleau; Gaston D Calfa; Meredith C Lane; Asher J Albertson; Jennifer L Larimore; Shinichi Kudo; Dawna L Armstrong; Alan K Percy; Lucas Pozzo-Miller
Journal:  Neurobiol Dis       Date:  2009-05-12       Impact factor: 5.996

4.  The role of different X-inactivation pattern on the variable clinical phenotype with Rett syndrome.

Authors:  T Ishii; Y Makita; A Ogawa; S Amamiya; M Yamamoto; A Miyamoto; J Oki
Journal:  Brain Dev       Date:  2001-12       Impact factor: 1.961

5.  Downregulation of CNPase in a MeCP2 deficient mouse model of Rett syndrome.

Authors:  Wei Wu; Weizhong Gu; Xuefeng Xu; Shiqiang Shang; Zhengyan Zhao
Journal:  Neurol Res       Date:  2012-01-13       Impact factor: 2.448

6.  The transcriptional repressor Mecp2 regulates terminal neuronal differentiation.

Authors:  Valéry Matarazzo; Deborah Cohen; Amy M Palmer; P Jeanette Simpson; Babar Khokhar; Shih-Jung Pan; Gabriele V Ronnett
Journal:  Mol Cell Neurosci       Date:  2004-09       Impact factor: 4.314

7.  Fetal alcohol syndrome in association with Rett syndrome.

Authors:  B Zoll; P Huppke; A Wessel; I Bartels; F Laccone
Journal:  Genet Couns       Date:  2004

8.  Mecp2 deficiency leads to delayed maturation and altered gene expression in hippocampal neurons.

Authors:  Richard D Smrt; Julialea Eaves-Egenes; Basam Z Barkho; Nicholas J Santistevan; Chunmei Zhao; James B Aimone; Fred H Gage; Xinyu Zhao
Journal:  Neurobiol Dis       Date:  2007-04-27       Impact factor: 5.996

Review 9.  The role of MeCP2 in the brain.

Authors:  Jacky Guy; Hélène Cheval; Jim Selfridge; Adrian Bird
Journal:  Annu Rev Cell Dev Biol       Date:  2011-06-29       Impact factor: 13.827

10.  Adult neural function requires MeCP2.

Authors:  Christopher M McGraw; Rodney C Samaco; Huda Y Zoghbi
Journal:  Science       Date:  2011-06-02       Impact factor: 47.728

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  39 in total

1.  Genetic Determinants of Epigenetic Patterns: Providing Insight into Disease.

Authors:  Emma Cazaly; Jac Charlesworth; Joanne L Dickinson; Adele F Holloway
Journal:  Mol Med       Date:  2015-03-26       Impact factor: 6.354

Review 2.  Diverse and dynamic DNA modifications in brain and diseases.

Authors:  Matthew J Armstrong; Yulin Jin; Emily G Allen; Peng Jin
Journal:  Hum Mol Genet       Date:  2019-11-21       Impact factor: 6.150

Review 3.  DNA methylation regulated gene expression in organ fibrosis.

Authors:  Xiangyu Zhang; Min Hu; Xing Lyu; Chun Li; Victor J Thannickal; Yan Y Sanders
Journal:  Biochim Biophys Acta Mol Basis Dis       Date:  2017-05-10       Impact factor: 5.187

4.  Altered gut microbiota in Rett syndrome.

Authors:  Francesco Strati; Duccio Cavalieri; Davide Albanese; Claudio De Felice; Claudio Donati; Joussef Hayek; Olivier Jousson; Silvia Leoncini; Massimo Pindo; Daniela Renzi; Lisa Rizzetto; Irene Stefanini; Antonio Calabrò; Carlotta De Filippo
Journal:  Microbiome       Date:  2016-07-30       Impact factor: 14.650

5.  Ethanol deregulates Mecp2/MeCP2 in differentiating neural stem cells via interplay between 5-methylcytosine and 5-hydroxymethylcytosine at the Mecp2 regulatory elements.

Authors:  Vichithra Rasangi Batuwita Liyanage; Robby Mathew Zachariah; James Ronald Davie; Mojgan Rastegar
Journal:  Exp Neurol       Date:  2015-01-22       Impact factor: 5.330

6.  Bioinformatic Analysis of DNA Methylation in Neural Progenitor Cell Models of Alcohol Abuse.

Authors:  Eileen N Oni; Ronald P Hart
Journal:  Curr Pharmacol Rep       Date:  2016-08-05

7.  Chronic Ethanol Exposure Alters DNA Methylation in Neural Stem Cells: Role of Mouse Strain and Sex.

Authors:  Shayan Amiri; James R Davie; Mojgan Rastegar
Journal:  Mol Neurobiol       Date:  2019-08-14       Impact factor: 5.590

8.  CUL3 Deficiency Causes Social Deficits and Anxiety-like Behaviors by Impairing Excitation-Inhibition Balance through the Promotion of Cap-Dependent Translation.

Authors:  Zhaoqi Dong; Wenbing Chen; Chao Chen; Hongsheng Wang; Wanpeng Cui; Zhibing Tan; Heath Robinson; Nannan Gao; Bin Luo; Lei Zhang; Kai Zhao; Wen-Cheng Xiong; Lin Mei
Journal:  Neuron       Date:  2019-11-25       Impact factor: 17.173

Review 9.  Abnormalities of the DNA methylation mark and its machinery: an emerging cause of neurologic dysfunction.

Authors:  Jacqueline Weissman; Sakkubai Naidu; Hans T Bjornsson
Journal:  Semin Neurol       Date:  2014-09-05       Impact factor: 3.420

Review 10.  Reciprocal control of translation and transcription in autism spectrum disorder.

Authors:  Francesco Longo; Eric Klann
Journal:  EMBO Rep       Date:  2021-05-11       Impact factor: 9.071

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