Literature DB >> 17988628

The story of Rett syndrome: from clinic to neurobiology.

Maria Chahrour1, Huda Y Zoghbi.   

Abstract

The postnatal neurodevelopmental disorder Rett syndrome (RTT) is caused by mutations in the gene encoding methyl-CpG binding protein 2 (MeCP2), a transcriptional repressor involved in chromatin remodeling and the modulation of RNA splicing. MECP2 aberrations result in a constellation of neuropsychiatric abnormalities, whereby both loss of function and gain in MECP2 dosage lead to similar neurological phenotypes. Recent studies demonstrate disease reversibility in RTT mouse models, suggesting that the neurological defects in MECP2 disorders are not permanent. To investigate the potential for restoring neuronal function in RTT patients, it is essential to identify MeCP2 targets or modifiers of the phenotype that can be therapeutically modulated. Moreover, deciphering the molecular underpinnings of RTT is likely to contribute to the understanding of the pathogenesis of a broader class of neuropsychiatric disorders.

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Year:  2007        PMID: 17988628     DOI: 10.1016/j.neuron.2007.10.001

Source DB:  PubMed          Journal:  Neuron        ISSN: 0896-6273            Impact factor:   17.173


  529 in total

1.  Correction of respiratory disorders in a mouse model of Rett syndrome.

Authors:  Ana P L Abdala; Mathias Dutschmann; John M Bissonnette; Julian F R Paton
Journal:  Proc Natl Acad Sci U S A       Date:  2010-10-04       Impact factor: 11.205

2.  Genome-wide analysis reveals methyl-CpG-binding protein 2-dependent regulation of microRNAs in a mouse model of Rett syndrome.

Authors:  Hao Wu; Jifang Tao; Pauline J Chen; Atif Shahab; Weihong Ge; Ronald P Hart; Xiaoan Ruan; Yijun Ruan; Yi E Sun
Journal:  Proc Natl Acad Sci U S A       Date:  2010-10-04       Impact factor: 11.205

Review 3.  Complexities of Rett syndrome and MeCP2.

Authors:  Rodney C Samaco; Jeffrey L Neul
Journal:  J Neurosci       Date:  2011-06-01       Impact factor: 6.167

Review 4.  The contribution of GABAergic dysfunction to neurodevelopmental disorders.

Authors:  Kartik Ramamoorthi; Yingxi Lin
Journal:  Trends Mol Med       Date:  2011-04-21       Impact factor: 11.951

Review 5.  Oxytocin receptor and Mecp2 308/Y knockout mice exhibit altered expression of autism-related social behaviors.

Authors:  Roger L H Pobbe; Brandon L Pearson; D Caroline Blanchard; Robert J Blanchard
Journal:  Physiol Behav       Date:  2012-03-03

6.  Tsix-Mecp2 female mouse model for Rett syndrome reveals that low-level MECP2 expression extends life and improves neuromotor function.

Authors:  Lieselot L G Carrette; Roy Blum; Weiyuan Ma; Raymond J Kelleher; Jeannie T Lee
Journal:  Proc Natl Acad Sci U S A       Date:  2018-07-23       Impact factor: 11.205

7.  Brain activity mapping in Mecp2 mutant mice reveals functional deficits in forebrain circuits, including key nodes in the default mode network, that are reversed with ketamine treatment.

Authors:  Miriam Kron; C James Howell; Ian T Adams; Michael Ransbottom; Diana Christian; Michael Ogier; David M Katz
Journal:  J Neurosci       Date:  2012-10-03       Impact factor: 6.167

8.  Oligodendrocyte lineage cells contribute unique features to Rett syndrome neuropathology.

Authors:  Minh Vu Chuong Nguyen; Christy A Felice; Fang Du; Matthew V Covey; John K Robinson; Gail Mandel; Nurit Ballas
Journal:  J Neurosci       Date:  2013-11-27       Impact factor: 6.167

9.  Non-cell autonomous influence of MeCP2-deficient glia on neuronal dendritic morphology.

Authors:  Nurit Ballas; Daniel T Lioy; Christopher Grunseich; Gail Mandel
Journal:  Nat Neurosci       Date:  2009-02-22       Impact factor: 24.884

10.  Loss of MeCP2 from forebrain excitatory neurons leads to cortical hyperexcitation and seizures.

Authors:  Wen Zhang; Matthew Peterson; Barbara Beyer; Wayne N Frankel; Zhong-wei Zhang
Journal:  J Neurosci       Date:  2014-02-12       Impact factor: 6.167

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