Literature DB >> 17289941

Reversal of neurological defects in a mouse model of Rett syndrome.

Jacky Guy1, Jian Gan, Jim Selfridge, Stuart Cobb, Adrian Bird.   

Abstract

Rett syndrome is an autism spectrum disorder caused by mosaic expression of mutant copies of the X-linked MECP2 gene in neurons. However, neurons do not die, which suggests that this is not a neurodegenerative disorder. An important question for future therapeutic approaches to this and related disorders concerns phenotypic reversibility. Can viable but defective neurons be repaired, or is the damage done during development without normal MeCP2 irrevocable? Using a mouse model, we demonstrate robust phenotypic reversal, as activation of MeCP2 expression leads to striking loss of advanced neurological symptoms in both immature and mature adult animals.

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Year:  2007        PMID: 17289941      PMCID: PMC7610836          DOI: 10.1126/science.1138389

Source DB:  PubMed          Journal:  Science        ISSN: 0036-8075            Impact factor:   47.728


  15 in total

Review 1.  DNA methylation and Rett syndrome.

Authors:  Skirmantas Kriaucionis; Adrian Bird
Journal:  Hum Mol Genet       Date:  2003-08-19       Impact factor: 6.150

2.  Purification, sequence, and cellular localization of a novel chromosomal protein that binds to methylated DNA.

Authors:  J D Lewis; R R Meehan; W J Henzel; I Maurer-Fogy; P Jeppesen; F Klein; A Bird
Journal:  Cell       Date:  1992-06-12       Impact factor: 41.582

3.  MeCP2 is a transcriptional repressor with abundant binding sites in genomic chromatin.

Authors:  X Nan; F J Campoy; A Bird
Journal:  Cell       Date:  1997-02-21       Impact factor: 41.582

4.  Deficiency of methyl-CpG binding protein-2 in CNS neurons results in a Rett-like phenotype in mice.

Authors:  R Z Chen; S Akbarian; M Tudor; R Jaenisch
Journal:  Nat Genet       Date:  2001-03       Impact factor: 38.330

5.  A mouse Mecp2-null mutation causes neurological symptoms that mimic Rett syndrome.

Authors:  J Guy; B Hendrich; M Holmes; J E Martin; A Bird
Journal:  Nat Genet       Date:  2001-03       Impact factor: 38.330

6.  Rett syndrome is caused by mutations in X-linked MECP2, encoding methyl-CpG-binding protein 2.

Authors:  R E Amir; I B Van den Veyver; M Wan; C Q Tran; U Francke; H Y Zoghbi
Journal:  Nat Genet       Date:  1999-10       Impact factor: 38.330

7.  Selective dendritic alterations in the cortex of Rett syndrome.

Authors:  D Armstrong; J K Dunn; B Antalffy; R Trivedi
Journal:  J Neuropathol Exp Neurol       Date:  1995-03       Impact factor: 3.685

Review 8.  Rett syndrome: a prototypical neurodevelopmental disorder.

Authors:  Jeffrey L Neul; Huda Y Zoghbi
Journal:  Neuroscientist       Date:  2004-04       Impact factor: 7.519

9.  Mild overexpression of MeCP2 causes a progressive neurological disorder in mice.

Authors:  Ann L Collins; Jonathan M Levenson; Alexander P Vilaythong; Ronald Richman; Dawna L Armstrong; Jeffrey L Noebels; J David Sweatt; Huda Y Zoghbi
Journal:  Hum Mol Genet       Date:  2004-09-06       Impact factor: 6.150

10.  A method for the generation of conditional gene repair mutations in mice.

Authors:  I Dragatsis; S Zeitlin
Journal:  Nucleic Acids Res       Date:  2001-02-01       Impact factor: 16.971

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  492 in total

Review 1.  Stress and the epigenetic landscape: a link to the pathobiology of human diseases?

Authors:  Sarah E Johnstone; Stephen B Baylin
Journal:  Nat Rev Genet       Date:  2010-10-05       Impact factor: 53.242

2.  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

3.  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 4.  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 5.  Networking in autism: leveraging genetic, biomarker and model system findings in the search for new treatments.

Authors:  Jeremy Veenstra-VanderWeele; Randy D Blakely
Journal:  Neuropsychopharmacology       Date:  2011-09-21       Impact factor: 7.853

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

Review 7.  Stem cells and modeling of autism spectrum disorders.

Authors:  Beatriz C G Freitas; Cleber A Trujillo; Cassiano Carromeu; Marianna Yusupova; Roberto H Herai; Alysson R Muotri
Journal:  Exp Neurol       Date:  2012-10-02       Impact factor: 5.330

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

Review 10.  Recent advances in MeCP2 structure and function.

Authors:  Kristopher C Hite; Valerie H Adams; Jeffrey C Hansen
Journal:  Biochem Cell Biol       Date:  2009-02       Impact factor: 3.626

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