Literature DB >> 16380085

Brain magnetic resonance study of Mecp2 deletion effects on anatomy and metabolism.

Véronique Saywell1, Angèle Viola, Sylviane Confort-Gouny, Yann Le Fur, Laurent Villard, Patrick J Cozzone.   

Abstract

Rett syndrome, a neurodevelopmental X-linked disorder, represents the most important genetic cause of severe mental retardation in the female population and results from a mutation in the gene encoding methyl-CpG-binding protein 2 (MECP2). We report here the first characterization of Mecp2-null mice, by in vivo magnetic resonance imaging and spectroscopy, delineating the cerebral phenotype associated with the lack of Mecp2. We performed a morphometric study that revealed a size reduction of the whole brain and of structures involved in cognitive and motor functions (cerebellum and motor cortex). Significant metabolic anomalies, including reduced N-acetylaspartate, myo-inositol, and glutamine plus glutamate, and increased choline levels were evidenced. These findings indicate that not only neuronal but also glial metabolism is affected in Mecp2-null mice. Furthermore, we uncovered an important reduction of brain ATP level, a hitherto undetected anomaly of energy metabolism that may reflect and contribute to cerebral injury and dysfunction.

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Year:  2005        PMID: 16380085     DOI: 10.1016/j.bbrc.2005.12.080

Source DB:  PubMed          Journal:  Biochem Biophys Res Commun        ISSN: 0006-291X            Impact factor:   3.575


  30 in total

1.  Mitochondrial Electron Transport Chain Complex Dysfunction in MeCP2 Knock-Down Astrocytes: Protective Effects of Quercetin Hydrate.

Authors:  Arpita Dave; Foram Shukla; Hemendra Wala; Prakash Pillai
Journal:  J Mol Neurosci       Date:  2018-12-06       Impact factor: 3.444

Review 2.  Behavioral and Neuroanatomical Phenotypes in Mouse Models of Autism.

Authors:  Jacob Ellegood; Jacqueline N Crawley
Journal:  Neurotherapeutics       Date:  2015-07       Impact factor: 7.620

Review 3.  Astrocytes conspire with neurons during progression of neurological disease.

Authors:  James C McGann; Daniel T Lioy; Gail Mandel
Journal:  Curr Opin Neurobiol       Date:  2012-04-03       Impact factor: 6.627

4.  Involvement of MeCP2 in Regulation of Myelin-Related Gene Expression in Cultured Rat Oligodendrocytes.

Authors:  Kedarlal Sharma; Juhi Singh; Prakash P Pillai; Emma E Frost
Journal:  J Mol Neurosci       Date:  2015-07-05       Impact factor: 3.444

5.  Surface- and voxel-based brain morphologic study in Rett and Rett-like syndrome with MECP2 mutation.

Authors:  Tadashi Shiohama; Jacob Levman; Emi Takahashi
Journal:  Int J Dev Neurosci       Date:  2019-01-25       Impact factor: 2.457

6.  Modulation of RhoGTPases improves the behavioral phenotype and reverses astrocytic deficits in a mouse model of Rett syndrome.

Authors:  Bianca De Filippis; Alessia Fabbri; Daiana Simone; Rossella Canese; Laura Ricceri; Fiorella Malchiodi-Albedi; Giovanni Laviola; Carla Fiorentini
Journal:  Neuropsychopharmacology       Date:  2011-12-07       Impact factor: 7.853

Review 7.  Neuroimaging endophenotypes in animal models of autism spectrum disorders: lost or found in translation?

Authors:  Marija M Petrinovic; Basil Künnecke
Journal:  Psychopharmacology (Berl)       Date:  2013-07-14       Impact factor: 4.530

8.  Temporal and regional alterations in NMDA receptor expression in Mecp2-null mice.

Authors:  Mary E Blue; Walter E Kaufmann; Joseph Bressler; Charlotte Eyring; Cliona O'driscoll; Sakkubai Naidu; Michael V Johnston
Journal:  Anat Rec (Hoboken)       Date:  2011-09-08       Impact factor: 2.064

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.  Does the effectiveness of the ketogenic diet in different epilepsies yield insights into its mechanisms?

Authors:  Adam L Hartman
Journal:  Epilepsia       Date:  2008-11       Impact factor: 5.864

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