Literature DB >> 31229631

Transcriptomic Studies in Mouse Models of Rett Syndrome: A Review.

Albert Sanfeliu1, Walter E Kaufmann2, Michael Gill1, Paolo Guasoni3, Daniela Tropea4.   

Abstract

Rett Syndrome (RTT) is a neurological disorder mainly associated with mutations in the X-linked gene coding for the methyl-CpG binding protein 2 (MECP2). To assist in studying MECP2's function, researchers have generated Mecp2 mouse mutants showing that MECP2's product (MeCP2) mostly functions as a transcriptional regulator. During the last two decades, these models have been used to determine the genes that are regulated by MeCP2, slowly dissecting the etiological mechanisms underlying RTT. In the present review, we describe the findings of these transcriptomic studies, and highlight differences between them, and discuss how studies on these genetic models can sharpen our understanding of the human disorder. We conclude that - while there's large variability regarding the number of differentially expressed genes identified - there are overlapping features that inform on the biology of RTT.
Copyright © 2019 IBRO. Published by Elsevier Ltd. All rights reserved.

Entities:  

Keywords:  Mecp2; Rett syndrome; mouse models; transcriptomics

Mesh:

Substances:

Year:  2019        PMID: 31229631     DOI: 10.1016/j.neuroscience.2019.06.013

Source DB:  PubMed          Journal:  Neuroscience        ISSN: 0306-4522            Impact factor:   3.590


  5 in total

1.  Analysis of the circRNA and T-UCR populations identifies convergent pathways in mouse and human models of Rett syndrome.

Authors:  Edilene Siqueira; Aida Obiols-Guardia; Olga C Jorge-Torres; Cristina Oliveira-Mateos; Marta Soler; Deepthi Ramesh-Kumar; Fernando Setién; Daniëlle van Rossum; Ainhoa Pascual-Alonso; Clara Xiol; Cristina Ivan; Masayoshi Shimizu; Judith Armstrong; George A Calin; R Jeroen Pasterkamp; Manel Esteller; Sonia Guil
Journal:  Mol Ther Nucleic Acids       Date:  2021-12-22       Impact factor: 8.886

Review 2.  Proteomic and transcriptional changes associated with MeCP2 dysfunction reveal nodes for therapeutic intervention in Rett syndrome.

Authors:  Ketan Marballi; Jessica L MacDonald
Journal:  Neurochem Int       Date:  2021-05-26       Impact factor: 4.297

3.  MeCP2 gates spatial learning-induced alternative splicing events in the mouse hippocampus.

Authors:  David V C Brito; Kubra Gulmez Karaca; Janina Kupke; Lukas Frank; Ana M M Oliveira
Journal:  Mol Brain       Date:  2020-11-17       Impact factor: 4.041

Review 4.  Transcriptomic and Epigenomic Landscape in Rett Syndrome.

Authors:  Domenico Marano; Salvatore Fioriniello; Maurizio D'Esposito; Floriana Della Ragione
Journal:  Biomolecules       Date:  2021-06-30

Review 5.  Role of phosphodiesterases in the pathophysiology of neurodevelopmental disorders.

Authors:  Sébastien Delhaye; Barbara Bardoni
Journal:  Mol Psychiatry       Date:  2021-01-07       Impact factor: 15.992

  5 in total

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