| Literature DB >> 31379106 |
Rahul Krishnaraj1, Florencia Haase2, Bronte Coorey2, Edward J Luca3, Ingar Wong2, Alexandra Boyling2, Carolyn Ellaway1,4,5, John Christodoulou1,4,5,6, Wendy A Gold1,2,4,7.
Abstract
The discovery that Rett syndrome is caused by mutations in the MECP2 gene has provided a major breakthrough in our understanding of the disorder. However, despite this, there is still limited understanding of the underlying pathophysiology of the disorder hampering the development of curative treatments. Over the years, a number of animal models have been developed contributing to our knowledge of the role of MECP2 in development and improving our understanding of how subtle expression levels affect brain morphology and function. Transcriptomic and proteomic studies of animal models are useful in identifying perturbations in functional pathways and providing avenues for novel areas of research into disease. This review focuses on published transcriptomic and proteomic studies of mouse models of Rett syndrome with the aim of providing a summary of all the studies, the reported dysregulated genes and functional pathways that are found to be perturbed. The 36 articles identified highlighted a number of dysfunctional pathways as well as perturbed biological networks and cellular functions including synaptic dysfunction and neuronal transmission, inflammation, and mitochondrial dysfunction. These data reveal biological insights that contribute to the disease process which may be targeted to investigate curative treatments.Entities:
Keywords: MECP2; Rett syndrome; mouse model; pathways; proteomic; transcriptomic
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Year: 2019 PMID: 31379106 DOI: 10.1002/humu.23887
Source DB: PubMed Journal: Hum Mutat ISSN: 1059-7794 Impact factor: 4.878