Unconventional transcriptional response to environmental enrichment in a mouse model of Rett syndrome.
BACKGROUND: Rett syndrome (RTT) is an X-linked postnatal neurodevelopmental disorder caused by mutations in the gene encoding methyl-CpG binding protein 2 (MeCP2) and one of the leading causes of mental retardation in females. RTT is characterized by psychomotor retardation, purposeless hand movemen...
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doaj-4e5d8ffb00d34f72b756a599caf461e62020-11-25T02:32:11ZengPublic Library of Science (PLoS)PLoS ONE1932-62032010-01-0157e1153410.1371/journal.pone.0011534Unconventional transcriptional response to environmental enrichment in a mouse model of Rett syndrome.Bredford KerrPamela A SilvaKatherina WalzJuan I YoungBACKGROUND: Rett syndrome (RTT) is an X-linked postnatal neurodevelopmental disorder caused by mutations in the gene encoding methyl-CpG binding protein 2 (MeCP2) and one of the leading causes of mental retardation in females. RTT is characterized by psychomotor retardation, purposeless hand movements, autistic-like behavior and abnormal gait. We studied the effects of environmental enrichment (EE) on the phenotypic manifestations of a RTT mouse model that lacks MeCP2 (Mecp2(-/y)). PRINCIPAL FINDINGS: We found that EE delayed and attenuated some neurological alterations presented by Mecp2(-/y) mice and prevented the development of motor discoordination and anxiety-related abnormalities. To define the molecular correlate of this beneficial effect of EE, we analyzed the expression of several synaptic marker genes whose expression is increased by EE in several mouse models. CONCLUSIONS/SIGNIFICANCE: We found that EE induced downregulation of several synaptic markers, suggesting that the partial prevention of RTT-associated phenotypes is achieved through a non-conventional transcriptional program.http://europepmc.org/articles/PMC2902516?pdf=render |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Bredford Kerr Pamela A Silva Katherina Walz Juan I Young |
spellingShingle |
Bredford Kerr Pamela A Silva Katherina Walz Juan I Young Unconventional transcriptional response to environmental enrichment in a mouse model of Rett syndrome. PLoS ONE |
author_facet |
Bredford Kerr Pamela A Silva Katherina Walz Juan I Young |
author_sort |
Bredford Kerr |
title |
Unconventional transcriptional response to environmental enrichment in a mouse model of Rett syndrome. |
title_short |
Unconventional transcriptional response to environmental enrichment in a mouse model of Rett syndrome. |
title_full |
Unconventional transcriptional response to environmental enrichment in a mouse model of Rett syndrome. |
title_fullStr |
Unconventional transcriptional response to environmental enrichment in a mouse model of Rett syndrome. |
title_full_unstemmed |
Unconventional transcriptional response to environmental enrichment in a mouse model of Rett syndrome. |
title_sort |
unconventional transcriptional response to environmental enrichment in a mouse model of rett syndrome. |
publisher |
Public Library of Science (PLoS) |
series |
PLoS ONE |
issn |
1932-6203 |
publishDate |
2010-01-01 |
description |
BACKGROUND: Rett syndrome (RTT) is an X-linked postnatal neurodevelopmental disorder caused by mutations in the gene encoding methyl-CpG binding protein 2 (MeCP2) and one of the leading causes of mental retardation in females. RTT is characterized by psychomotor retardation, purposeless hand movements, autistic-like behavior and abnormal gait. We studied the effects of environmental enrichment (EE) on the phenotypic manifestations of a RTT mouse model that lacks MeCP2 (Mecp2(-/y)). PRINCIPAL FINDINGS: We found that EE delayed and attenuated some neurological alterations presented by Mecp2(-/y) mice and prevented the development of motor discoordination and anxiety-related abnormalities. To define the molecular correlate of this beneficial effect of EE, we analyzed the expression of several synaptic marker genes whose expression is increased by EE in several mouse models. CONCLUSIONS/SIGNIFICANCE: We found that EE induced downregulation of several synaptic markers, suggesting that the partial prevention of RTT-associated phenotypes is achieved through a non-conventional transcriptional program. |
url |
http://europepmc.org/articles/PMC2902516?pdf=render |
work_keys_str_mv |
AT bredfordkerr unconventionaltranscriptionalresponsetoenvironmentalenrichmentinamousemodelofrettsyndrome AT pamelaasilva unconventionaltranscriptionalresponsetoenvironmentalenrichmentinamousemodelofrettsyndrome AT katherinawalz unconventionaltranscriptionalresponsetoenvironmentalenrichmentinamousemodelofrettsyndrome AT juaniyoung unconventionaltranscriptionalresponsetoenvironmentalenrichmentinamousemodelofrettsyndrome |
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