Abstract:
:Fragile X syndrome, the most frequent form of hereditary mental retardation, is due to a mutation of the fragile X mental retardation 1 (FMR1) gene on the X chromosome. Like fragile X patients, FMR1-knockout (FMR1-KO) mice lack the normal fragile X mental retardation protein (FMRP) and show both cognitive alterations and an immature neuronal morphology. We reared FMR1-KO mice in a C57BL/6 background in enriched environmental conditions to examine the possibility that experience-dependent stimulation alleviates their behavioral and neuronal abnormalities. FMR1-KO mice kept in standard cages were hyperactive, displayed an altered pattern of open field exploration, and did not show habituation. Quantitative morphological analyses revealed a reduction in basal dendrite length and branching together with more immature-appearing spines along apical dendrites of layer five pyramidal neurons in the visual cortex. Enrichment largely rescued these behavioral and neuronal abnormalities while increasing alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid (AMPA) glutamate receptor subunit 1 (GluR1) levels in both genotypes. Enrichment did not, however, affect FMRP levels in the WT mice. These data suggest that FMRP-independent pathways activating glutamatergic signaling are preserved in FMR1-KO mice and that they can be elicited by environmental stimulation.
journal_name
Proc Natl Acad Sci U S Aauthors
Restivo L,Ferrari F,Passino E,Sgobio C,Bock J,Oostra BA,Bagni C,Ammassari-Teule Mdoi
10.1073/pnas.0504984102keywords:
subject
Has Abstractpub_date
2005-08-09 00:00:00pages
11557-62issue
32eissn
0027-8424issn
1091-6490pii
0504984102journal_volume
102pub_type
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pub_type: 杂志文章
doi:10.1073/pnas.92.3.826
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更新日期:1977-08-01 00:00:00
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更新日期:1969-09-01 00:00:00
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journal_title:Proceedings of the National Academy of Sciences of the United States of America
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更新日期:1982-08-01 00:00:00
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更新日期:1998-03-17 00:00:00
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journal_title:Proceedings of the National Academy of Sciences of the United States of America
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更新日期:1995-06-06 00:00:00
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journal_title:Proceedings of the National Academy of Sciences of the United States of America
pub_type: 杂志文章
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更新日期:1979-02-01 00:00:00