| Early postnatal behavioral changes in the Mecp2-308 truncation mouse model of Rett syndrome. | |
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MedLine Citation:
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PMID: 19958389 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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In a mouse model of Rett syndrome (RTT) which expresses a truncated form of methyl-CpG-binding protein 2 (Mecp2) gene (Mecp2-308), we performed a neurobehavioral evaluation across the life span, starting from soon after birth till adulthood. A focus was made on those developmental phases and behavioral domains which have not been previously investigated. The results evidenced subtle anomalies on postnatal days (pnds) 3 to 9 (so-called presymptomatic phase) in spontaneous movements by hemizygous neonatal male mice. Specifically as early as pnd 3, mutant pups exhibited more intense curling and more side responses and on pnd 9 more pivoting and head rising behaviors than wild type (wt) littermates. A significant decrease in ultrasonic vocalization rate, also emerged in Mecp2-308 pups. The same mice were also characterized by increased anxiety-like behaviors (open-field and zero-maze tests) during the early symptomatic phase, in the absence of changes in cognitive passive-avoidance task and rotarod performances. Upon the clearly symptomatic stage, 5-month-old Mecp2-308 mice were also associated with reduced spontaneous home-cage motor activity, motor coordination impairments (rotarod and dowel tests), and a more marked profile of D-amphetamine (10 mg/kg) released stereotyped behavioral syndrome than wt mice. Present results provide an interesting timeline of the progression of symptoms in the Mecp2-308 model and emphasize the need for increased attention to the presymptomatic phase which may be especially informative in mouse models of human neurodevelopmental disorders. This analysis has provided evidence of precocious behavioral markers of RTT and has identified an early developmental window of opportunities on which potential therapies could be investigated. |
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Authors:
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B De Filippis; L Ricceri; G Laviola |
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Publication Detail:
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Type: Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't Date: 2009-11-02 |
Journal Detail:
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Title: Genes, brain, and behavior Volume: 9 ISSN: 1601-183X ISO Abbreviation: Genes Brain Behav. Publication Date: 2010 Mar |
Date Detail:
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Created Date: 2010-05-07 Completed Date: 2010-08-10 Revised Date: - |
Medline Journal Info:
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Nlm Unique ID: 101129617 Medline TA: Genes Brain Behav Country: England |
Other Details:
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Languages: eng Pagination: 213-23 Citation Subset: IM |
Affiliation:
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Section of Behavioural Neuroscience, Department of Cell Biology & Neuroscience, Istituto Superiore di Sanita, Roma, Italy. |
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| MeSH Terms | |
Descriptor/Qualifier:
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Animals Anxiety / genetics Cognition / physiology DNA Primers Darkness Disease Models, Animal Female Humans Light Male Methyl-CpG-Binding Protein 2 / genetics* Mice Motor Activity / genetics, physiology Psychomotor Performance / physiology Rett Syndrome / epidemiology, genetics* Sequence Deletion Stereotyped Behavior / physiology Vocalization, Animal / physiology |
| Grant Support | |
ID/Acronym/Agency:
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5301/530H//PHS HHS; 7NR1/1/NR/NINR NIH HHS |
| Chemical | |
Reg. No./Substance:
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0/DNA Primers; 0/MECP2 protein, human; 0/Mecp2 protein, mouse; 0/Methyl-CpG-Binding Protein 2 |
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