期刊
NEUROBIOLOGY OF LEARNING AND MEMORY
卷 97, 期 2, 页码 229-234出版社
ACADEMIC PRESS INC ELSEVIER SCIENCE
DOI: 10.1016/j.nlm.2011.12.006
关键词
Fragile X premutation; Transgenic mice; Motor function; Skilled reaching; Fragile X-associated disorders
资金
- National Institute of Health (NIH) [NINDS RL1 NS062411, TL1 DA024854]
- National Institute of Dental and Craniofacial Research (NIDCR) [UL1 DE019583]
- National Center for Research Resources (NCRR), National Institutes of Health (NIH) [UL1 RR024146]
- NIH Roadmap for Medical Research
The fragile X premutation is a tandem CGG trinucleotide repeat expansion in the fragile X mental retardation 1 (FMR1) gene between 55 and 200 repeats in length. A CGG knock-in (CGG RI) mouse has been developed that models the neuropathology and cognitive deficits reported in fragile X premutation carriers. Previous studies have demonstrated that CGG KI mice have spatiotemporal information processing deficits and impaired visuomotor function that worsen with increasing CGG repeat length. Since skilled forelimb reaching requires integration of information from the visual and motor systems, skilled reaching performance could identify potential visuomotor dysfunction in CGG la mice. To characterize motor deficits associated with the fragile X premutation, 6 month old female CGG RI mice heterozygous for trinucleotide repeats ranging from 70-200 CGG in length were tested for their ability to learn a skilled forelimb reaching task. The results demonstrate that female CGG KI mice show deficits for learning a skilled forelimb reaching task compared to wildtype littermates, and that these deficits worsen with increasing CGG repeat lengths. (C) 2011 Elsevier Inc. All rights reserved.
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