期刊
BEHAVIOURAL BRAIN RESEARCH
卷 222, 期 1, 页码 117-121出版社
ELSEVIER
DOI: 10.1016/j.bbr.2011.03.039
关键词
Fragile X premutation; Transgenic mice; Motor function; Endophenotype; Neurodevelopmental disorder; Neurodegenerative disorder; FXTAS
资金
- National Institute of Health (NIH) [NINDS RL1 NS062411, TL1 DA024854]
- National Institute of Dental and Craniofacial Research (NIDCR), NeuroTherapeutics Research Institute (NTRI) consortium [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 on the FMR1 gene between 55 and 200 repeats in length. A CGG knock-in (CGG KI) mouse with CGG trinucleotide repeat lengths between 70 and 350 has been developed and used to model the histopathology and cognitive deficits reported in carriers of the fragile X premutation. Previous studies have shown that CGG KI mice show progressive deficits in processing spatial and temporal information. To characterize the motor deficits associated with the fragile X premutation, male and female CGG KI mice ranging from 2 to 16 months of age with trinucleotide repeats ranging from 72 to 240 CGG in length were tested for their ability to perform a skilled ladder rung walking test. The results demonstrate that both male and female CGG KI mice showed a greater number of foot slips as a function of increased CGG repeat length, independent of the age of the animal or general activity level. (C) 2011 Elsevier B.V. All rights reserved.
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