Motor deficits on a ladder rung task in male and female adolescent and adult CGG knock-in mice

Michael R. Hunsaker, Ramona E. von Leden, Binh T. Ta, Naomi J. Goodrich-Hunsaker, Gloria Arque, Kyoungmi Kim, Rob Willemsen, Robert F Berman

Research output: Contribution to journalArticlepeer-review

35 Scopus citations

Abstract

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.

Original languageEnglish (US)
Pages (from-to)117-121
Number of pages5
JournalBehavioural Brain Research
Volume222
Issue number1
DOIs
StatePublished - Sep 12 2011

Keywords

  • Endophenotype
  • Fragile X premutation
  • FXTAS
  • Motor function
  • Neurodegenerative disorder
  • Neurodevelopmental disorder
  • Transgenic mice

ASJC Scopus subject areas

  • Behavioral Neuroscience

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