Cited by 179Genetics and Neurodevelopmental DisordersMemory and Neural MechanismsDown syndrome and intellectual disability research

Fmr1 knockout mouse has a distinctive strain-specific learning impairment

Dobkin, Carl, Rabe, Ausma, Dumas, Ruth, El Idrissi, Abdeslem, Haubenstock, Harriet, Ted Brown, W·2000·Neuroscience

Abstract

The Fmr1 gene knockout mouse is a model for the human Fragile X mental retardation syndrome. Fmr1 knockout mice with a C57BL/6-129/OlaHsd hybrid background have been reported to have only a very mild deficiency in learning the Morris water maze task. We compared the effect of this knockout mutation on learning in mice with either an FVB/N-129/OlaHsd hybrid background or a C57BL/6 background. When FVB-129 mice were tested in a cross-shaped water maze task, the knockout mice showed a pronounced deficiency in their ability to learn the position of a hidden escape platform in comparison to normal littermates. In contrast, knockout mice with a C57BL/6 background learned the maze just as well as their normal littermates. Fear conditioning did not reveal differences between knockout and normal mice in either background. These results show that silencing the Fmr1 gene clearly interfered with learning a specific visuospatial task in FVB/N-129 hybrid mice but not in C57BL/6 mice. The strain dependence may model the influence of genetic background in the human Fragile X syndrome.

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Crossref · 10.1016/s0306-4522(00)00292-x

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Authors
Dobkin, C., Rabe, A., Dumas, R., El Idrissi, A., Haubenstock, H., & Ted Brown, W.
Corresponding author
Dobkin, Carl
Venue
Neuroscience
Publisher
Elsevier BV
Published
2000-09
Volume
100
Issue
2
Pages
423-429
ISSN
0306-4522 (ISSN-L)
OpenAlex
W2001065634
Cited by
179
References
9
Citations by year
2026: 2 · 2025: 1 · 2024: 3 · 2023: 2 · 2021: 2 · 2020: 5 · 2019: 8 · 2018: 7 · 2017: 5 · 2016: 7 · 2015: 5 · 2014: 16 · 2013: 8 · 2012: 5
FWCI
4.68
Type
journal article
Language
en
Open access
closed
Locations
  • Neuroscience (publishedVersion)
  • PubMed (publishedVersion)
Funders
  • FRAXA Research Foundation
  • New York State Developmental Disabilities Planning Council
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
  • Life Sciences › Neuroscience › Cognitive Neuroscience › Memory and Neural Mechanisms
  • Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Down syndrome and intellectual disability research
Keywords
Fmr1 knockout mice, fragile X syndrome, learning disabilities, visuospatial learning, Morris water maze, fear conditioning, genetic background, strain dependence, C57BL/6 mice, FMRP
MeSH terms
Animals, Conditioning, Psychological, Fear, Female, Fragile X Syndrome, Learning Disabilities, Male, Mice, Inbred BALB C, Species Specificity, Mice, Knockout, Maze Learning, Mice

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