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Cited by 178Genetics and Neurodevelopmental DisordersAutism Spectrum Disorder ResearchWilliams Syndrome Research

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Genetic-Background Modulation of Core and Variable Autistic-Like Symptoms in Fmr1 Knock-Out Mice

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Pietropaolo, Susanna, Guilleminot, Aurélie, Martin, Benoît, D'Amato, Francesca R., Crusio, Wim E.·2011·PLoS ONE
DOI 10.1371/journal.pone.0017073PMC PMC3043074
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Abstract

BACKGROUND: No animal models of autism spectrum disorders (ASD) with good construct validity are currently available; using genetic models of pathologies characterized by ASD-like deficits, but with known causes, may be therefore a promising strategy. The Fmr1-KO mouse is an example of this approach, modeling Fragile X syndrome, a well-known genetic disorder presenting ASD symptoms. The Fmr1-KO is available on different genetic backgrounds (FVB versus C57BL/6), which may explain some of the conflicting results that have been obtained with these mutants up till now. METHODS: Fmr1 KO and their wild-type littermates on both the FVB and C57BL/6 genetic backgrounds were examined on a battery of tests modeling the clinical symptoms of ASD, including the triad of core symptoms (alterations in social interaction and communication, presence of repetitive behaviors), as well as the secondary symptoms (disturbances in sensori-motor reactivity and in circadian patterns of activity, epileptic events). RESULTS: Fmr1-KO mice displayed autistic-like core symptoms of altered social interaction and occurrence of repetitive behaviors with additional hyperactivity. The genetic background modulated the effects of the Fmr1 deletion and it appears that the C57BL/6 background may be more suitable for further research on core autistic-like symptoms. CONCLUSIONS: The Fmr1-mouse line does not recapitulate all of the main core and secondary ASD symptoms, but still can be useful to elucidate the neurobiological mechanisms underlying specific ASD-like endophenotypes.

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Figures

Figure 1 Schematic representation of the apparatus used for the three compartment test. The apparatus consisted of three rectangular compartments made of transparent Plexiglas. A stimulus cage (wire mesh, 10 cm in diameter, here represented as a dark grey circle) allowing visual, olfactory and partial tactile contact was placed in each side compartment. Performance in the task was evaluated across three 5-min trials, based on the relative time spent in the contact areas (highlighted in light grey).
Figure 1 Schematic representation of the apparatus used for the three compartment test. The apparatus consisted of three rectangular compartments made of transparent Plexiglas. A stimulus cage (wire mesh, 10 cm in diameter, here represented as a dark grey circle) allowing visual, olfactory and partial tactile contact was placed in each side compartment. Performance in the task was evaluated across three 5-min trials, based on the relative time spent in the contact areas (highlighted in light grey).CC BY · PubMed Central
Figure 2 Sociability (A) and preference for social novelty (B) in the three compartment test. Exploration of each stimulus was assessed by measuring the time spent in each contact area, i.e., a 20×22 cm area containing the stimulus cage. Data are mean ± SEM. *p<0.05. The dotted line represents chance level (50%).
Figure 2 Sociability (A) and preference for social novelty (B) in the three compartment test. Exploration of each stimulus was assessed by measuring the time spent in each contact area, i.e., a 20×22 cm area containing the stimulus cage. Data are mean ± SEM. *p<0.05. The dotted line represents chance level (50%).CC BY · PubMed Central
Figure 3 Aggression and affiliation in the direct social interaction with a juvenile male mouse. A: Attack latency (for non-attacking mice the maximum value of 180s was assigned). B: Time spent in affiliative behavior. Data are mean ± SEM. *p<0.05.
Figure 3 Aggression and affiliation in the direct social interaction with a juvenile male mouse. A: Attack latency (for non-attacking mice the maximum value of 180s was assigned). B: Time spent in affiliative behavior. Data are mean ± SEM. *p<0.05.CC BY · PubMed Central
Figure 4 Startle reactivity and prepulse inhibition (PPI). A-B: Startle reactivity during the first and the last block of pulse alone trials; C–D: PPI expressed as the linear coefficient of the reactivity data. Data are mean ± SEM. *p<0.05.
Figure 4 Startle reactivity and prepulse inhibition (PPI). A-B: Startle reactivity during the first and the last block of pulse alone trials; C–D: PPI expressed as the linear coefficient of the reactivity data. Data are mean ± SEM. *p<0.05.CC BY · PubMed Central
Figure 5 Locomotor habituation and circadian modulation of activity. A-B: Locomotion during the first testing hour; C–D: Total activity during the subsequent 24 hrs; E: 24 hr activity profile. Data are mean ± SEM. *p<0.05.
Figure 5 Locomotor habituation and circadian modulation of activity. A-B: Locomotion during the first testing hour; C–D: Total activity during the subsequent 24 hrs; E: 24 hr activity profile. Data are mean ± SEM. *p<0.05.CC BY · PubMed Central
Figure 6 Social and non-social behaviors in the direct social interaction with an adult female. A: Time spent on affiliative behaviors towards an adult female. B: Frequency of attempts to mount the female. C: Self-grooming. D. Time spent on non-social activities. Data are mean ± SEM. *p<0.05.
Figure 6 Social and non-social behaviors in the direct social interaction with an adult female. A: Time spent on affiliative behaviors towards an adult female. B: Frequency of attempts to mount the female. C: Self-grooming. D. Time spent on non-social activities. Data are mean ± SEM. *p<0.05.CC BY · PubMed Central
Figure 7 Ultrasonic vocalizations during the direct social interaction with an adult female. A: Frequency. B: Mean duration. Data are mean ± SEM. *p<0.05.
Figure 7 Ultrasonic vocalizations during the direct social interaction with an adult female. A: Frequency. B: Mean duration. Data are mean ± SEM. *p<0.05.CC BY · PubMed Central
Figure 8 EEG analysis. Left: Examples of bipolar electrocorticographic recordings from each experimental group, illustrating the clear absence of any seizures, absences, or interictal events. Right: Electrode placements. 1 and 2: frontal cortex. 3 and 4: parietal cortex. Ref: cerebellar reference electrode.
Figure 8 EEG analysis. Left: Examples of bipolar electrocorticographic recordings from each experimental group, illustrating the clear absence of any seizures, absences, or interictal events. Right: Electrode placements. 1 and 2: frontal cortex. 3 and 4: parietal cortex. Ref: cerebellar reference electrode.CC BY · PubMed Central

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Research works printed but not linked: 1

  1. BK Yee, 2005, The expression of prepulse inhibition of the acoustic startle reflex as a function of three pulse stimulus intensities, three prepulse stimulus intensities, and three levels of startle responsiveness in C57BL6/J mice., Behav Brain Res, 163, 265, 10.1016/j.bbr.2005.05.013 · DOI

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Metadata

Authors
Pietropaolo, S., Guilleminot, A., Martin, B., D'Amato, F. R., & Crusio, W. E.
Affiliations
  • Centre National de la Recherche Scientifique (FR)
  • Inserm (FR)
  • National Research Council (IT)
Corresponding author
Crusio, Wim E.
ORCID
Venue
PLoS ONE
Publisher
Public Library of Science (PLoS)
Published
2011-02-22
Volume
6
Issue
2
Pages
e17073
ISSN
1932-6203 (ISSN-L)
DOI
10.1371/journal.pone.0017073
PMC
PMC3043074
OpenAlex
W2002465714
Cited by
178
References
82
Citations by year
2026: 7 · 2025: 7 · 2024: 15 · 2023: 11 · 2022: 13 · 2021: 12 · 2020: 10 · 2019: 12 · 2018: 10 · 2017: 10 · 2016: 15 · 2015: 15 · 2014: 15 · 2013: 9 · 2012: 11
FWCI
8.18
Type
journal article
Language
en
Open access
Licence
Locations
  • PLoS ONE (publishedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • HAL (submittedVersion, other-oa)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
Funders
  • March of Dimes Foundation (12-FY05-1198)
  • Institut National de la Santé et de la Recherche Médicale
  • Centre National de la Recherche Scientifique (12-FY05-1198)
  • Université de Bordeaux (12-FY05-1198)
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
  • Life Sciences › Neuroscience › Cognitive Neuroscience › Autism Spectrum Disorder Research
  • Life Sciences › Neuroscience › Developmental Neuroscience › Williams Syndrome Research
Keywords
autism spectrum disorder, Fmr1 knockout mice, fragile X syndrome, genetic background, FVB mice, C57BL/6 mice, social interaction deficits, repetitive behavior, hyperactivity, circadian activity, animal models of autism, endophenotypes
MeSH terms
Animals, Autistic Disorder, Behavior, Animal, Disease Models, Animal, Learning, Male, Mice, Inbred C57BL, Motor Activity, Phenotype, Mice, Knockout, Genetic Heterogeneity, Genetic Predisposition to Disease, Mice, Fragile X Messenger Ribonucleoprotein 1, Endophenotypes

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