ArticleTranslational psychiatry2020
Reduced axonal caliber and structural changes in a rat model of Fragile X syndrome with a deletion of a K-Homology domain of Fmr1.
Article in Translational psychiatry, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.
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Who cites it
7 citing papers in PubMed, 8 citations in OpenAlex.
- A high-resolution phenotypic screen identifies regulators of CNS axon diameter in zebrafish.PLoS biology · 2026Article
- Visual function and autism spectrum disorder.Journal of neural transmission (Vienna, Austria : 1996) · 2026Review
- Altered olfactory responses in Fmr1 KO mice.Scientific reports · 2025Article
- Importin 13-dependent axon diameter growth regulates conduction speeds along myelinated CNS axons.Nature communications · 2024Article
- Mapping the neuroanatomical abnormalities in a phenotype of male compulsive rats.Behavioral and brain functions : BBF · 2023Article
- Reduced brain volume and white matter alterations in Shank3-deficient rats.Autism research : official journal of the International Society for Autism Research · 2021Article
- Bundle-Specific Axon Diameter Index as a New Contrast to Differentiate White Matter Tracts.Frontiers in neuroscience · 2021Article
Corrections and comments
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Authors and funding
7 authors at 3 institutions in 3 countries.
Funding
Abstract
Fragile X syndrome (FXS) is a neurodevelopmental disorder that is caused by mutations in the FMR1 gene. Neuroanatomical alterations have been reported in both male and female individuals with FXS, yet the morphological underpinnings of these alterations have not been elucidated. In the current study, we found structural changes in both male and female rats that model FXS, some of which are similarly impaired in both sexes, including the superior colliculus and periaqueductal gray, and others that show sex-specific changes. The splenium of the corpus callosum, for example, was only impaired in males. We also found reduced axonal caliber in the splenium, offering a mechanism for its structural changes. Furthermore, we found that overall, male rats have higher brain-wide diffusion than female rats. Our results provide insight into which brain regions are vulnerable to a loss of Fmr1 expression and reveal an impairment at the level of the axon that could cause structural changes in white matter regions.
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Registered trials
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