AUTHOR=Saré R. Michelle , Song Alex , Levine Merlin , Lemons Abigail , Loutaev Inna , Sheeler Carrie , Hildreth Christine , Mfon Angel , Smith Carolyn Beebe 

TITLE=Behavioral and Molecular Consequences of Chronic Sleep Restriction During Development in Fragile X Mice

JOURNAL=Frontiers in Neuroscience

VOLUME=16

YEAR=2022

URL=https://www.frontiersin.org/journals/neuroscience/articles/10.3389/fnins.2022.834890

DOI=10.3389/fnins.2022.834890

ISSN=1662-453X

ABSTRACT=<p>Sleep is critical for brain development and synaptic plasticity. In male wild-type mice, chronic sleep restriction during development results in long-lasting impairments in behavior including hypoactivity, decreased sociability, and increased repetitive behavior. Disordered sleep is characteristic of many neurodevelopmental disorders. Moreover, the severity of behavioral symptoms is correlated with the degree of disordered sleep. We hypothesized that chronic developmental sleep restriction in a mouse model of fragile X syndrome (FXS) would exacerbate behavioral phenotypes. To test our hypothesis, we sleep-restricted <italic>Fmr1</italic> knockout (KO) mice for 3 h per day from P5 to P52 and subjected mice to behavioral tests beginning on P42. Contrary to our expectations, sleep restriction improved the hyperactivity and lack of preference for social novelty phenotypes in <italic>Fmr1</italic> KO mice but had no measurable effect on repetitive activity. Sleep restriction also resulted in changes in regional distribution of myelin basic protein, suggesting effects on myelination. These findings have implications for the role of disrupted sleep in the severity of symptoms in FXS.</p>