Characteristics of Sleep and Wakefulness inWild-Derived Inbred          Mice

Hiyoshi, Hideyuki; Terao, Akira; Okamatsu‐Ogura, Yuko; Kimura, Kazuhiro

doi:10.1538/expanim.63.205

Cited by 8 publications

(6 citation statements)

References 25 publications

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“…Typically, mice are polyphasic, exhibiting multiple sleep bouts of sleep several minutes long within a 24-h period, and they are largely nocturnal, with more activity at night and consequently more sleep in the light period of the day ( 38 ). Analysis of the bouts of activity and sleep that make up the 24-h patterns revealed differences in the hemizygous Gria3 A653T mice, against a background of high variability between littermate individuals (as has been reported for wild-type animals previously ( 39 )).…”

Section: Resultsmentioning

confidence: 99%

A point mutation in the ion conduction pore of AMPA receptor GRIA3 causes dramatically perturbed sleep patterns as well as intellectual disability

Davies

Brown

Cais

et al. 2017

Human Molecular Genetics

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The discovery of genetic variants influencing sleep patterns can shed light on the physiological processes underlying sleep. As part of a large clinical sequencing project, WGS500, we sequenced a family in which the two male children had severe developmental delay and a dramatically disturbed sleep-wake cycle, with very long wake and sleep durations, reaching up to 106-h awake and 48-h asleep. The most likely causal variant identified was a novel missense variant in the X-linked GRIA3 gene, which has been implicated in intellectual disability. GRIA3 encodes GluA3, a subunit of AMPA-type ionotropic glutamate receptors (AMPARs). The mutation (A653T) falls within the highly conserved transmembrane domain of the ion channel gate, immediately adjacent to the analogous residue in the Grid2 (glutamate receptor) gene, which is mutated in the mouse neurobehavioral mutant, Lurcher. In vitro, the GRIA3(A653T) mutation stabilizes the channel in a closed conformation, in contrast to Lurcher. We introduced the orthologous mutation into a mouse strain by CRISPR-Cas9 mutagenesis and found that hemizygous mutants displayed significant differences in the structure of their activity and sleep compared to wild-type littermates. Typically, mice are polyphasic, exhibiting multiple sleep bouts of sleep several minutes long within a 24-h period. The Gria3A653T mouse showed significantly fewer brief bouts of activity and sleep than the wild-types. Furthermore, Gria3A653T mice showed enhanced period lengthening under constant light compared to wild-type mice, suggesting an increased sensitivity to light. Our results suggest a role for GluA3 channel activity in the regulation of sleep behavior in both mice and humans.

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Section: Resultsmentioning

confidence: 99%

A point mutation in the ion conduction pore of AMPA receptor GRIA3 causes dramatically perturbed sleep patterns as well as intellectual disability

Davies

Brown

Cais

et al. 2017

Human Molecular Genetics

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“…Currently, the most widely used animal models in sleep research are rodents, particularly rats and mice. However, unlike humans, rodents are nocturnal and exhibit polyphasic sleep–wake cycles [ 15 , 19 , 27 ]. Thus, non-human primates would represent valuable and more translational animal models of sleep.…”

Section: Discussionmentioning

confidence: 99%

Investigation of sleep–wake rhythm in non-human primates without restraint during data collection

Ishikawa

Sakai²,

Maki³

et al. 2017

Exp. Anim.

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To understand sleep mechanisms and develop treatments for sleep disorders, investigations using animal models are essential. The sleep architecture of rodents differs from that of diurnal mammals including humans and non-human primates. Sleep studies have been conducted in non-human primates; however, these sleep assessments were performed on animals placed in a restraint chair connected via the umbilical area to the recording apparatus. To avoid restraints, cables, and other stressful apparatuses and manipulations, telemetry systems have been developed. In the present study, sleep recordings in unrestrained cynomolgus monkeys (Macaca fascicularis) and common marmoset monkeys (Callithrix jacchus) were conducted to characterize normal sleep. For the analysis of sleep–wake rhythms in cynomolgus monkeys, telemetry electroencephalography (EEG), electromyography (EMG), and electrooculography (EOG) signals were used. For the analysis of sleep–wake rhythms in marmosets, telemetry EEG and EOG signals were used. Both monkey species showed monophasic sleep patterns during the dark phase. Although non-rapid eye movement (NREM) deep sleep showed higher levels at the beginning of the dark phase in cynomolgus monkeys, NREM deep sleep rarely occurred during the dark phase in marmosets. Our results indicate that the use of telemetry in non-human primate models is useful for sleep studies, and that the different NREM deep sleep activities between cynomolgus monkeys and common marmoset monkeys are useful to examine sleep functions.

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“…For instance, inbred mouse strains are generally nocturnal but can exhibit phenotypic differences in total amounts of sleep, distributions of sleep throughout the day, and consolidation of sleep periods. These can be correlated with differences in whole brain monoamine contents, which are under elaborate genetic control (Hiyoshi et al 2014). However, this approach has not been widely applied to ontogenetic changes in sleep.…”

Section: Negative Impacts Of Disrupted Sleep During Mammalian Developmentioning

confidence: 99%

Sleep and Development in Genetically Tractable Model Organisms

Kayser

Biron

2016

Genetics

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Sleep is widely recognized as essential, but without a clear singular function. Inadequate sleep impairs cognition, metabolism, immune function, and many other processes. Work in genetic model systems has greatly expanded our understanding of basic sleep neurobiology as well as introduced new concepts for why we sleep. Among these is an idea with its roots in human work nearly 50 years old: sleep in early life is crucial for normal brain maturation. Nearly all known species that sleep do so more while immature, and this increased sleep coincides with a period of exuberant synaptogenesis and massive neural circuit remodeling. Adequate sleep also appears critical for normal neurodevelopmental progression. This article describes recent findings regarding molecular and circuit mechanisms of sleep, with a focus on development and the insights garnered from models amenable to detailed genetic analyses.

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Characteristics of Sleep and Wakefulness inWild-Derived Inbred Mice

Cited by 8 publications

References 25 publications

A point mutation in the ion conduction pore of AMPA receptor GRIA3 causes dramatically perturbed sleep patterns as well as intellectual disability

A point mutation in the ion conduction pore of AMPA receptor GRIA3 causes dramatically perturbed sleep patterns as well as intellectual disability

Investigation of sleep–wake rhythm in non-human primates without restraint during data collection

Sleep and Development in Genetically Tractable Model Organisms

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