Hyperactivity and male‐specific sleep deficits in the 16p11.2 deletion mouse model of autism
Open accessAbstract
Sleep disturbances and hyperactivity are prevalent in several neurodevelopmental disorders, including autism spectrum disorders (ASDs) and attention deficit‐hyperactivity disorder (ADHD). Evidence from genome‐wide association studies indicates that chromosomal copy number variations (CNVs) are associated with increased prevalence of these neurodevelopmental disorders. In particular, CNVs in chromosomal region 16p11.2 profoundly increase the risk for ASD and ADHD, disorders that are more common in males than females. We hypothesized that mice hemizygous for the 16p11.2 deletion (16p11.2 del/+) would exhibit sex‐specific sleep and activity alterations. To test this hypothesis, we recorded activity patterns using infrared beam breaks in the home‐cage of adult male and female 16p11.2 del/+ and wildtype (WT) littermates. In comparison to controls, we found that both male and female 16p11.2 del/+ mice exhibited robust home‐cage hyperactivity. In additional experiments, sleep was assessed by polysomnography over a 24‐hr period. 16p11.2 del/+ male, but not female mice, exhibited significantly more time awake and significantly less time in non‐rapid‐eye‐movement (NREM) sleep during the 24‐hr period than wildtype littermates. Analysis of bouts of sleep and wakefulness revealed that 16p11.2 del/+ males, but not females, spent a significantly greater proportion of wake time in long bouts of consolidated wakefulness (greater than 42 min in duration) compared to controls. These changes in hyperactivity, wake time, and wake time distribution in the males resemble sleep disturbances observed in human ASD and ADHD patients, suggesting that the 16p11.2 del/+ mouse model may be a useful genetic model for studying sleep and activity problems in human neurodevelopmental disorders. Autism Res 2016. © 2016 International Society for Autism Research, Wiley Periodicals, Inc. Autism Res 2017, 10: 572–584. © 2016 International Society for Autism Research, Wiley Periodicals, Inc.
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Hyperlocomotion is broadly reported in 16p11.2+/− mice, another behavioral feature that does not coincide directly with human symptomology.
“Hyperlocomotion is also broadly reported in 16p11.2+/− mice [4, 5, 59, 61, 63, 64, 70], another behavioral feature that does not coincide directly with human symptomology.”
From 16p11.2 Copy Number Variations and Neurodevelopmental Disorders (2020) · cites this paper as [70]
Sleep abnormalities and anxiety have been reported in 16p11.2+/− mice (Mills).
“Sleep abnormalities [69, 70] and anxiety [66] have been reported in 16p11.2+/− mice (Mills).”
From 16p11.2 Copy Number Variations and Neurodevelopmental Disorders (2020) · cites this paper as [70]
Male 16p11.2+/− mice (Mills) exhibit sleep deficits and impairments in a reward-directed learning task, while females do not.
“Additionally, male 16p11.2+/− mice (Mills) exhibit sleep deficits [70], and impairments in a reward-directed learning task [67], while females do not.”
From 16p11.2 Copy Number Variations and Neurodevelopmental Disorders (2020) · cites this paper as [70]
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References 71 · Cited by 83
- Authors
- Angelakos, C. C., Watson, A. J., O'Brien, W. T., Krainock, K. S., Nickl‐Jockschat, T., & Abel, T.
- Affiliations
- University of Pennsylvania (US)
- Department of Neuroscience University of Pennsylvania Philadelphia PA 19104
- Department of Biology University of Pennsylvania Philadelphia PA 19104
- Jülich Aachen Research Alliance (DE)
- Corresponding author
- Abel, Ted
- Venue
- Autism Research
- Publisher
- Wiley
- Published
- 2016-10-14
- Volume
- 10
- Issue
- 4
- Pages
- 572-584
- ISSN
- 1939-3792 (ISSN-L)
- 1939-3806
- PMC
- PMC6201314
- OpenAlex
- W2550071093
- Cited by
- 83
- References
- 71
- Citations by year
- 2026: 8 · 2025: 9 · 2024: 9 · 2023: 12 · 2022: 7 · 2021: 6 · 2020: 8 · 2019: 10 · 2018: 9 · 2017: 5
- FWCI
- 5.08
- Type
- journal article
- Language
- en
- Open access
- green
- europepmc.org
- Locations
- Autism Research (publishedVersion)
- PubMed (publishedVersion)
- Europe PMC (PubMed Central) (acceptedVersion)
- PubMed Central (acceptedVersion)
- RWTH Publications (RWTH Aachen) (submittedVersion)
- Funders
- Simons Foundation
- Deutsche Forschungsgemeinschaft
- National Defense Science and Engineering Graduate
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (U54HD086984)
- Air Force Office of Scientific Research
- Topics
- Life Sciences › Neuroscience › Cognitive Neuroscience › Sleep and Wakefulness Research
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genomic variations and chromosomal abnormalities
- Life Sciences › Neuroscience › Cognitive Neuroscience › Autism Spectrum Disorder Research
- Keywords
- 16p11.2 deletion, autism spectrum disorder, attention deficit hyperactivity disorder, sleep disorders, hyperactivity, sex differences, home cage activity, wakefulness, NREM sleep, chromosomal copy number variation, mouse model, neurodevelopmental disorders
- MeSH terms
- Autism Spectrum Disorder, Adult, Animals, Attention Deficit Disorder with Hyperactivity, Chromosome Deletion, Circadian Rhythm, Disease Models, Animal, Female, Humans, Hyperkinesis, Male, Mice, Inbred C57BL, Models, Genetic, Sex Factors, Sleep Stages, Polysomnography, Genetic Predisposition to Disease, Mice, Genome-Wide Association Study, DNA Copy Number Variations, Mice, 129 Strain
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