Sleep problems in Rett syndrome animal models: A systematic review

Due to the discovery of Rett Syndrome (RTT) genetic mutations, animal models have been developed. Sleep research in RTT animal models may unravel novel neural mechanisms for this severe neurodevelopmental heritable rare disease. In this systematic literature review we summarize the findings on sleep...

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Published inJournal of neuroscience research Vol. 99; no. 2; pp. 529 - 544
Main Authors Zhang, Xinyan, Lin, Jian‐Sheng, Spruyt, Karen
Format Journal Article
LanguageEnglish
Published United States Wiley Subscription Services, Inc 01.02.2021
Wiley
Subjects
Animal models
Apnea
circadian rhythm
Circadian rhythms
Cognitive science
Drosophila
EEG
Humanities and Social Sciences
Impact analysis
Life Sciences
Literature reviews
monkey
Monkeys
mouse
Mutation
Neurodevelopmental disorders
Rare diseases
REM sleep
Rett syndrome
Rodents
Sleep
Sleep and wakefulness
Sleep disorders
sleep
mouse
monkey
Drosophila
Rett syndrome
circadian rhythm
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Summary:Due to the discovery of Rett Syndrome (RTT) genetic mutations, animal models have been developed. Sleep research in RTT animal models may unravel novel neural mechanisms for this severe neurodevelopmental heritable rare disease. In this systematic literature review we summarize the findings on sleep research of 13 studies in animal models of RTT. We found disturbed efficacy and continuity of sleep in all genetically mutated models of mice, cynomolgus monkeys, and Drosophila. Models presented highly fragmented sleep with distinct differences in 24‐hr sleep/wake cyclicity and circadian arrhythmicity. Overall, animal models mimic sleep complaints reported in individuals with RTT. However, contrary to human studies, in mutant mice, attenuated sleep delta waves, and sleep apneas in non‐rapid eye movement sleep were reported. Future studies may focus on sleep structure and EEG alterations, potential central mechanisms involved in sleep fragmentation and the occurrence of sleep apnea across different sleep stages. Given that locomotor dysfunction is characteristic of individuals with RTT, studies may consider to integrate its potential impact on the behavioral analysis of sleep.
Bibliography:Edited by Christopher Colwell. Reviewed by Shu‐qun Shi and Angelisa Frasca.
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ISSN:0360-4012
1097-4547
DOI:10.1002/jnr.24730