title: Daily rhythmic behaviors and thermoregulatory patterns are disrupted in adult female MeCP2-deficient mice.
abstract: Mutations in the X-linked gene encoding Methyl-CpG-binding protein 2 (MECP2) have been associated with neurodevelopmental and neuropsychiatric disorders including Rett Syndrome, X-linked mental retardation syndrome, severe neonatal encephalopathy, and Angelman syndrome. Although alterations in the performance of MeCP2-deficient mice in specific behavioral tasks have been documented, it remains unclear whether or not MeCP2 dysfunction affects patterns of periodic behavioral and electroencephalographic (EEG) activity. The aim of the current study was therefore to determine whether a deficiency in MeCP2 is sufficient to alter the normal daily rhythmic patterns of core body temperature, gross motor activity and cortical delta power. To address this, we monitored individual wild-type and MeCP2-deficient mice in their home cage environment via telemetric recording over 24 hour cycles. Our results show that the normal daily rhythmic behavioral patterning of cortical delta wave activity, core body temperature and mobility are disrupted in one-year old female MeCP2-deficient mice. Moreover, female MeCP2-deficient mice display diminished overall motor activity, lower average core body temperature, and significantly greater body temperature fluctuation than wild-type mice in their home-cage environment. Finally, we show that the epileptiform discharge activity in female MeCP2-deficient mice is more predominant during times of behavioral activity compared to inactivity. Collectively, these results indicate that MeCP2 deficiency is sufficient to disrupt the normal patterning of daily biological rhythmic activities.
authors.affiliation: Division of Genetics and Development, Toronto Western Research Institute, University Health Network, Toronto, Ontario, Canada.
authors.forename: Robert G
authors.initials: RG
authors.lastname: Wither
authors.forename: Sinisa
authors.initials: S
authors.lastname: Colic
authors.forename: Chiping
authors.initials: C
authors.lastname: Wu
authors.forename: Berj L
authors.initials: BL
authors.lastname: Bardakjian
authors.forename: Liang
authors.initials: L
authors.lastname: Zhang
authors.forename: James H
authors.initials: JH
authors.lastname: Eubanks
mesh_terms: D000818:Animals; D001522:Behavior, Animal; D001833:Body Temperature Regulation / Q000235:genetics*; D002940:Circadian Rhythm / Q000235:genetics*; D004569:Electroencephalography; D005260:Female; D051783:Methyl-CpG-Binding Protein 2 / Q000172:deficiency* / Q000235:genetics; D051379:Mice; D018345:Mice, Knockout; D009043:Motor Activity; D013686:Telemetry
chemical_list: D051783:Methyl-CpG-Binding Protein 2