Osman S Ipsiroglu1, Katarina Wind2, Yi-Hsuan Amy Hung2, Mai Berger3, Forson Chan3, Wayne Yu4, Sylvia Stockler5, Joanne Weinberg4. 1. Sleep/Wake-Behavior Research Lab, BC Children's Hospital Research Institute, Vancouver, BC, Canada; Departments of Pediatrics/Psychiatry, Sleep Medicine Center, BC Children's Hospital, University of British Columbia, Vancouver, BC, Canada. Electronic address: oipsiroglu@bcchr.ca. 2. Sleep/Wake-Behavior Research Lab, BC Children's Hospital Research Institute, Vancouver, BC, Canada; Department of Cellular & Physiological Sciences, Faculty of Medicine, University of British Columbia, Vancouver, BC, Canada. 3. Sleep/Wake-Behavior Research Lab, BC Children's Hospital Research Institute, Vancouver, BC, Canada. 4. Department of Cellular & Physiological Sciences, Faculty of Medicine, University of British Columbia, Vancouver, BC, Canada. 5. Department of Pediatrics, Division of Biochemical Diseases, BC Children's Hospital, University of British Columbia, Vancouver, BC, Canada.
Abstract
INTRODUCTION: Clinical research and studies using animal models have revealed a complex and relatively under-explored interaction between prenatal alcohol exposure (PAE) and alterations in sleep-wake behaviors. OBJECTIVES: To utilize a structured naturalistic observation-based methodology, consisting of descriptive elements, to provide insight into possible links between altered sleep and disruptive daytime presentations in children and adolescents with fetal alcohol spectrum disorder (FASD). To apply a similar structured behavioral observation protocol in a PAE animal model to compare outcomes from the experimental and clinical studies utilizing naturalistic observational methodology. METHODS: Forty pediatric patients with FASD (1.8-17.5 yrs, median age 9.4 yrs) and chronic sleep problems were assessed. In the PAE animal model, male offspring from PAE, Pair-Fed (PF), and ad libitum-fed Control (C) groups (n = 8/group) were assessed in the juvenile/preadolescent (23-25 days of age) and adolescent/pubertal (35-36 days of age) periods. RESULTS: In the clinical setting, we found that 95% of children with FASD showed disruptive or externalizing behaviors, 73% showed internalizing behaviors, 93% had circadian rhythm sleep disorders, all had chronic insomnia, and 85% had restless sleep, often with tossing/turning/kicking movements indicative of non-restorative sleep with hypermotor events. In the daytime, individuals showed excessive daytime sleepiness as well as hyperactive/hyperkinetic behaviors, an urge-to-move, and involuntary movements suggestive of hyperarousability. Alterations in sleep/wake behaviors in the PAE animal model paralleled the clinical data in many aspects, demonstrating greater sleep latencies, less total time asleep, more total time awake and longer awake bouts, more position changes, more time in transition, and longer transition bouts in PAE compared to PF and/or control animals. CONCLUSIONS: Thus, our findings provide support for the power and validity of naturalistic observational paradigms in revealing dysregulated sleep-wake behaviors and their association and/or exacerbating relationship with day and nighttime behavioral problems, such as disruptive behaviors, externalizing and internalizing disorders, and daytime sleepiness.
INTRODUCTION: Clinical research and studies using animal models have revealed a complex and relatively under-explored interaction between prenatal alcohol exposure (PAE) and alterations in sleep-wake behaviors. OBJECTIVES: To utilize a structured naturalistic observation-based methodology, consisting of descriptive elements, to provide insight into possible links between altered sleep and disruptive daytime presentations in children and adolescents with fetal alcohol spectrum disorder (FASD). To apply a similar structured behavioral observation protocol in a PAE animal model to compare outcomes from the experimental and clinical studies utilizing naturalistic observational methodology. METHODS: Forty pediatric patients with FASD (1.8-17.5 yrs, median age 9.4 yrs) and chronic sleep problems were assessed. In the PAE animal model, male offspring from PAE, Pair-Fed (PF), and ad libitum-fed Control (C) groups (n = 8/group) were assessed in the juvenile/preadolescent (23-25 days of age) and adolescent/pubertal (35-36 days of age) periods. RESULTS: In the clinical setting, we found that 95% of children with FASD showed disruptive or externalizing behaviors, 73% showed internalizing behaviors, 93% had circadian rhythm sleep disorders, all had chronic insomnia, and 85% had restless sleep, often with tossing/turning/kicking movements indicative of non-restorative sleep with hypermotor events. In the daytime, individuals showed excessive daytime sleepiness as well as hyperactive/hyperkinetic behaviors, an urge-to-move, and involuntary movements suggestive of hyperarousability. Alterations in sleep/wake behaviors in the PAE animal model paralleled the clinical data in many aspects, demonstrating greater sleep latencies, less total time asleep, more total time awake and longer awake bouts, more position changes, more time in transition, and longer transition bouts in PAE compared to PF and/or control animals. CONCLUSIONS: Thus, our findings provide support for the power and validity of naturalistic observational paradigms in revealing dysregulated sleep-wake behaviors and their association and/or exacerbating relationship with day and nighttime behavioral problems, such as disruptive behaviors, externalizing and internalizing disorders, and daytime sleepiness.
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