Sleep in children with Smith-Magenis syndrome: a case-control actigraphy study

TY - JOUR

T1 - Sleep in children with Smith-Magenis syndrome

T2 - a case-control actigraphy study

AU - Trickett, Jayne

AU - Oliver, Chris

AU - Heald, Mary

AU - Denyer, Hayley

AU - Surtees, Andrew

AU - Clarkson, Emma

AU - Gringras, Paul

AU - Richards, Caroline

PY - 2020/4/15

Y1 - 2020/4/15

N2 - Study Objectives: 1) To compare both actigraphy and questionnaire assessed sleep quality and timing in children with Smith-Magenis syndrome (SMS) to a chronologically age-matched typically developing (TD) group. 2) To explore associations between age, nocturnal and diurnal sleep quality and daytime behaviour.Methods: Seven nights of actigraphy data were collected from 20 children with SMS (mean age 8.70; SD 2.70) and 20 TD children. Daily parent/teacher ratings of behaviour and sleepiness were obtained. Mixed linear modelling was used to explore associations between total sleep time and daytime naps and behaviour.Results: Sleep in children with SMS was characterised by shorter total sleep time (TST), extended night waking, shorter sleep onset, more daytime naps and earlier morning waking compared to the TD group. Considerable inter-daily and inter-individual variability in sleep quality was found in the SMS group, so caution in generalising results is required. An expected inverse association between age and TST was found in the TD group, but no significant association was found for the SMS group. No between group differences in sleep hygiene practices were identified. A bidirectional negative association between TST and nap duration was found for the SMS group. In the SMS group increased afternoon sleepiness was associated with increased irritability (p=.007) and overactivity (p=.005).Conclusion: These findings evidence poor sleep quality in SMS and the need to implement evidence-based interventions in this population.

AB - Study Objectives: 1) To compare both actigraphy and questionnaire assessed sleep quality and timing in children with Smith-Magenis syndrome (SMS) to a chronologically age-matched typically developing (TD) group. 2) To explore associations between age, nocturnal and diurnal sleep quality and daytime behaviour.Methods: Seven nights of actigraphy data were collected from 20 children with SMS (mean age 8.70; SD 2.70) and 20 TD children. Daily parent/teacher ratings of behaviour and sleepiness were obtained. Mixed linear modelling was used to explore associations between total sleep time and daytime naps and behaviour.Results: Sleep in children with SMS was characterised by shorter total sleep time (TST), extended night waking, shorter sleep onset, more daytime naps and earlier morning waking compared to the TD group. Considerable inter-daily and inter-individual variability in sleep quality was found in the SMS group, so caution in generalising results is required. An expected inverse association between age and TST was found in the TD group, but no significant association was found for the SMS group. No between group differences in sleep hygiene practices were identified. A bidirectional negative association between TST and nap duration was found for the SMS group. In the SMS group increased afternoon sleepiness was associated with increased irritability (p=.007) and overactivity (p=.005).Conclusion: These findings evidence poor sleep quality in SMS and the need to implement evidence-based interventions in this population.

KW - actigraphy

KW - behavior

KW - intellectual disability

KW - sleep

KW - Smith–Magenis syndrome

UR - http://www.scopus.com/inward/record.url?scp=85076245020&partnerID=8YFLogxK

U2 - 10.1093/sleep/zsz260

DO - 10.1093/sleep/zsz260

M3 - Article

C2 - 31630201

AN - SCOPUS:85076245020

VL - 43

JO - Sleep

JF - Sleep

SN - 0161-8105

IS - 4

M1 - zsz260

ER -