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The developmental trajectory of sleep in children with Smith-Magenis syndrome compared to typically developing peers: A three-year follow-up study

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posted on 2023-10-12, 08:56 authored by Georgie Agar, Chris Oliver, Jayne Spiller, Caroline Richards

Study Objectives

To determine the trajectory of: (i) objective sleep parameters and (ii) caregiver-reported sleep questionnaire scores over 3 years in children with Smith-Magenis syndrome (SMS) compared to age-matched typically developing (TD) controls. We also aimed to (iii) describe individual profiles of change in sleep parameters over time.


Methods

Week-long, overnight actigraphy and questionnaire data from 13 children with SMS and 13 age-matched TD children were collected at Time 1 and Time 2 (3 years later). Independent samples t-tests, paired samples t-tests, and Bayesian analyses were used to compare sleep parameters and sleep questionnaire scores between groups at each time point and compare data within groups to assess change over time.


Results

Sleep parameters were consistently more disrupted in the SMS group than the TD group, with significantly reduced sleep efficiency, increased wake after sleep onset and earlier get up times at both time points. This was mirrored in the questionnaire data, with children with SMS evidencing higher scores for overall sleep disturbance, night waking, and daytime sleepiness. While TD sleep parameters demonstrated expected developmental changes over 3 years, in the SMS group sleep parameters and variability between and within children remained largely stable. However, some children with SMS showed substantial variation in sleep parameters over time. Questionnaire scores remained stable over 3 years in both groups.


Conclusions

Overall, sleep disturbance appears to be a stable feature of SMS, indicative of a divergent sleep trajectory compared to TD peers. Proactive intervention approaches should be considered for poor sleep in SMS.

Funding

This study was funded by the charity Cerebra, via a PhD fellowship awarded to the first author.

History

Author affiliation

School of Psychology and Vision Science, University of Leicester

Version

  • VoR (Version of Record)

Published in

Sleep Advances

Volume

4

Issue

1

Publisher

Oxford University Press (OUP)

eissn

2632-5012

Copyright date

2023

Available date

2023-10-12

Language

en

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