posted on 2025-07-17, 05:56authored byNatasha Juanita Sneddon
<p dir="ltr">Background: Sleep disturbances are common, and impair quality of life, among Huntington disease genetic expansion carriers (HDGECs). </p><p dir="ltr">Methods: This thesis used various methods to summarise prevalence of sleep and circadian disturbances amongst HDGECs. We conducted a systematic review of the literature reporting the prevalence of sleep and circadian disturbances in HDGECs. Where four or more studies used the same measure of sleep or circadian outcomes, we metaanalysed data using a Random Effects Model (Metafor Package (version 4.6-0) in R (version 4.4.0)). The thesis concludes with recommendations for consideration within future studies of sleep and circadian disturbances within HDGECs. </p><p dir="ltr">Results: The 25 papers included in our systematic review revealed sleep and circadian disturbances are prevalent among HDGECs. There was a large variation in prevalence due to heterogeneity of measures used and HD disease staging, as well as a limited number of participants within studies. Our meta-analysis found prevalence (mean %) for abnormal sleep quality (61%), periodic limb movements (35%), sleep medication (29%), excessive daytime sleepiness (15%), abnormal apnoea-hypopnoea index (9.97%), rapid eye movement sleep without atonia (5%), and rapid eye movement sleep behaviour disorder (3%). </p><p dir="ltr">Conclusion: Sleep disturbances are prevalent among HDGECs. Prevalence varied across populations studied and measures used. Future studies should consider measures that will be appropriate for testing pertinent outcomes among HDGECs, use of the Huntington disease integrated staging system [2], focusing on pre-manifest participants, considering covariates, and conducting randomised controlled trials.</p>
History
Table of Contents
1. Introduction -- 2. What is the prevalence of sleep disturbances in Huntington disease expansion carriers? A systematic review and meta-analysis -- 3. Recommendations for Future Research -- 4. Conclusion -- A. Systematic Literature Review: Database-specific search terms -- B. Systematic Literature Review: PRISMA 2020 Main Checklist -- References
Notes
Thesis by publication
Awarding Institution
Macquarie University
Degree Type
Thesis MRes
Degree
Master of Research
Department, Centre or School
Macquarie Medical School
Year of Award
2025
Principal Supervisor
Clement Loy
Additional Supervisor 1
Angela D'Rozario
Rights
Copyright: The Author
Copyright disclaimer: https://www.mq.edu.au/copyright-disclaimer