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Title: Circadian rhythms, sleep and behaviour in intellectual and developmental disabilities : a systematic review of sleep and challenging behaviour and actigraphic assessment of circadian functioning in MPS III (Sanfilippo syndrome)
Author: Mumford, Rachel Anne
Awarding Body: University of Manchester
Current Institution: University of Manchester
Date of Award: 2013
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Sleep disturbance and behavioural difficulties are both prevalent problems in the intellectual and developmental disability population and can have a significant impact on quality of life for the individual and their family. This thesis investigated sleep, behaviour and circadian rhythm functioning in children with intellectual and developmental disabilities, and is presented in three sections. The first two papers have been prepared in accordance with the author guidelines of the journals proposed for submission, excluding tables and figures for ease of reading. The first paper is a systematic review of the literature examining the relationship between sleep disturbance and challenging behaviour in children with intellectual and developmental disabilities. 15 studies were included in the review and overall there were consistent findings of an association between the presence of sleep disruption and increased behavioural difficulties. A causal relationship could not be inferred due to the cross-sectional methodology of studies. Other factors, such as parental wellbeing, child level of intellectual disability and comorbidity of physical health conditions, need to be considered to understand the complexity of this relationship. Children with the neurodevelopmental disorder mucopolysaccharidosis type III (MPS III or Sanfilippo syndrome) present with high rates of sleep disturbance and challenging behaviour. The second paper investigates circadian rhythm functioning and activity levels in children with MPS III, compared to typically developing controls. Objective measurement of circadian rhythm and activity levels was obtained through actigraphic recording for 7-10 days. Children with MPS III had increased fragmentation of circadian rhythm, less stability of rhythm in relation to external cues and a differential pattern of activity across the day compared to controls. Overall, results were indicative of a disruption of circadian rhythm function in children with MPS III. The implications for clinical practice and future research are discussed. The third paper provides a critical appraisal of the overall research process, including further consideration of the strengths and limitations, implications for clinical practice, wider context of the research and personal reflections. An account of the project that was originally proposed with the MPS III population is also presented, alongside reflections on its termination.
Supervisor: Hare, Dougal; Bunton, Penny Sponsor: Not available
Qualification Name: Thesis (D.Clin.Psy.) Qualification Level: Doctoral
EThOS ID:  DOI: Not available
Keywords: sleep, circadian rhythm, behaviour, mucopolysaccharidosis type III, Sanfilippo