Sensory profiles in autism spectrum disorder and Williams syndrome Magdalena Glod Thesis submitted in candidature for the degree of Doctor of Philosophy Newcastle University Institute of Neuroscience May 2017 Abstract This thesis explored sensory profiles in children with autism spectrum disorder (ASD) and Williams syndrome (WS). The thesis begins with two review papers: the psychological correlates of sensory processing patterns in individuals with ASD were evaluated in a systematic review, followed by a mixed-methods review of sensory processing in Williams syndrome. Next, an investigation of changes in sensory symptoms across different age groups in children with ASD and WS was undertaken and revealed that level of sensory atypicalities in both disorders across age groups were very similar. This was followed with an examination of the sensory profiles of children with ASD without learning disability, ASD with learning disability and WS indicating that the distinction between the diagnostic group based on sensory behaviours and socio-communicative characteristics could hardly be made. A factorial validity of the Spence Children’s Anxiety Scale-Parent version was then examined. The conventional SCAS-P structure in the ASD sample was not confirmed, raising concerns regarding the validity of the tool. Further exploration of sensory profiles in ASD and WS was then undertaken, where sensory processing clusters of children with both disorders were examined. The relationships between sensory processing and other clinical features were described and the mediating role of anxiety and intolerance of uncertainty between sensory processing abnormalities and repetitive behaviours was demonstrated. Next, the first comparison of sensory profiles in child-parent dyads in ASD and typical development (TD) was reported indicating some divergent patterns. Finally, a novel direct assessment of auditory and tactile sensory processing was developed and found to show promise as a measure for use with young children with ASD and WS. The synthesis of the evidence across chapters was then discussed and strengths and weaknesses of the current work presented. Suggestions for future research and the implications for clinical and research endeavours were considered. iii Dedication For mum For my family And for friends who became family v Acknowledgments I will never be able to thank my supervisors enough; Jacqui Rodgers, Debbie Riby and Emma Honey for their guidance, support and patience throughout my PhD. I am extremely grateful for the support of the children and families for their time and enthusiasm to take part in my studies. Recruiting participants would not be possible without the help of the Williams Syndrome Foundation, ASD-UK and schools in the North East of England. I would also like to acknowledge Cathy Creswell, Polly Waite, Ruth Jameson, Mikle South and Helen McConachie for their help with data acquisition for analysis undertaken in Chapter 6. Cathy Creswell also described the anxious sample presented in that chapter. The work in this PhD thesis was supported with funding from the Estate of David Murray Garside. vii Table of Contents Abstract ................................................................................................................................... iii Dedication ................................................................................................................................. v Acknowledgments ................................................................................................................. vii List of Tables .......................................................................................................................... xv List of Figures ..................................................................................................................... xviii List of Publications.........................................................................................................…...xix Chapter 1. General introduction to autism spectrum disorder and Williams syndrome.1 1.1 Autism spectrum disorder ................................................................................................ 1 1.2 Williams syndrome .......................................................................................................... 2 1.3 Sensory processing ........................................................................................................... 3 Chapter 2. Psychological correlates of sensory processing patterns in individuals with autism spectrum disorder: a systematic review .................................................................... 6 2.1 Background ...................................................................................................................... 6 2.1.1 Sensory atypicalities in ASD ..................................................................................... 6 2.1.2 Sensory processing patterns in ASD ......................................................................... 6 2.1.3 Symptom co-morbidity .............................................................................................. 6 2.1.4 Previous reviews ....................................................................................................... 7 2.1.5 Aim of the review ....................................................................................................... 7 2.2 Method ............................................................................................................................. 8 2.2.1 Inclusion/ exclusion criteria ...................................................................................... 8 2.2.2 Search Strategy ......................................................................................................... 8 2.2.3 Electronic search .................................................................................................... 10 Screening of electronic search results .................................................................................. 11 2.2.4 Final selection ......................................................................................................... 11 2.2.5 Critical evaluation .................................................................................................. 21 2.3 Results ............................................................................................................................ 21 2.3.1 Evaluation grid – papers’ quality ........................................................................... 21 2.3.2 Participants’ section ............................................................................................... 39 2.3.3 Sensory measures section ........................................................................................ 39 2.3.4 Psychological correlate measure section ................................................................ 40 ix 2.3.5 Results – associations ............................................................................................. 41 2.3.6 Participants ............................................................................................................. 42 2.3.7 Psychological constructs ........................................................................................ 44 Restricted and repetitive behaviours (RRBs) ...................................................................... 47 Emotional and behavioural functioning ............................................................................... 47 Physical skills ...................................................................................................................... 51 2.4 Discussion ...................................................................................................................... 51 2.4.1 Evaluation grid - papers’ quality............................................................................ 51 2.4.2 Sensory processing patterns and correlates ........................................................... 53 2.4.3 Study limitations ..................................................................................................... 55 2.4.4 Conclusions ............................................................................................................. 55 Chapter 3. Sensory processing in Williams syndrome: a narrative review ..................... 57 3.1 Background .................................................................................................................... 57 3.1.1 Previous reviews ..................................................................................................... 57 3.1.2 Aims ........................................................................................................................ 58 3.2 Method ........................................................................................................................... 58 3.2.1 Inclusion/ exclusion criteria ................................................................................... 58 3.2.2 Search Strategy ....................................................................................................... 58 3.2.3 Electronic search .................................................................................................... 61 3.2.4 Screening of electronic search results .................................................................... 61 3.3 Results...........................................................................................................................