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F INAL R EPORT
WORD Reference: SCS/09/012 Date of Report: 15 th January 2013
Lead applicant: Professor Susan Leekam
Organisation: School of Psychology, Cardiff University
Title: Final title
Repetitive behaviours, anxiety and sensory problems in children with autism and correlates of anxiety in their parents (PhD Mirko Uljarevic)
Original Title: Developing effective early intervention for children with Autism
Spectrum Disorders (ASD)
Total cost:
£60,000.00
Start date: 1 st January 2010 End date: 15 th January 2013
This project aimed to:
1.
provide new data on factors that modulate repetitive behaviours in children with ASD;
2.
provide new data on risk factors for anxiety in parents of children with ASD;
3.
increase understanding about the effectiveness of interventions for ASD.
Aim One - Understanding repetitive behaviours in children with ASD
It has been hypothesised that anxiety and sensory problems are the triggers for repetitive behaviours, however, this hypothesis has not been formally tested before. This PhD project provided an original contribution to the literature on repetitive behaviours, sensory problems and anxiety in ASD by:
 validating the Repetitive Behaviours Questionnaire-2 (RBQ-2; Leekam et al., 2007), a questionnaire that provides detailed assessment of repetitive behaviours in a population of children and adolescents with ASD. Instruments previously used in the research on repetitive behaviours are limited in the sense that they either do not

SCHEDULE 4 provide detailed enough assessment of repetitive behaviours or that they are only appropriate for either very young children or ASD individuals with ASD. Therefore, validation of a new, detailed instrument is a significant contribution to the literature and future research.
 reporting that 49% of children with ASD met the criteria for elevated overall anxiety and that all children with ASD exhibited some form of sensory problems, with majority of children having problems in more than one sensory modality at the same time and majority of children showing paradoxical response to sensory stimuli from their environment i. e. being both hyper-responsive and hypo-responsive to the stimulation;
 by using complex mediation models, this project showed that anxiety was related to particular types of repetitive behaviour – insistence on sameness and that this relationship was mediated by two types of sensory problems, namely sensory sensitivity and sensory avoiding.
Aim Two - What are the risk factors for anxiety in parents of children with ASD?
Previous research has shown that anxiety and depression are very frequent in parents of children with ASD; however, research to date has mainly considered their problems to be a consequence of the burden caused by raising a child with autism. Although the everyday lives of parents of children with ASD are undoubtedly stressful, and from the general anxiety literature it is clear that prolonged elevated levels of stress is a risk factor for anxiety, it is important to highlight that several theories suggest that the link between life events and psychiatric symptoms is frequently moderated by individual differences. This PhD project provided an original contribution to the literature on anxiety in parents of children with ASD by developing theoretical model that suggested that certain parents’ own personal characteristics might on the one hand put them at increased risk for developing affective problems, and on the other, protect them from affective problems despite the problems they face in their everyday lives. In particular Intolerance of Uncertainty and Sensory Problems in parents were hypothesised to be major risk factors for the anxiety in parents of children with
ASD. This research project showed that:

44% of parents had clinically significant levels of anxiety;

58% of parents have elevated sensory sensitivity scores (interestingly, sensory problems in children and parents were highly associated);#

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 after controlling for characteristics of children, the factor that distinguished group of anxious parents from non-anxious parents was higher levels of intolerance of uncertainty, increased sensory sensitivity and higher use of avoidant coping.
These findings provide a solid background for developing a research program on parents, with the ultimate goal of helping parents and families adapt to the challenges of caring for individuals with ASD.
Aim Three - Increase understanding about the effectiveness of interventions for ASD
It was originally planned to compare two comprehensive intervention programmes that are widely used in Wales but without strong evidence base: Intensive interaction and
Portage. In the original plan, the baseline data were supposed to be collected immediately prior to the onset of the intervention, recording the age at which the child began treatment, the intensity of the treatment (in hours per week) and then it was planned to use timed post-test measures 16 weeks later. There were supposed to be 16 children in Intensive Interaction group and 16 in Portage group and, where possible, it was planned that interventions would be delivered for exactly the same number of hours per week. However, as noted in the
Quarterly reports for June and September 2011, due to reduced referral through services, this plan had to be changed and new, revised proposal was to conduct a case study of two children diagnosed with ASD who would receive Intensive Interaction across the period of 12 weeks.
Intensive interaction is widely used in Wales by speech and language therapists. It is an approach aimed at facilitating the development of the most fundamental social and communication abilities in individuals with severe and complex learning difficulties.
Emphasis is on overall style of interaction rather than pre-specified objectives and there are some evidence suggesting that intensive interaction can improve self-motivation, lead to improved responsiveness, communication, greater acceptance of physical contact, greater tolerance and enjoyment of interaction as well as the development of relationships. It is hypothesized that mechanism behind those beneficial effect is the reduction of over-arousal and anxiety. However, at present, these empirical evidence come from few single case studies, narrative case studies and anecdotal reports of teachers who use intensive interaction with individuals with learning difficulties (Nindt, 1999; Nindt & Powell, 2000). It was felt that, although these anecdotal data are promising, it was necessary to gather scientific data on the efficacy of this intervention and also about the possible mechanism that might be behind possible positive effects for the child with ASD.
Therefore, new revised proposal had two aims: to examine effects of therapy on the child’s level of functioning in terms of repetitive behaviours and social and communicative

SCHEDULE 4 abilities and also to investigate the process of therapy itself. This case study was important in its own right but it also built a good baseline for the larger study in the future, with the possibility of including other types of intervention as well as a comparison and with the goal of isolating active components behind the different interventions.
Outcomes of this part of the project:
 study used multiple-baseline single case design the social attention of two 3year-olds with autism who received an imitation-based intervention from a speech pathologist was studied. Each child received 10 or 12 one-hour intervention sessions preceded by a free-play session with a researcher.
Outcome measures were social attention with the therapist and with the researcher. Social attention did not increase over the course of the intervention phase for either child, nor during free play with the researcher. Thus, there was no evidence of cumulative effects of imitation, or generalisation to free play.
More analyses on the collected data for this part of the project are planned for the future.
Grant outputs:
During this PhD project, one review paper on repetitive behaviours in ASD has been published in highly regarded peer-reviewed academic journal (Psychological Bulletin, impact factor: 15.575). This paper has been cited 37 times so far by other researchers (google scholar statistic). Furthermore, 2 research papers have been submitted and currently under review and two more publications are in preparation (expected submission date September 2013).
Results from this project were presented at 4 international and one national UK conferences. PhD student has also given three academic talks on this research.
Finally, PhD student Mirko Uljarević is expected to submit his PhD thesis by
September 6 th
.
List of grant outputs:
Academic Papers:
Leekam, S. R., Prior, M. R., Uljarević, M. (2011). Restricted and repetitive behaviors in autism spectrum disorders: A review of research in the last decade. Psychological
Bulletin, 137 (4), 562-593.
Lidstone, J., Uljarevic, M., Sullivan, J., Rodgers, J., McConachie, H., Freeston, M., Le
Couteur, A., Prior, M., Leekam S. (2013). Relations among restricted and repetitive behaviours, anxiety and sensory symptoms in individuals with autism spectrum disorders. Research in Autism Spectrum Disorders. (Submitted).

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Lidstone, J., Uljarević, M., Leekam, S. R., Kanaris, H., Mullis, J., McKigney, A-M., &
Paradice, R., (2013). Early intervention for children with autism: Adult imitation of children’s actions. Submitted.
Uljarević, M., Leekam S. (2013). Intolerance of Uncertainty and Coping Strategies As
Correlates of Anxiety in Mothers of Children with ASD. (In preparation)
Uljarević, M., Leekam S. (2013). Sensory Processing in children with ASD and their parents.
(In preparation).
Conference proceedings
Uljarević, M., Lidstone, J., Leekam, S., Freeston, M., Le Couteur, A., McConachie, H.,
Rodgers, J., & Prior, M. Correlates of anxiety in mothers of children with autism spectrum disorder. The European Academy of Childhood Disability annual meeting,
October, 2013.
Uljarević, M., Lidstone, J., Leekam, S., Freeston, M., Le Couteur, A., McConachie, H.,
Rodgers, J., & Prior, M. Intolerance of Uncertainty and Coping Strategies As Correlates of Anxiety in Mothers of Children with ASD. Poster presented at the International
Meeting for Autism Research (IMFAR), May 2013, San Sebastian, Spain.
Uljarević, M., Lidstone, J., Leekam, S., Freeston, M., Le Couteur, A., McConachie, H.,
Rodgers, J., & Prior, M. Sensory processing and repetitive motor behaviours in children with Autism Spectrum Disorders (ASD). Poster presented at the British Psychological
Society Developmental Section Annual Conference, 5 th
-7 th
September, 2012 University of Strathclyde, Glasgow.
Uljarević, M., Lidstone, J., Leekam, S. R., Kanaris, H., McKigney, A. M., Mullis, J.,
Paradice, R. Anxiety in Children with Autism Spectrum Disorders is Associated with
Affective Symptoms in Their Mothers. Poster presented at the International Meeting for
Autism Research (IMFAR), May 2012, Toronto, Canada.
Lidstone, J., Uljarević, M., Leekam, S.R., Kanaris, H., McKigney, A.-M., Mullis, J., &
Paradice, R. (2012). How do the functions of restricted and repetitive behaviours vary with developmental level in children with autism spectrum disorders? Poster presented at the International Meeting for Autism Research, Canada, May 2012, Toronto, Canada.
Academic talks:
Uljarević, M., Lidstone, J., Leekam, S. R. Sensory symptoms and anxiety: A study of children with ASD and their parents. Talk given to the Bristol Autism Research Group (BARG),
18 th September 2012, School of Experimental Psychology, Bristol.

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Uljarević, M.
Sensory problems, anxiety and repetitive behaviours in children with ASD .
School of Psychology, Cardiff University, seminar, 29th June 2012.
Uljarević, M. The relationship between Anxiety, Sensory Processing and Repetitive
Behaviours in children and adolescents with ASD. Postgraduate Student Conference,
Gregynog, December 2011.
Raising awareness of research and dissemination of results to public:
Results from this projects have been widely dissemination including presentations to parents, teachers, and policy makers, including the visit by the Deputy Minister for Social
Services Gwenda Thomas. Please see below for a full list of dissemination activities:
Uljarević, M., Lidstone, J., Leekam, S. R. Anxiety and stress in parents of children with autism. Talk given at the dissemination event for parents of children with ASD,
September 17 th
, 2012, School of Biosciences, Cardiff University.
Uljarević, M. Iimplementation of the Wales Autism Intervention Survey and research findings on anxiety and coping in parents of children with Autism Spectrum Disorders. Invited talk to the Autism Support Team, Mynachdy Centre, Cardiff.
Uljarević, M. Anxiety and Stress in Parents of children with ASD. Invited talk to Cardiff and the Vale NAS Parent Group, Cardiff, 26 th
May 2011.
Uljarevic, M. Repetitive behaviours and anxiety in children with autism. Hollies School,
November, 2010
Uljarević, M., Lidstone, J., & Leekam, S. R. Anxiety and sensory problems in children with
ASD. Invited talk to teachers from the Ashgrove Specialist School, Cardiff, November
2010.
Uljarević, M. Identifying the risk factors linked to anxiety in children with ASD. Open
Afternoon for the Deputy Minister for Social Services Gwenda Thomas, WARC funders and collaborators. September, 2010.