Sensory integration-based interventions for children with autism spectrum disorder. Erin E. Barton. University of Colorado Denver

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1 1 Sensory integration-based interventions for children with autism spectrum disorder Erin E. Barton University of Colorado Denver Brian Reichow University of Connecticut Health Center

2 2 Abstract Background: Although not a diagnostic criteria until the fifth revision of the American Psychiatric Association Diagnostic and Statistical Manual, children with autism spectrum disorders (ASD) are often reported to show atypical responses and behaviors to sensory stimuli. Interventions based on sensory integration theory are often used to address their atypical behavior. Although frequently used, the empirical evidence supporting these interventions has been criticized and questioned. The purpose of this review protocol is to systematically analyze the evidence for sensory integration-based interventions with children who have ASDs. Methods/Design: This protocol has been registered and published PROSPERO (CRD ). We will search Medline, PsycINFO, EMBASE, CINHAL, ERIC, ClinicalTrials.gov, and the Cochrane Registry of Controlled Trials (CENTRAL) for all relevant studies. Our inclusion criteria are: (a) the use of experimental designs to compare sensory integration-based interventions to another treatment or control, (b) the inclusion of participants with diagnoses of autism spectrum disorders, and (c) articles written in English. We will extract data from included studies, evaluate each study s risk of bias, and evaluate the effects of the sensory integration-based interventions for each study. Where possible, quantitative syntheses methods will be used to combine study results to report average effects; if heterogeneity is found, we will explore relations using subgroup and sensitivity analyses. If meta-analysis is inappropriate, we will provide narrative reviews of the study findings. Keywords: autism spectrum disorder, ASD, sensory integration, occupational therapy, OT, complementary and alternative medicine, systematic review

3 3 Background Description of the Condition Autism is a neurobiological developmental disorder that manifests with much variability. Today there are several different conditions related to autism commonly known as autism spectrum disorders (Volkmar, State, & Klin, 2009). Although there are many commonalities, there is no single behavioral marker for autism. Characteristics include qualitative impairments in social interactions, communication, and restricted, repetitive, and stereotyped patterns of behavior. About 60% of children with autism experience significant cognitive delays (Fombonne, 2005), and about 30% 50% will not develop functional speech; however, these numbers are decreasing with early diagnosis and treatment (Chakrabarti & Fombonne, 2005). Children with autism also might display atypical responses to sensory experiences; however, this is a characteristic that might be seen in many developmental behavioral disorders (e.g., ADHD, childhood anxiety disorders; American Academy of Pediatrics, 2012). The exact prevalence of sensory abnormalities in children with autism is difficult to estimate given the idiosyncratic and heterogeneous nature of the disability. Some estimates suggest as many as 90% of children with autism have sensory abnormalities in multiple domains and these difference persist across age and IQ (Leekham, Nieto, Libby, Wing, & Gould, 2007). Description of the intervention Sensory integration-based interventions are designed to address sensory processing difficulties in young children. In recent years, sensory integration-based interventions have gained popularity and are the most frequently requested intervention for children with autism, despite having limited empirical support. The use of sensory integration-based interventions is a particularly common practice for children with autism (Green, Pituch, Itchon, Choi, O Reilly, & Sigafoos, 2006; Olson & Moulton, 2004). The driving principle for the use of

4 4 sensory integration-based interventions is to improve sensory processing and increase adaptive functioning in individuals with sensory dysfunction (Ayers, 1972). This principle is based on sensory integration theory, which purports that controlled sensory experiences will help children appropriately respond to sensory input (Ayers, 1979). A variety of sensory integration-based interventions have been developed to create these controlled sensory experiences for young children with autism. These include: weighted vests, massage therapy, brushing, therapy balls, swings, auditory interventions, and so on. Review Purpose Although the use sensory integration-based interventions are widespread, the research on sensory integration-based interventions is limited by a lack of controlled, rigorous studies and clear guidelines and procedures for their use (Reichow et al., 2010; Schaaf, 2011). The purpose of this review is to synthesize this research and determine the evidence for practice. The literature review will examine comprehensive sensory integration treatments, sensory diets, touch-based treatments, weighted / pressure vests, auditory interventions, and therapy balls. The literature review is limited to peer-reviewed, published studies that experimentally examined the effects of sensory integration-based interventions on young children with ASDs (younger than 6 years old). Methods/Design Study Selection Types of studies. All relevant randomized control trials, quasi-randomized control trials (i.e., trials where a quasi-random method of allocation is used, such as alternation or date of birth), controlled clinical trials (CCT), and single-subject experimental design (SSED) studies that allow for the possible assessment of a functional relation will be included. Pre/Post quasi-experimental design studies in which a comparison group is not used, or SSED studies without at least 3 attempts to demonstrate a treatment effect will be excluded.

5 5 We will include cross-over trials but will analyze the results at the conclusion of the first phase. Types of participants. We will include studies with children who have autistic disorder, Asperger s disorder, pervasive developmental disorder, not otherwise specified, or atypical autism who are younger than 18 years old at the onset of treatment. Participants will not be excluded based on IQ or presence of comorbidities. Setting. We will not exclude studies based on the location in which the sensory integration therapy is provided. Types of interventions. Sensory integration therapy as defined above, compared with no treatment or waitlist controls, or treatment as usual (TAU). TAU often combines a variety of treatment components, sometimes referred to as eclectic. Types of outcome measures. We will examine the following types of primary outcomes: sensory processing, attention, challenging behaviors, appropriate behaviors, social skills, language, engagement, stereotypy, self-regulation, accuracy of responses, and motor skills. Search Methods We will search Medline, PsycINFO, EMBASE, CINHAL, and ERIC for all relevant studies. We will not use any filters, such as language or randomization, to prevent missing any relevant studies. A copy of our search strategy can be downloaded as a supplemental file from PROSPERO. We will identify unpublished and ongoing trials by (a) searching trial registries (ClinicalTrials.gov, Cochrane Central Register of Controlled Trials [CENTRAL]), (b) searching reference lists of included studies, and (c) correspondence with authors of the included studies. The search strategy is shown in Appendix A. Data Collection and Analysis

6 6 Selection of studies. Two graduate assistants will independently screen the titles and abstracts yielded by the search against the inclusion criteria. Full texts of all articles that appear relevant will then be completed by the two authors (EB and BR). We will resolve disagreements through mediation and if necessary, a third opinion. The reason for exclusion of all trials in which full texts are reviewed will be recorded and presented in a flow diagram. Data extraction and management. Keith Kravec (KK) and Hannah Embree (HE), graduate students in early childhood special education, will independently extract data from each trial using a data collection form. We will extract the following information: (a) sample size, (b) ages, (c) diagnoses, (d) independent confirmation of diagnoses, (e) Sensoryintegration based intervention type, (f) research design, (g) duration of intervention, (h) intervention materials, (i) intervention setting, (k) intervention fidelity measurement, (l) interventionist, (m) interventionist training, (n) dependent variables, (o) measurement tools and procedures, (p) assessors, (q) reliability of assessment reported, (r) study rigor, and (s) reported outcomes. Again, disagreements will be resolved through mediation and if necessary a third opinion (Erin Barton; EB). Assessment of risk of bias. Because we anticipate locating group research design trials and single subject experimental design trials we will assess methodological rigor (risk of bias) using the evaluative method developed by Reichow and colleagues (Reichow, Volkmar, Chicchetti, 2008; Reichow, 2011). The evaluative method contains two rubrics (one for group design studies and one for single subject experimental design studies) that assess six primary quality indicators and up to 8 secondary quality indicators. The method has been used to evaluate multiple treatment approaches including social skills (Reichow & Volkmar, 2010), psychotropic medication (Siegel & Beaulieu, 2012), and sight word instruction (Spector, 2011). Initial psychometric testing indicated the method produces highly valid and reliable results (Cicchetti, 2011; Reichow et al., 2007).

7 7 Measurement of treatment effect. For group research design studies reporting continuous data, we will analyze study data by calculating the standardized mean difference effect size using Hedges g (Hedges & Olkin, 1985). For single subject experimental design studies, we will calculate a success estimate using the methods proposed by Reichow & Volkmar (2010). If studies report high degrees of missing data, we will contact the study author to inquire about getting the data. If we are unable to locate the data, we will only use the data that are available (i.e., we will not impute missing data). Assessment of heterogeneity. If we conduct a meta-analysis of group research design studies, we will assess heterogeneity using the Chi-square test (Q-statistic) and I 2 (Higgins et al., 2002). The Q-statistics provides information about whether the heterogeneity is greater than that expected by chance, and I2 provides the percentage of between study variability. If we find heterogeneity in the meta-analysis, we will conduct both subgroup analyses (e.g., intervention density, participant characteristics, intervention characteristics) and sensitivity analyses (e.g., removal of studies with high risk of bias, removal of CCTs, conduction of meta-analysis using different statistical techniques), if data permit. Assessment of publication bias. If we identify greater than 10 studies, we will use a funnel plot to examine publication bias (sometimes referred to as reporting bias). The funnel plot will show the estimated differences in treatment effects against their standard error. We have set a threshold of 10 studies because funnel plots with low number of studies have been shown to be error prone and difficult to interpret (Sterne et al., 2008). Data syntheses. If data permit, we will combine means of studies by conducting a meta-analysis of all group research design studies. We anticipate using a random effects meta-analysis due to likely variation within and between studies with respect to intervention protocol. We will provide a narrative description of the single subject experimental design studies since unbiased meta-analytic techniques have not been established (Wolery et al.,

8 8 2010). We also will provide a narrative description of results for the group research studies if meta-analysis is inappropriate.

9 9 References: Ayers, A. J. (1972). Sensory integration and learning disorders. Los Angeles: Western Psychological Services. Ayres, A. J. (1979). Sensory integration and the child. Los Angeles: Western Psychological Services. Chakrabarti, S., & Fombonne, E. (2005). Pervasive developmental disorders in preschool children: Confirmation of high prevalence. American Journal of Psychiatry, 162, Cicchetti, D. (Ed.). (2011). Allostatic Load, [Special Issue]. Development and Psychopathology, Part 1, 23(3), ; Part 2, 23(4), Fombonne E. (2005). Epidemiological studies of pervasive developmental disorders. In F. R. Volkmar, R. Paul, A. Klin, D. Cohen (Eds). Handbook of Autism and Pervasive Developmental Disorders. (pp ). Hoboken, NJ: Wiley. Green, V., Pituch, K.A., Itchon, J., Choi, A., O Reilly, M. & Sigafoos, J. (2006) Internet survey of treatments used by parents of children with autism. Research in Developmental Disabilities, 26, Hedges, L., & Olkin, I. Statistical Models For Meta-Analysis. New York: Academic Press, Higgins, J. P. T., & Thompson, S. G. (2002). Quantifying heterogeneity in a meta-analysis. Statistics in Medicine, 21, Leekam, S. R., Nieto, C., Libby, S. J., Wing, L., & Gould, J. (2007). Describing the sensory abnormalities of children and adults with autism. Journal of Autism and Developmental Disorders, 37, Olson, L. J., & Moulton, H. J. (2004). Use of weighted vests in pediatric occupational therapy practice. Physical and Occupational Therapy in Pediatrics, 24, Reichow, B. (2011). Development, procedures, and application of the evaluative method for determining evidence-based practices in autism. In B. Reichow, P. Doehring, D. V. Cicchetti, & F. R. Volkmar (Eds.). Evidence-based practices and treatments for children with autism (pp ). New York, NY: Springer. Reichow, B., Barton, E. E., Volkmar, F. R., & Cicchetti, D. V. (2007, May). The status of research on interventions for young children with autism spectrum disorders. Poster presented at the International Meeting for Autism Research, Seattle, WA. Reichow, B., Barton, E. E., Neely, J., Good, L., & Wolery, M. (2010). Effects of weighted vests on the engagement of children with developmental delays and autism. Focus on Autism and Developmental Disabilities, 25, Reichow, B., Barton, E. E., Good, L., & Wolery, M. (2009). Effects of wearing a pressure vest on engagement of a child with developmental disabilities. Journal of Autism and Developmental Disorders, 39, Reichow, B., & Volkmar, F. R. (2010). Best-evidence synthesis of social skills interventions for individuals with autism spectrum disorders. Journal of Autism and Developmental Disorders, 40, Reichow, B., Volkmar, F. R., & Cicchetti, D. V. (2008). Development of an evaluative method for determining the strength of research evidence in autism. Journal of Autism and Developmental Disorders, 38, Schaaf, R. C. (2011). Interventions that address sensory dysfunction for individuals with Autism Spectrum Disorders: Preliminary evidence for the superiority of sensory integration compared to other sensory approaches. In B. Reichow, P. Doehring, D. V. Cicchetti, & F. R. Volkmar (Eds.). Evidence-based practices and treatments for children with autism. New York, NY: Springer.

10 Siegel, M., & Beaulieu, A. A. (2012). Psychotropic medications in children with autism spectrum disorders: A systematic review and synthesis for evidence-based practice. Journal of Autism and Developmental Disorders, 42, Spector, J. E. (2011). Sight word instruction for students with autism: An evaluation of the evidence base. Journal of Autism and Developmental Disorders, 41, Sterne, J. A. C., Egger M., & Moher, D. (2008). Addressing reporting biases. In J. P. T. Higgins, S. Green (Eds.) (pp ). Cochrane Handbook for Systematic Reviews of Interventions. Chichester: John Wiley & Sons. Volkmar, F. R., State, M., & Klin, A. (2009). Autism and autism spectrum disorders: diagnostic issues for the coming decade. Journal of Child Psychology and Psychiatry, 50, Wolery, M., Busick, M., Reichow, B., & Barton, E. E. (2010). Comparison of overlap methods for quantitatively synthesizing single subject data. Journal of Special Education, 44,

11 11 List of Abbreviations APA American Psychiatric Association ASD autism spectrum disorder DSM-IV Diagnostic and Statistical Manual, 4 th. edition DSM-5 Diagnostic and Statistical Manual, 5 th edition OT occupational therapist SIT sensory integration therapy

12 12 Competing Interests Drs. Erin E. Barton and Brian Reichow each receive royalties from book publications and honoraria for lectures on autism spectrum disorders, but declare no competing interests with the content and scope of this review. Author s Contributions EB and BR drafted and contributed to the development of this protocol. Graduate assistants will screen the abstracts and titles and retrieve potentially eligible papers and then BR and EB will make decisions about eligibility. We will resolve any disagreements by conference until there is agreement. BR and EB will independently extract data, again resolving any disagreements by conference until agreement is reached. Authors Information Erin E. Barton, Ph.D., is an assistant professor of early childhood special education at University of Colorado Denver. Brian Reichow, Ph.D., is an assistant professor of community medicine and health care and research director at the AJ Pappanikou Center of Excellence on Developmental Disabilities. Part of his work on this project was completed while he was associate research scientist at Yale Child Study Center. Acknowledgments The authors would like to acknowledge the assistance of Hannah Embree and Keith Kravec for their assistance with this review.

13 13 Appendix A. Search Strategy Medline 1. Weighted vest 2. Pressure vest 3. Topic=(("sensory intervention" OR "sensory treatment" OR "sensory integration") AND "autism") 4. Sensory diet 5. Sensory dysfunction 6. (Topic=("PDD-NOS") OR (Topic=("pervasive developmental disorder") AND Topic=("sensory") 7. 1 and 2 and 3 and 4 and 5 8. limit 6 to (English language and humans and ( all infant (birth to 23 months) or all child (0 to 18 years) ) PsycINFO and ERIC 1. weighted vest 2. pressure vest 3. all((autism spectrum disorder OR children with autism OR students with autism)) AND all((sensory treatment OR sensory intervention)) 4. sensory diet AND autism 5. sensory diet AND asd 6. all(sensory diet) AND all((pdd-nos OR pervasive developmental disorder )) 7. all(sensory diet) AND all((students with autism)) 8. all(sensory diet) AND all((children with autism)) 9. all("children with asd") AND all((sensory intervention OR sensory treatment OR sensory integration)) 10. all(pdd-nos OR "pervasive developmental disorder" ) AND all((sensory interventions OR sensory treatments OR sensory integration)) 11. all("sensory integration") AND all(("autism spectrum disorder" OR children with autism OR students with autism)) and 2 and 3 and 4 and 5 and 6 and 7 and 8 and 9 and 10 and limit 12 to (peer reviewed journal and english language)

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