Abstract
The findings of this study enhance occupational therapy practitioners’ understanding of the association between sensory features and high-order repetitive and restricted behaviors, which may assist in planning more efficient interventions.
Sensory features have long been associated with autism spectrum disorder (ASD) and are currently included in the Diagnostic and Statistical Manual of Mental Disorders (5th ed.; DSM–5; American Psychiatric Association [APA], 2013) classification system for ASD, under the category of “restricted, repetitive patterns of behaviors, interests, or activities” (RRBIs) as they relate to “hyper- or hypo-reactivity to sensory input or unusual interests in sensory aspects of the environment.” The professional literature on ASD reports that approximately 45% to 95% of this population experience atypical sensory features, presenting across all sensory modalities, throughout their life span (Ben-Sasson et al., 2009). Sensory features strongly affect the daily routines of children with ASD and those of their families, leading most parents to seek occupational therapy evaluation and intervention for their children (Kirby et al., 2017).
Sensory features in ASD include sensory modulation disorders, which consist of three atypical sensory response patterns: (1) hyperresponsiveness, reflecting a low sensory threshold that may lead to an aversive reaction or avoidance of sensory input; (2) hyporesponsiveness, relating to a high sensory threshold that can result in a delayed or diminished response to sensory input; and (3) sensory-seeking behaviors, relating to a craving for or attraction to certain sensory stimuli (Ausderau et al., 2014; Baranek et al., 2006). Distinct sensory patterns previously found in children with ASD suggest that age and IQ may moderate certain sensory patterns in this population. For example, hyporesponsiveness characterized children with ASD who had a lower proxy IQ and those who were younger (i.e., toddlers; Ausderau et al., 2014), whereas hyperresponsiveness and sensory-seeking behaviors were highest among children in the 6- to 9-yr-old age group and decreased thereafter (Ben-Sasson et al., 2009). Thus, it is important to explore the sensory features of children with ASD while focusing on a narrow age band and taking their cognitive level into consideration.
Association Between Sensory Features and Other RRBIs in Autism Spectrum Disorder
RRBIs is an umbrella term that refers to a group of heterogeneous behaviors marked by repetition, rigidity, invariance, and inappropriateness to the place and context of the behavior. These behaviors can range from low order (stereotypy, self-injury) to high order (compulsions, rituals/sameness, restricted interests; Lin & Koegel, 2018; Turner, 1999). RRBIs—specifically, high-order RRBIs, such as restricted interests and challenging behaviors in response to changes to preferred routines—often interfere with learning skills and the ability to complete self-care and leisure activities and are considered challenging for family members and educators (Gal, 2011; Lin & Koegel, 2018). Previous studies have shown an association between lower IQ levels and repetitive movements, although high-order RRBIs are thought to be more prevalent among children with ASD who do not have a concomitant intellectual disability (Bodfish et al., 2000; South et al., 2005). Indeed, past studies have revealed that RRBIs—specifically, low-order RRBIs—are associated with sensory features in ASD (Chen et al., 2009; Gal et al., 2002), although there is a paucity of knowledge concerning the possible association of high-order RRBIs and sensory features in ASD, specifically among children with ASD who have average to above-average cognitive abilities. Few studies have addressed the association between limited aspects of sensory patterns, mostly hyperresponsiveness, with RRBIs or have examined a specific aspect of high-order RRBIs, such as an insistence on sameness (Black et al., 2017; Schulz & Stevenson, 2019). Boyd et al. (2010) found a strong association between hyperresponsiveness (but not hyporesponsiveness) and stereotypy, compulsions, and rituals in young children with ASD or other developmental disorders, controlling for mental age, and sensory seeking was significantly associated only with rituals and sameness behaviors.
The extant literature also has explored sensory modalities associated with RRBIs, focusing sometimes on the association of symptoms of an individual sensory modality with RRBIs (Foss-Feig et al., 2012). Studies have revealed a positive association between modulation disorders in visual, auditory, and tactile modalities and the intensity and frequency of RRBIs in ASD (Chen et al., 2009; Schulz & Stevenson, 2019), although this association has not been examined across various subtypes of RRBIs. In addition, the expression of sensory patterns among children with ASD has context-specific qualities, relating specifically to social versus nonsocial contexts (Baranek et al., 2013; Bodfish et al., 2000). Socially oriented stimuli include those found in a human context, such as faces, voices, and interpersonal touch, and are reported by parents of children with ASD as enjoyable for their children (Dickie et al., 2009). Researchers have found that hyporesponsiveness in children with ASD was more pronounced in social contexts compared with typically developing children or those with other developmental disabilities (Baranek et al., 2013). Social reinforcement may also contribute to the appearance of certain repetitive behaviors in ASD (Turner, 1999); however, parents of children with ASD report repetitive behaviors in response to nonsocial sensory stimuli, such as lights, objects, and so forth (Dickie et al., 2009).
In the current study, we addressed the nuanced associations between high-order RRBIs and different sensory patterns, sensory modalities, and sensory contexts, as assessed with well-validated measures. We hypothesized that sensory patterns (hyperresponsiveness, hyporesponsiveness, and sensory-seeking behaviors) would be associated with, and predict, high-order RRBIs, including an insistence on sameness, narrow interests, rigid routines, and rituals, among children with ASD who are cognitively able. We further hypothesized that sensory modalities (vision, hearing, etc.) and sensory contexts (social and nonsocial) would be associated with high-order RRBIs.
Method
Participants
Participants in this study included 39 children (34 boys, 5 girls; M age = 7.99 yr, SD = 1.15) studying in the general education system in Israel with a clinical diagnosis of ASD. The ASD diagnosis was determined by a physician or a psychologist according to the DSM–5 criteria and/or the Autism Diagnostic Observation Schedule–Generic (Lord et al., 2000). The two inclusion criteria for this study were (1) confirmation of the ASD diagnosis through the Childhood Asperger Syndrome Test (CAST; Williams et al., 2005), which sets a cutoff score as >15 for a potential ASD diagnosis, and (2) a receptive language (as a proxy for IQ) score >75, as confirmed by the Peabody Picture Vocabulary Test (3rd ed.; PPVT–III; Dunn & Dunn, 1997). The children’s mean receptive language score on the PPVT–III was 99.19 (SD = 12.53, range = 76–128). Children with seizure disorders, acute or chronic medical conditions, or known genetic conditions were excluded. The original sample included 41 children with ASD; 2 were dropped from the study because they did not meet the inclusion criteria.
Instruments
The Sensory Experiences Questionnaire, Version 2.1 (SEQ; Baranek, 1999; Baranek et al., 2006), is a 43-item caregiver instrument that examines sensory features in young children with ASD and other developmental disabilities, in line with the evolving conceptual model of sensory processing among children with ASD. Data collection for the current study was held before the updated version of the SEQ was completed; thus, we used Version 2.1. The quantitative SEQ items focus on three patterns of sensory responsiveness, within both social and nonsocial contexts. The items also can be grouped by modality (auditory, tactile, visual, vestibular/proprioceptive, and gustatory/olfactory). Items are rated on a Likert scale that ranges from 1 (almost never) to 5 (almost always). The SEQ has well-established reliability and validity (e.g., Baranek et al., 2006; Boyd et al., 2010). The internal consistencies of the subscales are as follows: Hyperresponsiveness, α = .73; Hyporesponsiveness, α = .75; Sensory Seeking, α = .80; Social Context, α = .69; and Nonsocial Context, α = .78 (Little et al., 2011). Higher scores indicate higher levels of sensory features. Summary scores may be interpreted as within the typical range, the at-risk range, or the deficient range for sensory processing function on the basis of the distributions (means and standard deviations) of the normative sample. To minimize cultural and linguistic biases, for this study the SEQ was translated to Hebrew and then back-translated by two bilingual occupational therapists and approved by Neta Katz Zetler. The internal consistency of the translated SEQ Total score was high (α = .87), as it was for the sensory patterns (hyperresponsiveness, α = .83; hyporesponsiveness, α = .74; sensory-seeking behaviors, α = .82; social context, α = .75; and nonsocial context, α = .88). Raw scores were used for the RRBIs correlational analyses.
The Repetitive Behavior Scale–Revised (RBS–R; Bodfish et al., 1999; Lam & Aman, 2007) is a 43-item caregiver questionnaire that has six subscales: (1) Stereotyped Behavior, (2) Self-Injurious Behavior, (3) Compulsive Behavior, (4) Routine Behavior, (5) Sameness Behavior, and (6) Restricted Interests. Items are rated on a scale that ranges from 0 (behavior does not occur) to 3 (behavior occurs and is a severe problem). Factor loadings range from 0.51 to 0.66 and account for 47.5% of the variance (Lam & Aman, 2007). The subscales’ internal consistency ranges from .78 to .91, interrater reliability ranges from .57 to .73, and strong discriminant validity between adults with cognitive delays with and without autism has been observed (Bodfish et al., 2000). We used a translated version of the RBS–R that received approval from the authors. We focused on the high-order RRBIs subscales as measured by raw scores, excluding the Stereotyped Behavior subscale, which represents low-order RRBIs and has been associated with relevant sections of the SEQ used in this study (Boyd et al., 2010). Moreover, we omitted the Self-Injurious Behavior subscale from the RBS–R Total score, in accordance with the current DSM–5 definition of repetitive and restricted behaviors that do not include self-injurious behavior. The internal consistency of the RBS–R Total score in the current study, not including the above-mentioned subscales, was .95, and the internal consistency values of the of the relevant subscales ranged from .79 to .91.
Procedure
This study was approved by ethics committees at the University of Haifa and the Israeli Ministry of Education. Flyers explaining the purpose of the study were sent to families of children with ASD by their teachers. All participating parents signed a written consent form and completed the CAST. The PPVT–III was administered at the children’s school setting, in a quiet room, by a trained occupational therapist. After this, parents completed the SEQ and the RBS–R and returned the questionnaires by mail. Questionnaires were coded by a trained occupational therapist with 20 yr of experience.
Data Analysis
We used descriptive statistics to examine the sensory features of children with ASD in comparison with those of typically developing children on the SEQ across patterns (hyporesponsiveness, hyperresponsiveness, and sensory-seeking behaviors), modalities, and contexts. We examined the association between sensory features and RRBIs by computing the correlation between the revised RBS–R Total raw score and the SEQ Total raw score. Next, we calculated Pearson correlations to determine the association between sensory response patterns across sensory modalities (auditory, tactile, visual, vestibular/proprioceptive, and gustatory/olfactory), as well as across contexts (social and nonsocial) and the separate RRB subscales tapping high-order RRBIs (including Compulsive Behavior, Routine Behavior, Sameness Behavior, and Restricted Interests). We also conducted a linear regression analysis to examine whether sensory patterns predicted RRBIs among children with ASD. IBM SPSS Statistics (Version 23) was used for data analyses. Significance was set at p ≤ .05, and Bonferroni corrections were applied to the correlational analyses.
Results
Parents of 39 children with ASD completed the SEQ and the RBS–R. The children’s ages ranged from 6 yr, 3 mo, to 10 yr, 6 mo (skewness = 0.14, kurtosis = –0.73), thus suggesting a normal distribution of age. No statistically significant correlations were found between sensory and repetitive behaviors and the children’s age or proxy IQ.
Sensory Experiences Questionnaire Distribution of Scores Among Children With Autism Spectrum Disorder
We determined the level of sensory features across sensory response patterns and contexts among children with ASD in the sample (categorically defined as typical, at risk, or deficient) using the SEQ manual. A higher percentage of children with ASD showed atypical sensory features in the hyperresponsiveness pattern, followed by hyporesponsiveness and then by sensory-seeking behaviors (Table 1). In regard to context, a higher percentage of children with ASD showed atypical sensory features in the social context than in the nonsocial context (see Table 1).
SEQ Distribution of Scores Among Children With ASD (N = 39)
Note. ASD = autism spectrum disorder; SEQ = Sensory Experiences Questionnaire.
Association Between Sensory Features and RRBIs
We examined the association between sensory features and RRBIs using a correlation procedure. A statistically significant correlation was found between the RBS–R (total score for RBS high-order behaviors) and the SEQ Total scores (r = .81, p < .01). In regard to the association between RRBIs and SEQ sensory patterns, the results demonstrated that sensory hyperresponsiveness was significantly correlated with the RBS–R Total score (r = .72, p < .01) as well as with all subscales of the RBS–R administered in this study, including Sameness Behavior (r = .67, p < .01), Ritualistic Behavior (r = .72, p < .01), Restricted Behavior (r = .69, p < .01), and Compulsive Behavior (r = .53, p < .01). Sensory hyporesponsiveness was significantly correlated with the RBS–R Total score (r = .68, p < .01), as well as with all of the RBS–R subscales (Sameness Behavior, r = .71, p < .01; Ritualistic Behavior, r = .67, p < .01; Restricted Behavior, r = .58, p < .01; and Compulsive Behavior, r = .49, p < .01). Similarly, sensory-seeking behaviors shared a statistically significant correlation with the RBS–R Total score (r = .70, p < .01) and all the RBS–R subscales (Sameness Behavior, r = .66, p < .01; Ritualistic Behavior, r = .68, p < .01; Restricted Behavior, r = .66, p < .01; and Compulsive Behavior, r = .55, p < .01).
We then used stepwise linear regression to examine the possible effect of each sensory pattern on the RBS–R Total scores of children with ASD and noted that the sensory hyperresponsiveness behaviors of children with ASD predicted 51% of the RBS–R Total score, whereas sensory-seeking behaviors predicted an additional 11% of the RBS–R Total score. Hyporesponsiveness did not contribute further to the prediction of the RBS–R Total score. Thus, the findings confirm our hypothesis predicting a statistically significant association between high-order RRBIs and sensory features across patterns in ASD, and hyperresponsiveness and sensory-seeking behaviors were predictive of high-order RRBIs.
Association Between RRBIs and Sensory Modalities
The sensory modalities most strongly associated with the higher order RRBIs as expressed in the RBS–R’s Total score were auditory (r = .71, p < .01) and visual (r = .72, p < .01), followed by tactile and gustatory/olfactory features (r = .49, p < .01, and r = .58, p < .01, respectively), and vestibular/proprioceptive features were the least correlated among the sensory modalities with the RBS–R Total score (r = .36, p < .05; Table 2). These results confirm that sensory features across modalities are associated with RRBIs.
Correlations Between Sensory Modalities and RBS–R Scores in Children With ASD
Note. ASD = autism spectrum disorder; ns = nonsignificant; RBS–R = Repetitive Behavior Scale–Revised; SEQ = Sensory Experiences Questionnaire.
p ≤ .05.
p ≤ .01.
Association Between RRBIs and the Social and Nonsocial Contexts of Sensory Features
Both social and nonsocial contexts on the SEQ correlated with high-order RRBIs, confirming our initial hypotheses. The social context of sensory features significantly correlated with the RBS–R Total score (r = .63, p < .01), as well as with insistence on sameness (r = .59, p < .01), rituals (r = .70, p < .01), restricted interests (r = .67, p < .01), and compulsive behaviors (r = .44, p < .01). In addition, the nonsocial context of sensory features was significantly correlated with the RBS–R Total score (r = .79, p < .01), as well as with all subscales of the RBS–R, including Sameness Behavior (r = .76, p < .01), Ritualistic Behavior (r = .75, p < .01), Restricted Behavior (r = .69, p < .01), and Compulsive Behavior (r = .61, p < .01).
Discussion
The definition of sensory symptoms in general, and in people with ASD in particular, has been an issue of some debate over the past few decades. Various attempts have been made to characterize specific sensory patterns on the basis of the combination of clinically meaningful symptom sets (Dunn, 1997; Miller et al., 2007). One such characterization divides sensory modulation disorders into three categories: (1) sensory underresponsivity/sensory hyporesponsiveness, (2) sensory overresponsivity/sensory hyperresponsiveness, and (3) sensory seeking (Miller et al., 2007). The results of the current study, which relate to children with ASD ages 6 to 10 yr, suggest that children with ASD who have no intellectual disabilities present with high levels of hyperresponsiveness and relatively lower levels of hyporesponsiveness and sensory-seeking behaviors.
Although sensory features were not considered in the Diagnostic and Statistical Manual of Mental Disorders (4th ed.; APA, 1994) as part of the ASD diagnosis, under the current ASD diagnostic criteria (DSM–5) they are considered a criterion for RRBIs. In contrast to previous knowledge concerning associations between sensory response patterns and specific high-order RRBIs, the results of the current study demonstrate that a statistically significant association exists between all three sensory response patterns and the various high-order RRBIs, including compulsive behavior, routine behavior, sameness behavior, and restricted interests. Moreover, in accordance with previous data (Chen et al., 2009; Schulz & Stevenson, 2019), our findings indicate that the pattern of sensory hyperresponsiveness predicted most of the high-order RRBIs for children with ASD (with proxy IQs >75), thus providing additional support for inclusion of some sensory features together with RRBIs in the DSM–5.
Theoretical explanations suggest that RRBIs may serve as a self-regulating mechanism for people with ASD in situations of sensory overarousal and stress (Boyd et al., 2010; Gal et al., 2002). For example, hyperresponsiveness to sensory stimuli might lead a person with ASD to experience sensory overload, which in turn can stimulate them to engage in repetitive movements as a calming protective behavior, or to insist on sameness (Schulz & Stevenson, 2019; Turner, 1999). Moreover, low-order RRBIs, such as stereotyped movements, may serve as a self-stimulation mechanism of people with ASD who exhibit hyporesponsiveness to sensory stimuli (Gal et al., 2002). Our study supports correlations of high-order RRBIs with both hypo- and hyperresponsiveness, suggesting that these repetitive behaviors may serve as self-regulating mechanisms in a deviation from typical sensory processing. However, in our sample of children with proxy IQs >75, it was mainly hyperresponsiveness that predicted these behaviors.
In accordance with previous findings (Chen et al., 2009; Schulz & Stevenson, 2019), our results reveal that the auditory, visual, and tactile features were most strongly associated with RRBIs in children with ASD. However, past results have at times related to only a limited aspect of RRBIs (Chen et al., 2009) or did not include an examination of the possible involvement of all sensory modalities (Schulz & Stevenson, 2019). Indeed, many of the high-order RRBI items presented in the RBS–R relate to the auditory and visual modalities (e.g., listening or watching the same movie or TV program over and over) as well as to the tactile modality. Although in this study we identified only a relatively weak correlation of vestibular/proprioceptive items with high-order RRBIs, the correlation between vestibular stimuli and low-order RRBIs has been noted in previous research. Gal and Dyck (2009) found that blind children were more likely to engage in rocking and repetitive head movements in comparison with children with low vision, suggesting that stereotyped movements such as proprioceptive and vestibular stimulation may serve as a compensation in situations that lack sensory input. When added to earlier research suggesting a correlation between specific stereotyped movements and specific disabilities, the current study further suggests that specific high-order RRBIs may serve as a compensation for the discomfort caused by visual, auditory, and tactile abnormalities.
This study strengthens past evidence concerning the contribution of both social and nonsocial contexts to the expression of sensory features and high-order RRBIs. Past studies have shown that the child’s behaviors may change depending on the environment in which they occur (Brown & Dunn, 2010) and that sensory experiences are perceived by children with ASD as occurring in a particular daily context (Dickie et al., 2009). Baranek et al. (2013) found that hyporesponsiveness behaviors of children with ASD were more pronounced within social context. Moreover, Kirby et al. (2017), who in their study used in-home naturalistic video recordings, found that hyperresponsiveness as well as sensory-seeking and repetitive/stereotypic behaviors were most commonly associated with nonsocial sensory stimuli in children with ASD. Additional contextual factors that have been suggested to contribute to the expression of sensory and repetitive behaviors in children with ASD include the child’s familiarity with their surroundings, the activities in which they are engaged, and the characteristics of the environment (Dickie et al., 2009; Kirby et al., 2017).
The current study enhances occupational therapists’ understanding of high-order RRBIs and their association with sensory features of children with ASD as embedded within social and nonsocial contexts. High-order RRBI, such as insistence on sameness and special restricted interests, may present various challenges to the child and their social environment; for example, these RRBIs may prove awkward in social situations or act counter to societal expectations for the child’s age and gender. They may have great advantages as well, however. For example, for some children with ASD their special abilities, once directed, guided, and expanded, could evolve into a meaningful occupation and even, later in life, a career (Gal, 2011). Therefore, when addressing RRBIs, occupational therapy practitioners may need to carefully consider the relative benefits of interventions aimed to reduce atypical behaviors that interfere with a child’s participation versus shaping occupations and empowering the child’s strengths in pursuit of increased participation (Gal & Ben-Sasson, 2021). In addition, occupational therapy practitioners should carefully examine each child’s sensory profile to determine the extent to which underlying sensory-processing challenges may be contributing to specific RRBIs and use evidence-based interventions, including strategies for self-regulation, to address them. Moreover, occupational therapy interventions should address the specific contexts that inhibit or support the child’s participation in daily life.
Despite the robustness of the current findings, caution should be taken in generalizing the results, given that this study relied solely on parental report questionnaires, which may be subjective. Despite the well-established validity and reliability of the SEQ and RBS–R, recent studies suggest that the proxy inferences related to the internal and unconfirmed states of others may be influenced by parents’ knowledge and experience (Wolff et al., 2019). Future studies might benefit from investigation of these variables using observational measures of sensory features and RRBIs in both standardized clinical and naturalistic contexts, as well as from first-hand accounts. In addition, the relatively small sample size, and the relative sample’s homogeneity with respect to age and IQ levels, limit the generalizability of the findings to the general population of people with ASD. Future research should include larger samples of people with ASD from all developmental levels and across the life span and should consider subtyping or moderation analyses to better understand the heterogeneous nature of associations between co-occurring sensory features and RRBIs.
Implications for Occupational Therapy Practice
This study has the following implications for occupational therapy practice: High-order RRBIs of children with ASD (with a proxy IQ >75) are associated with sensory features, especially the pattern of sensory hyperresponsiveness. Occupational therapists are encouraged to carefully assess children’s RRBIs in tandem with their sensory profiles in the context of daily life activities and routines and to consider evidence-based interventions to optimize participation in both social and nonsocial contexts.
Conclusion
This study implies that the unique sensory profile of children with ASD, across sensory modalities, patterns, and contexts, may contribute to the appearance of high-order RRBIs in daily life activities. Findings encourage the development of evidence-based interventions in occupational therapy, including sensory self-regulation strategies that may affect RRBIs of children with ASD and enhance their participation. Future studies are recommended to use first-hand accounts to investigate the association between sensory and repetitive behaviors of people with ASD across the life span.
Footnotes
Acknowledgments
We thank the families who participated in the study.
