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Running Head: Sensory and Social Subtypes with Autism Spectrum Disorders 1 1 Running head: Sensory and social subtypes with autism spectrum disorders 2 3 Sensory and social subtypes of Japanese individuals with autism spectrum disorders 4 5 Ayako Kaneko1,2*, Remi Ohshima3, Haruka Noda1,2,4, Tomoko Matsumaru5, Ryoichiro Iwanaga4, Masakazu 6 Ide1 7 8 1. Department of Rehabilitation for Brain Functions, Research Institute of National Rehabilitation Center for 9 Persons with Disabilities, Tokorozawa, Saitama, Japan 10 2. Japan Society for the Promotion of Science, Chiyoda, Tokyo, Japan 11 3. Faculty of Psychology, Mejiro University, Shinjuku, Tokyo, Japan 12 4. Department of Occupational therapy Science, Nagasaki University Graduate school of Biomedical sciences, 13 Nagasaki, Nagasaki, Japan 14 5. Faculty of Human sciences, Mejiro University, Shinjuku, Tokyo, Japan 15 2 1 Abstract 2 Studies have proposed that individuals with autism spectrum disorder (ASD) can be divided into several 3 subtypes depending on their sensory features. However, consideration of social communication features is also 4 crucial for configuring ASD subtypes, because social and sensory features are tightly interrelated. In this study, 5 we asked Japanese individuals with ASD to answer the Short Sensory Profile (SSP) and the Social 6 Responsiveness Scale, Second Edition (SRS-2), which measure sensory and social aspects, respectively. 7 Consequent latent profile analysis demonstrated that the participants could be divided into five subgroups: two 8 groups exhibited opposite or inconsistent patterns between the SSP and SRS-2 scores, while the other groups 9 exhibited consistent patterns. Our findings indicate the existence of diverse phenotypes in individuals with 10 ASD. 11 Keywords 12 Autism spectrum disorder; Subgroups; Hyper-reactivity; Hypo-reactivity; Sensory profile; Social 13 Responsiveness Scale 14 15 *Corresponding authors: Ayako Kaneko and Masakazu Ide 16 e-mail: [email protected] (A.K.), [email protected] (M.I.) 17 18 Compliance with Ethical Standards 19 This study was supported by the Japan Society for the Promotion of Science (No. 18J22022, No. 18H03140, 20 No. 21J01032). The authors declare that they have no conflicts of interest. All procedures involving human 21 participants were performed in accordance with the ethical standards of the ethics committee of Rikkyo 3 1 University and with the 1964 Declaration of Helsinki and its later amendments or comparable ethical 2 standards. All participants and their parents received a full explanation of the study procedures and then 3 provided written informed consent. 4 1 Introduction 2 A large body of studies across a broad range of research fields has focused on abnormal sensory features that 3 are found in over 90% of autism spectrum disorder (ASD) cases (Crane, Goddard, & Pring, 2009; Green, 4 Chandler, Charman, Simonoff, & Baird, 2016; Tomchek & Dunn, 2007). As a result of this increased 5 attention, in addition to core symptoms (i.e., difficulties regarding social communication and 6 restricted/repetitive behaviors [RRB]), atypical sensory processing is now included among the diagnosis 7 criteria for ASD in the fifth edition of the Diagnostic and Statistical Manual of Mental Disorders (DSM-5, 8 2013). Dunn (1997) proposed that sensory-processing disorders are comprised of several sub-components: 9 sensory sensitivity, sensory avoidance, low registration, and sensation seeking. In the Sensory Profile (SP; 10 Dunn, 1997), which is a tool that was developed to estimate individuals’ degree of abnormal sensory 11 processing, the former two components represent traits of hyper-reactivity to sensory stimuli, while the latter 12 two represent hypo-reactivity. 13 Several researchers have attempted to categorize individuals with ASD into subgroups based on 14 their sensory features. In a small sample study, 54 subjects with ASD were administered the short version of 15 the SP (SSP), and the obtained data underwent cluster analysis; consequently, three subgroups were identified: 16 sensory-based inattentive seeking (group 1), sensory modulation with movement sensitivity (group 2), and 17 sensory modulation with taste/smell sensitivity (group 3; Lane et al., 2010). The subjects from group 1 18 exhibited modest sensory problems, while the opposite trends were observed for group 2, with the constituent 19 subjects showing severe sensory problems. The subjects in group 3 exhibited remarkable problems in terms of 20 taste and smell, as well as sensitivity to their own body energy and postures. In order to confirm these findings, 21 Lane, Dennis, and Geraghty (2011) performed an analysis of 30 individuals with ASD; they consequently 5 1 confirmed the presence of the three subtypes identified in Lane et al. (2010); however, they observed five 2 clusters in total. They determined that the additional clusters could be considered subsets of group 1 and group 3 2, respectively. Furthermore, Lane, Molloy, and Bishop (2014), through analysis of a relatively large sample of 4 individuals with ASD (N = 228), identified four subgroups: sensory adaptive, taste/smell sensitive, postural 5 inattentive, and generalized sensory difference. Two of these four subgroups (sensory adaptive and generalized 6 sensory difference) corresponded to those proposed in Lane et al. (2010, 2011), while individuals from Lane et 7 al.’s (2010) subgroup of sensory modulation with taste/smell sensitivity (group 3) could be assumed to be 8 divided into two different subgroups in Lane et al. (2014): taste/smell sensitive and postural inattentive, 9 respectively. Additionally, Lane et al. (2014) compared the severity of autism symptoms, which were 10 evaluated using the Autism Diagnosis Observation Schedule (Lord et al., 2000), across the four groups, and 11 did not find any significant intergroup differences. Ausderau et al. (2014), using the Sensory Experience 12 Questionnaire (SEQ; Baranek, David, Poe, Stone, & Watson, 2006), classified a large number of subjects (N = 13 1294) into four subgroups, and found identical patterns concerning the clustering of the groups to those 14 reported by Lane et al. (2014). They also compared other features of ASD, measured using the Social 15 Responsiveness Scale-Preschool Version (SRS-P; Pine, Luby, Abbacchi, & Constantino, 2006), between the 16 four groups, and found that persons classified to the group with generalized sensory differences showed more 17 severe ASD symptoms than did the individuals from the other groups. These previous findings demonstrate 18 that the symptoms of ASD concerning difficulties regarding social communication and RRB are influenced by 19 sensory symptoms, and that consideration of sensory symptoms can be used to differentiate individuals with 20 ASD into several subgroups. 6 1 Few studies have aimed to identify subgroups by considering multiple, rather than single, aspects of 2 the pathognomonic condition of autism. Tomchek, Little, Myers, and Dunn (2018) attempted to categorize 3 children with ASD based on their development skills (i.e., adaptive behavior, receptive/expressive language, 4 and fine/gross motor skills), which were evaluated using a combination of measurements (the Independent 5 Behavior-Revised or the Developmental Profile II, the Preschool Language Scale: 4th edition or the Rosetti 6 Infant-Toddler Language Scale, and the Peabody Developmental Motor Scales). Additionally, the children’s 7 sensory-processing characteristics, measured using SSP, were simultaneously included in a cluster analysis. 8 Tomchek et al. (2018) consequently identified four subgroups (Group 1: Sensorimotor; Group 2: Selective- 9 Complex; Group 3: Perceive-Adaptable; Group 4: Vigilant-Engaged); the children in Group 4, which was 10 characterized by moderate hyper-reactivity and sensory seeking, showed higher developmental skill when 11 compared to persons in Group 3, which featured the children who exhibited the fewest sensory features. This 12 finding indicates that it is efficient to take several characteristics of autism into account, as this provides rich 13 information when compared to results obtained through analysis of sensory characteristics only. However, no 14 previous study has sought to categorize social communication and RRB concurrently with sensory features, 15 despite the fact that these are regarded as core features of ASD. 16 Although many studies have reported relationships between sensory features and difficulties 17 regarding social communication and/or RRB, these studies have not necessarily presented consistent results. 18 Some studies have reported that atypical sensory features are associated with difficulties in social 19 communication (Hilton et al., 2010) and atypical behaviors in social situations (Baker, Lane, Angley, & 20 Young, 2008; Lane, Young, Baker, & Angley, 2010). In contrast, Ashburner, Ziviani, and Rodger (2008) 21 reported that hypo-reactivity is related to atypical social communication, while hyper-reactivity is only related 7 1 to it in the auditory modality. Gabriels et al. (2008) reported that atypical sensory features are associated with 2 RRB, whereas Boyd et al. (2010) found that hypo-reactivity is not associated with RRB. These reports indicate 3 that, among individuals with ASD, there is a large diversity of phenotypes in relation to the combination of 4 sensory characteristics with other characteristics of ASD (i.e., social communication and RRB). Thus, it is 5 important to more accurately ascertain ASD subgroups by simultaneously considering several aspects of ASD 6 when performing clustering. 7
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