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Comparison of the Sensory Profile among Autistic Individuals and Individuals with Williams Syndrome

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Title: Comparison of the Sensory Profile among Autistic Individuals and Individuals with Williams Syndrome
Language: English
Authors: Masahiro Hirai (ORCID 0000-0003-2298-5632), Ayaka Ikeda, Takeo Kato, Takahiro Ikeda, Kosuke Asada, Yoko Hakuno, Kanae Matsushima, Tomonari Awaya, Shin Okazaki, Toshihiro Kato, Toshio Heike, Masatoshi Hagiwara, Takanori Yamagata, Kiyotaka Tomiwa, Ryo Kimura
Source: Journal of Autism and Developmental Disorders. 2025 55(6):2092-2099.
Availability: Springer. Available from: Springer Nature. One New York Plaza, Suite 4600, New York, NY 10004. Tel: 800-777-4643; Tel: 212-460-1500; Fax: 212-460-1700; e-mail: customerservice@springernature.com; Web site: https://link.springer.com/
Peer Reviewed: Y
Page Count: 8
Publication Date: 2025
Document Type: Journal Articles
Reports - Research
Descriptors: Sensory Experience, Profiles, Autism Spectrum Disorders, Genetic Disorders, Severity (of Disability), Symptoms (Individual Disorders), Personality Traits, Scores
DOI: 10.1007/s10803-023-06205-1
ISSN: 0162-3257
1573-3432
Abstract: Purpose: With the current study, we aimed to reveal the similarities and differences in sensory profiles between Williams syndrome (WS) and autism spectrum disorder. Methods: Using the sensory profile questionnaire completed by the caregivers, we analyzed the WS (n = 60, 3.4-19.8 years) and autistic (n = 39, 4.2-14.0 years) groups. Results: The Severity Analysis revealed a significant group difference in Sensory Sensitivity but not in Low Registration, Sensation Seeking, and Sensation Avoiding subscales. Age can modulate the subscale scores differently across groups. For Sensation Seeking, the scores of both groups decreased with development. However, the scores of Sensory Sensitivity decreased with age in the autistic group but not in the WS group. Sensation Avoiding scores increased with development in the WS group but not in the autistic group. No significant developmental changes were observed in Low Registration. Conclusion: This study highlights the cross-syndrome similarities and differences in sensory profiles and developmental changes in autistic individuals and individuals with WS.
Abstractor: As Provided
Entry Date: 2025
Accession Number: EJ1470827
Database: ERIC
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  Value: <anid>AN0185099706;aut01jun.25;2025May14.02:48;v2.2.500</anid> <title id="AN0185099706-1">Comparison of the Sensory Profile Among Autistic Individuals and Individuals with Williams Syndrome </title> <p>Purpose: With the current study, we aimed to reveal the similarities and differences in sensory profiles between Williams syndrome (WS) and autism spectrum disorder. Methods: Using the sensory profile questionnaire completed by the caregivers, we analyzed the WS (n = 60, 3.4–19.8 years) and autistic (n = 39, 4.2–14.0 years) groups. Results: The Severity Analysis revealed a significant group difference in Sensory Sensitivity but not in Low Registration, Sensation Seeking, and Sensation Avoiding subscales. Age can modulate the subscale scores differently across groups. For Sensation Seeking, the scores of both groups decreased with development. However, the scores of Sensory Sensitivity decreased with age in the autistic group but not in the WS group. Sensation Avoiding scores increased with development in the WS group but not in the autistic group. No significant developmental changes were observed in Low Registration. Conclusion: This study highlights the cross-syndrome similarities and differences in sensory profiles and developmental changes in autistic individuals and individuals with WS.</p> <p>Keywords: Autism spectrum disorder; Cross-syndrome; Sensory profile; Williams syndrome; Psychology and Cognitive Sciences Psychology</p> <p>Autism spectrum disorder (ASD) is a clinically heterogeneous disorder with a prevalence of 1 in every 44 people that is diagnosed by assessing difficulties in social communication, restricted interests, and atypical sensory processing according to the Diagnostic and Statistical Manual of Mental Disorders (DSM-5; American Psychiatric Association, [<reflink idref="bib1" id="ref1">1</reflink>]). The genetic background of ASD is generally heterogeneous (Lord et al., [<reflink idref="bib24" id="ref2">24</reflink>]), whereas Williams syndrome (WS) is a rare genetic disorder caused by the deletion of approximately 25–27 genes on chromosome 7 that has an estimated prevalence of 1 in every 10,000 people (Kozel et al., [<reflink idref="bib21" id="ref3">21</reflink>]). Individuals with WS exhibit hypersociability, which is characterized by a strong social interest in both familiar and unfamiliar people (e.g., Jones et al., [<reflink idref="bib16" id="ref4">16</reflink>]).</p> <p>It has been pointed out that autistic individuals and individuals with WS show opposite social phenotypes, as individuals with WS have high social motivation in approaching an unfamiliar person, in contrast to autistic individuals who show low social motivation (Chevallier et al., [<reflink idref="bib5" id="ref5">5</reflink>]). Indeed, recent cross-syndrome studies have demonstrated that social motivation in autistic individuals was lower compared to individuals with WS; however, profiles of social cognition in individuals with WS were partly similar to autistic individuals (Glod et al., [<reflink idref="bib11" id="ref6">11</reflink>]; Hirai et al., [<reflink idref="bib13" id="ref7">13</reflink>]).</p> <p>In addition to the similarities and differences in social phenotypes between autistic and WS groups (cf. Asada & Itakura, [<reflink idref="bib2" id="ref8">2</reflink>]; Klein-Tasman et al., [<reflink idref="bib20" id="ref9">20</reflink>]), similarities in atypical sensory characteristics across syndromes have been reported. An atypical sensory profile for autistic individuals was included in the DSM-5, which significantly correlates with the core features of ASD (Glod et al., [<reflink idref="bib8" id="ref10">8</reflink>]). Similarly, individuals with WS have atypical sensory characteristics such as enhanced sensory sensitivity (Glod et al. [<reflink idref="bib10" id="ref11">10</reflink>]). Hypersensitivity to certain sounds, such as machines, fireworks, and bursting balloons, has been reported in 85–95% of individuals with WS (Donnai & Karmiloff-Smith, [<reflink idref="bib6" id="ref12">6</reflink>]). Most children with WS have sensory difficulties, and based on a sensory profile questionnaire, these children were classified into two groups: high- and low-impairment groups (John & Mervis, [<reflink idref="bib15" id="ref13">15</reflink>]). Notably, the high-impairment group showed less adaptive function than the low-impairment group.</p> <p>However, to the best of our knowledge, only a few studies have directly compared the sensory profiles of autistic individuals and individuals with WS (Glod et al., [<reflink idref="bib9" id="ref14">9</reflink>], [<reflink idref="bib11" id="ref15">11</reflink>]). By comparing two typical syndromes with hypersensitivity, it is possible to reveal whether there are certain mechanisms common to both syndromes or whether the manifestation of hyper-/hypo-sensitivity varies from syndrome to syndrome. Moreover, it is expected to reveal the mechanisms of emergence of hyper-/hypo-sensitivity across syndromes by following the developmental trajectory. Most previous cross-syndrome studies have examined populations in certain age ranges and did not seem to focus on developmental trajectories; although several sensory profile studies on autistic individuals have examined developmental changes (Kern et al., [<reflink idref="bib17" id="ref16">17</reflink>]; Lane et al., [<reflink idref="bib22" id="ref17">22</reflink>]). A previous cross-syndrome study demonstrated that Social Responsiveness Scale-2 (SRS-2) scores (Hirai et al., [<reflink idref="bib13" id="ref18">13</reflink>]) can change across developmental stages. Therefore, age can affect various aspects of sensory/cognitive profiles in autistic individuals and individuals with WS; however, no empirical findings on cross-syndrome studies have been reported. Therefore, the current study aimed to assess the similarities and differences between the sensory characteristics of autistic individuals and individuals with WS, using a sensory profile questionnaire (Dunn, [<reflink idref="bib7" id="ref19">7</reflink>]). We further explored developmental changes in the sensory profiles of autistic individuals and individuals with WS.</p> <hd id="AN0185099706-2">Methods</hd> <p></p> <hd id="AN0185099706-3">Participants</hd> <p>The participants were individuals diagnosed with WS (<emph>n =</emph> 60; mean age [<emph>M</emph><subs>age</subs>] = 9.9 years, standard deviation [<emph>SD</emph>] = 4.0, range = 3.4–19.8 years; 25 male, 35 female), autistic individuals (<emph>n</emph> = 39; <emph>M</emph><subs>age</subs> = 9.2 years, <emph>SD</emph> = 2.9, range = 4.2–14.0 years; 36 male, 3 female), and their caregivers. The ages of the autistic individuals were not significantly different from those in the WS group, <emph>t</emph>(95.5) = 1.09, <emph>p</emph> = 0.28 (Table 1). The participants with WS were recruited through a non-profit organization, "Smirhythm," a self-help group, "Elfin Kansai," and Jichi Medical University, Osaka City General Hospital, Todaiji Ryoiku Hospital for Children, and Kyoto University. Autistic individuals were recruited from Jichi Medical University.</p> <p>Table 1 Descriptive statistics of participant characteristics</p> <p> <ephtml> <table frame="hsides" rules="groups"><thead><tr><th align="left" /><th align="left"><p>ASD (<italic>n</italic> = 39)</p></th><th align="left"><p>WS (<italic>n</italic> = 60)</p></th></tr></thead><tbody><tr><td align="left"><p>Numbers of male/female participants</p></td><td align="left"><p>36/3</p></td><td align="left"><p>25/35</p></td></tr><tr><td align="left"><p>Age in months (<italic>SD</italic>)</p></td><td align="left"><p>109.9 (34.9)</p></td><td align="left"><p>118.9 (47.5)</p></td></tr><tr><td align="left"><p>Age in years (<italic>SD</italic>)</p></td><td align="left"><p>9.2 (2.9)</p></td><td align="left"><p>9.9 (4.0)</p></td></tr><tr><td align="left"><p>Age in months range</p></td><td align="left"><p>50–168</p></td><td align="left"><p>41–237</p></td></tr><tr><td align="left"><p>Age in years range</p><p>PVT-R in months</p><p>RCPM</p><p>AQ</p><p>SCQ</p></td><td align="left"><p>4.2–14.0</p><p>104.8 (38.2)</p><p>26.5 (9.1)</p><p>27.4 (7.8)</p><p>12.8 (6.3)</p></td><td align="left"><p>3.4–19.8</p><p>81.3 (33.3) <italic>n</italic> = 26</p><p>13.4 (6.4) <italic>n</italic> = 27</p><p>21.3 (5.6) <italic>n</italic> = 28</p><p>10.9 (5.9) <italic>n</italic> = 28</p></td></tr></tbody></table> </ephtml> </p> <p> <emph>ASD</emph> autism spectrum disorder; <emph>WS</emph> Williams syndrome; <emph>SD</emph> standard deviation; <emph>n</emph> number of participants; <emph>PVT-R</emph> Picture Vocabulary Test－Revised; <emph>RCPM</emph> Raven's Colored Progressive Matrices; <emph>AQ</emph> Autism Spectrum Quotient; <emph>SCQ</emph> Social Communication Questionnaire</p> <p>WS was genetically confirmed using fluorescent in situ hybridization prior to participation. ASD was diagnosed based on the DSM-5 criteria by a pediatric neurologist at Jichi Medical University. Furthermore, the Social Communication Questionnaire (SCQ; Lord et al. [<reflink idref="bib23" id="ref20">23</reflink>]; Rutter et al. [<reflink idref="bib27" id="ref21">27</reflink>]) and Autism-Spectrum Quotient (AQ; Auyeung et al. [<reflink idref="bib3" id="ref22">3</reflink>]; Wakabayashi et al. [<reflink idref="bib30" id="ref23">30</reflink>]) were evaluated at Jichi Medical University. The cognitive profiles of the participants were measured via the Picture Vocabulary Test－Revised (PVT-R; Ueno et al. [<reflink idref="bib29" id="ref24">29</reflink>]) and Raven's Colored Progressive Matrices (RCPM; Sugishita and Yamazaki [<reflink idref="bib28" id="ref25">28</reflink>]) at Jichi Medical University (Table 1). Note that the WS sample from Kyoto University is a subset of the sample reported in previous studies (Kimura et al. [<reflink idref="bib18" id="ref26">18</reflink>], [<reflink idref="bib19" id="ref27">19</reflink>]). Overall, the cognitive profiles of the autistic individuals were comparable to those of typically developing individuals, whereas those with WS had cognitive profiles similar to those with intellectual disabilities.</p> <hd id="AN0185099706-4">Materials</hd> <p>The caregivers of the participants completed the Japanese translated version of the Sensory Profile (SP; Hagiwara et al., [<reflink idref="bib12" id="ref28">12</reflink>]) to measure sensory processing profiles (Dunn, [<reflink idref="bib7" id="ref29">7</reflink>]). The SP is used to rate sensory profiles and comprises 125 items covering a range of unusual sensory profiles. The caregivers rated observations of their children's symptoms on a five-point Likert scale (ranging from never [<reflink idref="bib1" id="ref30">1</reflink>] to always [<reflink idref="bib5" id="ref31">5</reflink>]). Note that the Japanese translated version of the SP differs from the original edition at two points. First, the original edition of SP targets ages 3–10, whereas the Japanese version of the SP targets ages 3–82. Second, the original version ranges from always (<reflink idref="bib1" id="ref32">1</reflink>) to never (<reflink idref="bib5" id="ref33">5</reflink>), but the Japanese version ranges from never (<reflink idref="bib1" id="ref34">1</reflink>) to always (<reflink idref="bib5" id="ref35">5</reflink>). Therefore, in the original edition of SP, the lower score has more problems with sensory processing; whereas in the Japanese version, a higher score indicates more problems with sensory processing.</p> <p>Sensory profiles measured using the SP can be categorized into one of the four sensory processing quadrants: Low Registration, do not respond to some of the usual sensory events that other people notice easily and respond to; Sensation Seeking, searching for ways of increasing sensory input actively; Sensation Avoiding, actively avoiding sensory stimuli; and Sensation Sensitivity, able to readily respond to sensory events that are not detected or noticed by other people. The maximum total scores for Low Registration, Sensation Seeking, Sensory Sensitivity, and Sensation Avoiding were 75, 130, 100, and 145, respectively.</p> <hd id="AN0185099706-5">Data Analysis</hd> <p>First, based on the SP subscales (Sensation Seeking, Sensation Avoiding, Sensory Sensitivity, and Low Registration) score, the degree of difficulty in each domain was categorized into three categories (similar to others, more than others, and much more than others) to calculate the percentage according to the criteria characterized by Japanese samples (Inada et al., [<reflink idref="bib14" id="ref36">14</reflink>]). The cut-off values for each subscale are presented in the supplementary information file (Supplementary information 1). Second, as the total subscore for each category was different, we applied a binomial logistic regression analysis to examine the relationship between the age and group for each subscale score. Likelihood ratio (LR) tests were performed to examine the significance of the regression coefficients for main effects and interactions. In addition, if the interaction was significant, a logistic regression analysis and LR test was performed for each group with subgroups to determine the simple main effect. A p-value of < 0.05 was considered statistically significant.</p> <hd id="AN0185099706-6">Results</hd> <p>Each subscale score in the autistic and WS groups is shown in Fig. 1; Table 2.</p> <p>Graph: Fig. 1 Scores for the Sensory Profile (Low Registration, Sensation Seeking, Sensory Sensitivity, and Sensation Avoiding)</p> <p>Table 2 Mean (SD) scores on outcome variables</p> <p> <ephtml> <table frame="hsides" rules="groups"><thead><tr><th align="left" /><th align="left"><p>ASD (<italic>n</italic> = 39)</p></th><th align="left"><p>WS (<italic>n</italic> = 60)</p></th></tr></thead><tbody><tr><td align="left"><p>SP quadrant</p></td><td align="left" /><td align="left" /></tr><tr><td align="left"><p>Low Registration</p></td><td char="." align="char"><p>32.3 (12.4)</p></td><td char="." align="char"><p>37.3 (12.1)</p></td></tr><tr><td align="left"><p>Sensation Seeking</p></td><td char="." align="char"><p>49.2 (16.6)</p></td><td char="." align="char"><p>50.5 (12.3)</p></td></tr><tr><td align="left"><p>Sensory Sensitivity</p></td><td char="." align="char"><p>35.2 (9.3)</p></td><td char="." align="char"><p>42.1 (10.3)</p></td></tr><tr><td align="left"><p>Sensation Avoiding</p></td><td char="." align="char"><p>66.6 (16.4)</p></td><td char="." align="char"><p>69.6 (17.6)</p></td></tr></tbody></table> </ephtml> </p> <p> <emph>ASD</emph> autism spectrum disorder; <emph>WS</emph> Williams syndrome; <emph>SD</emph> standard deviation; <emph>n</emph> number of participants</p> <hd id="AN0185099706-7">Severity Group Profile Analysis</hd> <p>The severity profiles of the autistic and WS groups are shown in Table 3. A chi-square analysis was applied to each SP quadrant. We did not find any significant group differences for Low Registration (χ<sups>2</sups>(<reflink idref="bib2" id="ref37">2</reflink>) = 0.51, <emph>p</emph> = 0.78), Sensation Seeking (χ<sups>2</sups>(<reflink idref="bib2" id="ref38">2</reflink>) = 2.27, <emph>p</emph> = 0.32), and Sensation Avoiding (χ<sups>2</sups>(<reflink idref="bib2" id="ref39">2</reflink>) = 0.51, <emph>p</emph> = 0.78). However, there was a significant group difference in Sensory Sensitivity (χ<sups>2</sups>(<reflink idref="bib2" id="ref40">2</reflink>) = 12.97, <emph>p</emph> < 0.01), which suggests that the proportion of severe cases in the WS group was significantly higher than that in the autistic group.</p> <p>Table 3 Severity profiles of each group on the sensory profile, with number of individuals (percentage) in each category</p> <p> <ephtml> <table frame="hsides" rules="groups"><thead><tr><th align="left"><p>Variable</p></th><th align="left"><p>Category</p></th><th align="left"><p>ASD</p><p>(<italic>n</italic> = 39)</p><p><italic>n</italic> (%)</p></th><th align="left"><p>WS</p><p>(<italic>n</italic> = 60)</p><p><italic>n</italic> (%)</p></th></tr></thead><tbody><tr><td align="left"><p>Low Registration</p></td><td align="left" /><td align="left" /><td align="left" /></tr><tr><td align="left" /><td align="left"><p>Similar to others</p></td><td align="left"><p>6 (15.3)</p></td><td align="left"><p>6 (10.0)</p></td></tr><tr><td align="left" /><td align="left"><p>More than others</p></td><td align="left"><p>22 (56.4)</p></td><td align="left"><p>20 (33.3)</p></td></tr><tr><td align="left" /><td align="left"><p>Much more than others</p></td><td align="left"><p>11 (28.2)</p></td><td align="left"><p>34 (56.7)</p></td></tr><tr><td align="left"><p>Chi-square</p></td><td align="left" /><td align="left" colspan="2"><p>0.51</p></td></tr><tr><td align="left"><p>Sensation Seeking</p></td><td align="left" /><td align="left" /><td align="left" /></tr><tr><td align="left" /><td align="left"><p>Similar to others</p></td><td align="left"><p>12 (30.8)</p></td><td align="left"><p>12 (20.0)</p></td></tr><tr><td align="left" /><td align="left"><p>More than others</p></td><td align="left"><p>17 (43.6)</p></td><td align="left"><p>35 (58.3)</p></td></tr><tr><td align="left" /><td align="left"><p>Much more than others</p></td><td align="left"><p>10 (25.6)</p></td><td align="left"><p>13 (21.7)</p></td></tr><tr><td align="left"><p>Chi-square</p></td><td align="left" /><td align="left" colspan="2"><p>2.27</p></td></tr><tr><td align="left"><p>Sensory Sensitivity</p></td><td align="left" /><td align="left" /><td align="left" /></tr><tr><td align="left" /><td align="left"><p>Similar to others</p></td><td align="left"><p>12 (30.8)</p></td><td align="left"><p>5 (8.3)</p></td></tr><tr><td align="left" /><td align="left"><p>More than others</p></td><td align="left"><p>19 (48.7)</p></td><td align="left"><p>26 (43.3)</p></td></tr><tr><td align="left" /><td align="left"><p>Much more than others</p></td><td align="left"><p>8 (20.5)</p></td><td align="left"><p>29 (48.3)</p></td></tr><tr><td align="left"><p>Chi-square</p></td><td align="left" /><td align="left" colspan="2"><p>12.97**</p></td></tr><tr><td align="left"><p>Sensation Avoiding</p></td><td align="left" /><td align="left" /><td align="left" /></tr><tr><td align="left" /><td align="left"><p>Similar to others</p></td><td align="left"><p>9 (23.0)</p></td><td align="left"><p>11 (18.3)</p></td></tr><tr><td align="left" /><td align="left"><p>More than others</p></td><td align="left"><p>18 (46.1)</p></td><td align="left"><p>27 (45.0)</p></td></tr><tr><td align="left" /><td align="left"><p>Much more than others</p></td><td align="left"><p>12 (30.8)</p></td><td align="left"><p>22 (36.7)</p></td></tr><tr><td align="left"><p>Chi-square</p></td><td align="left" /><td align="left" colspan="2"><p>0.51</p></td></tr></tbody></table> </ephtml> </p> <p> <emph>ASD</emph> autism spectrum disorder; <emph>WS</emph> Williams syndrome; <emph>n</emph> number of participants; **<emph>p</emph> < 0.01</p> <hd id="AN0185099706-8">SP Subscale Scores</hd> <p>The parameters of all models are reported in Table 4. For Low Registration, the main effect of the group was significant (χ<sups>2</sups>(<reflink idref="bib1" id="ref41">1</reflink>) = 30.7, <emph>p</emph> < 0.01); however, neither age (χ<sups>2</sups>(<reflink idref="bib1" id="ref42">1</reflink>) = 0.25, <emph>p</emph> = 0.62) nor interaction (χ<sups>2</sups>(<reflink idref="bib1" id="ref43">1</reflink>) = 2.76, <emph>p</emph> = 0.10) was significant. This suggests that the scores for the WS group were significantly higher than those for the autistic group (Fig. 2A).</p> <p>Graph: Fig. 2 Developmental trajectories of scores for the Sensory Profile (A: Low Registration, B: Sensation Seeking, C: Sensory Sensitivity, and D: Sensation Avoiding). Gray shaded area indicates the 95% confidence interval</p> <p>Table 4 Parameters for generalized linear model results in each subscore</p> <p> <ephtml> <table frame="hsides" rules="groups"><thead><tr><th align="left" rowspan="2" /><th align="left" rowspan="2"><p>Predictor</p></th><th align="left" rowspan="2"><p>Estimate</p></th><th align="left" rowspan="2"><p>SE</p></th><th align="left"><p>95% CI</p></th><th align="left" /></tr><tr><th align="left"><p>2.5%</p></th><th align="left"><p>97.5%</p></th></tr></thead><tbody><tr><td align="left"><p>Low Registration</p></td><td align="left" /><td align="left" /><td align="left" /><td align="left" /><td align="left" /></tr><tr><td align="left" /><td align="left"><p>(Intercept)</p></td><td align="left"><p>− 0.14</p></td><td align="left"><p>0.12</p></td><td align="left"><p>− 0.38</p></td><td align="left"><p>0.10</p></td></tr><tr><td align="left" /><td align="left"><p>group</p></td><td align="left"><p>0.030</p></td><td align="left"><p>0.15</p></td><td align="left"><p>− 0.26</p></td><td align="left"><p>0.32</p></td></tr><tr><td align="left" /><td align="left"><p>age</p></td><td align="left"><p>− 0.0013</p></td><td align="left"><p>0.0011</p></td><td align="left"><p>− 0.0034</p></td><td align="left"><p>0.00084</p></td></tr><tr><td align="left" /><td align="left"><p>group*age</p></td><td align="left"><p>0.0021</p></td><td align="left"><p>0.0013</p></td><td align="left"><p>− 0.00037</p></td><td align="left"><p>0.0046</p></td></tr><tr><td align="left"><p>Sensation Seeking</p></td><td align="left" /><td align="left" /><td align="left" /><td align="left" /><td align="left" /></tr><tr><td align="left" /><td align="left"><p>(Intercept)</p></td><td align="left"><p>0.19</p></td><td align="left"><p>0.097</p></td><td align="left"><p>0.0022</p></td><td align="left"><p>0.38</p></td></tr><tr><td align="left" /><td align="left"><p>group</p></td><td align="left"><p>− 0.50</p></td><td align="left"><p>0.12</p></td><td align="left"><p>− 0.73</p></td><td align="left"><p>− 0.28</p></td></tr><tr><td align="left" /><td align="left"><p>age</p></td><td align="left"><p>− 0.0063</p></td><td align="left"><p>0.00085</p></td><td align="left"><p>− 0.0080</p></td><td align="left"><p>− 0.0046</p></td></tr><tr><td align="left" /><td align="left"><p>group*age</p></td><td align="left"><p>0.0051</p></td><td align="left"><p>0.00099</p></td><td align="left"><p>0.0032</p></td><td align="left"><p>0.0070</p></td></tr><tr><td align="left"><p>Sensory Sensitivity</p></td><td align="left" /><td align="left" /><td align="left" /><td align="left" /><td align="left" /></tr><tr><td align="left" /><td align="left"><p>(Intercept)</p></td><td align="left"><p>-0.23</p></td><td align="left"><p>0.11</p></td><td align="left"><p>− 0.45</p></td><td align="left"><p>− 0.014</p></td></tr><tr><td align="left" /><td align="left"><p>group</p></td><td align="left"><p>-0.13</p></td><td align="left"><p>0.13</p></td><td align="left"><p>− 0.39</p></td><td align="left"><p>0.13</p></td></tr><tr><td align="left" /><td align="left"><p>age</p></td><td align="left"><p>-0.0035</p></td><td align="left"><p>0.00098</p></td><td align="left"><p>− 0.0054</p></td><td align="left"><p>− 0.0015</p></td></tr><tr><td align="left" /><td align="left"><p>group*age</p></td><td align="left"><p>0.0038</p></td><td align="left"><p>0.0011</p></td><td align="left"><p>0.0016</p></td><td align="left"><p>0.0060</p></td></tr><tr><td align="left"><p>Sensation Avoiding</p></td><td align="left" /><td align="left" /><td align="left" /><td align="left" /><td align="left" /></tr><tr><td align="left" /><td align="left"><p>(Intercept)</p></td><td align="left"><p>-0.16</p></td><td align="left"><p>0.089</p></td><td align="left"><p>− 0.33</p></td><td align="left"><p>0.019</p></td></tr><tr><td align="left" /><td align="left"><p>group</p></td><td align="left"><p>-0.21</p></td><td align="left"><p>0.11</p></td><td align="left"><p>− 0.42</p></td><td align="left"><p>− 0.00023</p></td></tr><tr><td align="left" /><td align="left"><p>age</p></td><td align="left"><p>-0.000072</p></td><td align="left"><p>0.00078</p></td><td align="left"><p>− 0.0016</p></td><td align="left"><p>0.0014</p></td></tr><tr><td align="left" /><td align="left"><p>group*age</p></td><td align="left"><p>0.0025</p></td><td align="left"><p>0.00090</p></td><td align="left"><p>0.00070</p></td><td align="left"><p>0.0042</p></td></tr></tbody></table> </ephtml> </p> <p> <emph>SE</emph> standard error; <emph>CI</emph> confidence interval * <emph>p</emph> < 0.05, ** <emph>p</emph> < 0.01, *** <emph>p</emph> < 0.001</p> <p>Regarding Sensation Seeking, the main effects of age (χ<sups>2</sups>(<reflink idref="bib1" id="ref44">1</reflink>) = 34.0, <emph>p</emph> < 0.01) and interaction (χ<sups>2</sups>(<reflink idref="bib1" id="ref45">1</reflink>) = 27.1, <emph>p</emph> < 0.01) were significant; however, the main effect of group (χ<sups>2</sups>(<reflink idref="bib1" id="ref46">1</reflink>) = 2.86, <emph>p</emph> = 0.09) was not significant. Therefore, we further explored the effect of age by dividing the scores into subgroups and found that scores in both groups decreased with development (autistic group: χ<sups>2</sups>(<reflink idref="bib1" id="ref47">1</reflink>) = 55.3, <emph>p</emph> < 0.01 and WS group: χ<sups>2</sups>(<reflink idref="bib1" id="ref48">1</reflink>) = 5.78, <emph>p</emph> = 0.01). The scores in the autistic group significantly decreased with development compared with those in the WS group (Fig. 2B).</p> <p>For the Sensory Sensitivity subscale, the main effects of group (χ<sups>2</sups>(<reflink idref="bib1" id="ref49">1</reflink>) = 48.7, <emph>p</emph> < 0.01) and interaction (χ<sups>2</sups>(<reflink idref="bib1" id="ref50">1</reflink>) = 11.5, <emph>p</emph> < 0.01) were significant, but not the main effect of age (χ<sups>2</sups>(<reflink idref="bib1" id="ref51">1</reflink>) = 1.42, <emph>p</emph> = 0.23). This suggests that scores in the WS group were significantly higher than those in the autistic group and that age can differently modulate scores across groups. Therefore, we further explored the effect of age by dividing the scores into subgroups (Fig. 2C) and found that scores in the autistic group decreased with development (χ<sups>2</sups>(<reflink idref="bib1" id="ref52">1</reflink>) = 12.5, <emph>p</emph> < 0.01), but not in the WS group (χ<sups>2</sups>(<reflink idref="bib1" id="ref53">1</reflink>) = 0.41, <emph>p</emph> = 0.52).</p> <p>For the Sensation Avoiding subscale, the main effects of age (χ<sups>2</sups>(<reflink idref="bib1" id="ref54">1</reflink>) = 20.1, <emph>p</emph> < 0.01) and interaction (χ<sups>2</sups>(<reflink idref="bib1" id="ref55">1</reflink>) = 7.50, <emph>p</emph> < 0.01) were significant but the group was not (χ<sups>2</sups>(<reflink idref="bib1" id="ref56">1</reflink>) = 3.82, <emph>p</emph> = 0.05). Therefore, we further explored the effect of age by dividing the scores into subgroups (Fig. 2D) and found that the scores increased with development in the WS group (χ<sups>2</sups>(<reflink idref="bib1" id="ref57">1</reflink>) = 27.5, <emph>p</emph> < 0.01) but not in the autistic group (χ<sups>2</sups>(<reflink idref="bib1" id="ref58">1</reflink>) = 0.008, <emph>p</emph> = 0.93).</p> <hd id="AN0185099706-9">Discussion</hd> <p>The current study aimed to explore the similarities and differences in sensory characteristics in autistic individuals and individuals with WS using caregivers' evaluations of sensory characteristics using the SP. Overall, we found similarities and differences in sensory profiles across the syndromes. Regarding the severity analysis, we found a significant group difference in Sensory Sensitivity, but not in Low Registration, Sensation Seeking, or Sensation Avoiding. Furthermore, we found that age could modulate the subscale scores differently across groups. For Sensation Seeking, the scores of both groups decreased with development, and the scores of Sensory Sensitivity decreased with age in the autistic group but not in the WS group. Additionally, Sensation Avoiding scores with development in the WS group increased but did not in the autistic group. Interestingly, no significant developmental changes were observed in the Low Registration quadrant.</p> <p>Our findings are partly consistent with previous cross-syndrome studies on sensory profiles in terms of no group differences in the Sensation Avoiding subscales (Glod et al., [<reflink idref="bib9" id="ref59">9</reflink>], [<reflink idref="bib11" id="ref60">11</reflink>]) and group differences in the Low Registration subscales (Glod et al., [<reflink idref="bib11" id="ref61">11</reflink>]). Although a significant difference was observed only in the scores of Low Registration in a previous study (Glod et al., [<reflink idref="bib11" id="ref62">11</reflink>]), we found significant group differences in Low Registration, Sensation Seeking, and Sensory Sensitivity scores. However, another study by Glod et al. reported no cross-syndrome differences (<emph>ps</emph> > 0.20) across subscores (Glod et al., [<reflink idref="bib9" id="ref63">9</reflink>]) (calculated the group difference based on the mean and standard deviations depicted in the paper). Our study was similar to the previous studies in terms of the ages of the participants (though the number of participants was different), yet we found differences in three categories from the SP. The sample size differences or other factors, such as cognitive factors, may have affected the results; however, currently we have no idea why group differences were observed in the current study. Therefore, further studies are needed to confirm whether there are consistent group differences in the SP scores.</p> <p>However, regarding severity analysis, our current results are consistent with previous studies in terms of the proportion of participants who were categorized as typical. For the autistic group, approximately 6.7–20.0% of autistic individuals fall within the typical range across subscales (Glod et al., [<reflink idref="bib11" id="ref64">11</reflink>]). In our study, 15.3–30.8% of autistic individuals fell in the typical range for SP subscale scores. Regarding WS, a previous study indicated that approximately 10% of individuals with WS fell within the typical range for sensory modulation (John & Mervis, [<reflink idref="bib15" id="ref65">15</reflink>]), whereas Glod and colleagues (Glod et al., [<reflink idref="bib11" id="ref66">11</reflink>]) showed that 7.7–15.4% of individuals with WS showed the typical sensory modulation. In our study, approximately 8.3–20.0% of participants with WS fell within the typical range for SP subscale scores. This suggests that the tendency for the severity level of the proportion of SP subscores in autistic individuals and individuals with WS seems concordant with previous studies.</p> <p>We further found that age affected each score differently across the groups. In the autistic group, Sensation Seeking scores increased with age, but Sensory Sensitivity scores decreased with age. No developmental changes were observed for Sensation Avoiding and Low Registration. The reduction in Sensation Seeking with age in the autistic group seems concordant with a previous study demonstrating that atypical visual, auditory, oral, and tactile processing changed over age groups, appearing to become similar to the control groups (Kern et al., [<reflink idref="bib17" id="ref67">17</reflink>]). In contrast with our findings, a recent large-sample study (<emph>N</emph> = 919) demonstrated that both Sensory Sensitivity and Sensation Avoiding frequency in the autistic group appeared to peak during middle childhood, around the age of 6–12 years. Further, it did not show a monotonic reduction during childhood but rather stabilized through adolescence (Lane et al., [<reflink idref="bib22" id="ref68">22</reflink>]). This might be because our sample size was relatively small compared to that of the study by Lane et al.; thus, our study might not be able to capture subtle developmental changes. This needs to be addressed in future studies.</p> <p>In the WS group, Sensation Seeking scores decreased with age, whereas Sensation Avoiding scores increased with age. Furthermore, no developmental changes were observed in Sensory Sensitivity. To the best of our knowledge, no studies have tested developmental changes in SP scores in individuals with WS. However, developmental changes in other traits for individuals with WS have been revealed, which show that the occurrence of verbal aggression and temper tantrums can decline with age, but not physical aggression before 19 years of age (Rice et al., [<reflink idref="bib26" id="ref69">26</reflink>]). The persistence of Sensory Sensitivity across ages in WS seems to be concordant with a previous study that demonstrated that hyperacusis can be found in both children and adults with WS (Blomberg et al., [<reflink idref="bib4" id="ref70">4</reflink>]).</p> <p>In summary, there were no group differences between the SP subscale scores of Sensation Seeking and Sensation Avoiding; however, there was a difference between groups in terms of Sensory Sensitivity and Low Registration. Development has been found to impact the differences in the subscale scores. However, further studies are required to fully elucidate the neural mechanisms underlying the developmental changes in sensory properties.</p> <p>This study had several limitations. First, although we found group differences in developmental changes of each subscale score in the SP, we could not fully capture the heterogeneity of SP across syndromes. Second, although we have revealed the differential developmental changes in SP scores across syndromes, the SP was evaluated by caregivers. It is possible that compensatory mechanisms during development may lead to different Sensory Sensitivity assessments by caregivers. Therefore, further studies will need to adopt objective measures to evaluate sensory characteristics. Third, although previous sensory profile studies have demonstrated that cognitive profile do not influence the sensory profile, and our preliminary analysis also demonstrated no significant correlation between Raven Colored Matrix scores and SP scores, in order to better understand the effect of cognitive profile on the sensory profiles, further studies are required. Finally, our study focused only on the Japanese population, and the scores might be susceptible to cultural norms. Therefore, cross-cultural studies may better capture factors.</p> <p>In conclusion, although previous studies demonstrated developmental changes in sensory profiles in typical adults (Pohl et al., [<reflink idref="bib25" id="ref71">25</reflink>]) or autistic individuals (Kern et al., [<reflink idref="bib17" id="ref72">17</reflink>]; Lane et al., [<reflink idref="bib22" id="ref73">22</reflink>]), to the best of our knowledge, this is the first study to reveal developmental changes in sensory characteristics using the SP in both autistic individuals and individuals with WS. As the number of participants was limited, more studies are needed to encompass a wide range of autistic individuals and individuals with WS to replicate the developmental changes in SP scores.</p> <hd id="AN0185099706-10">Acknowledgements</hd> <p>We are grateful to all caregivers for their participation. Further, we thank C. Matsui for helping to recruit participants and M. Sasaki for assistance in data collection.</p> <hd id="AN0185099706-11">Author Contributions</hd> <p>Masahiro Hirai and RK contributed to conceptualization. Masahiro Hirai and AI contributed to formal analysis. Masahiro Hirai contributed to Writing - original draft preparation. Masahiro Hirai, AI, KA, YH, KM, Toshihiro Kato and RK contributed to data curation. Masahiro Hirai, AI, KA, YH, KM, Toshihiro Kato and RK contributed to investigation. Masahiro Hirai, AI, Takeo Kato, TI, KA, YH, KM, TA, SO, Toshihiro Kato, TH, Masatoshi Hagiwara, TY, KT and RK contributed to writing - review and editing. Takeo Kato, TI, KM, TA, SO, Toshihiro Kato, TH, Masatoshi Hagiwara, TY, KT and RK contributed to resources. Masahiro Hirai and RK contributed to funding acquisition. Masahiro Hirai and RK contributed to project administration.</p> <hd id="AN0185099706-12">Funding</hd> <p>This study was funded by the Pfizer Health Research Foundation, Japan; Japan Society for the Promotion of Science (JSPS) KAKENHI (Grant Numbers: 18H01103, 21K18554); and a Grant-in-Aid for Scientific Research on Innovative Areas (Grant Number: 15H01585) awarded to MH. RK received grants from the Yuumi Memorial Foundation for Home Health, the SENSHIN Medical Research Foundation, and JSPS KAKENHI (Grant Numbers: 19K08251, 21H05326, and 22H00986, respectively). The grant-giving institutions played no role in the design, collection, analysis, or interpretation of data or the writing of the manuscript.</p> <hd id="AN0185099706-13">Data Availability</hd> <p>Data are available upon request from the corresponding author.</p> <hd id="AN0185099706-14">Declarations</hd> <p></p> <hd id="AN0185099706-15">Conflict of interest</hd> <p>The authors declare no conflict of interest relevant to the content of this article.</p> <hd id="AN0185099706-16">Ethical approval</hd> <p>Research involving human participants: This study was performed in line with the principles of the Declaration of Helsinki. 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  Data: Comparison of the Sensory Profile among Autistic Individuals and Individuals with Williams Syndrome
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  Data: <searchLink fieldCode="AR" term="%22Masahiro+Hirai%22">Masahiro Hirai</searchLink> (ORCID <externalLink term="http://orcid.org/0000-0003-2298-5632">0000-0003-2298-5632</externalLink>)<br /><searchLink fieldCode="AR" term="%22Ayaka+Ikeda%22">Ayaka Ikeda</searchLink><br /><searchLink fieldCode="AR" term="%22Takeo+Kato%22">Takeo Kato</searchLink><br /><searchLink fieldCode="AR" term="%22Takahiro+Ikeda%22">Takahiro Ikeda</searchLink><br /><searchLink fieldCode="AR" term="%22Kosuke+Asada%22">Kosuke Asada</searchLink><br /><searchLink fieldCode="AR" term="%22Yoko+Hakuno%22">Yoko Hakuno</searchLink><br /><searchLink fieldCode="AR" term="%22Kanae+Matsushima%22">Kanae Matsushima</searchLink><br /><searchLink fieldCode="AR" term="%22Tomonari+Awaya%22">Tomonari Awaya</searchLink><br /><searchLink fieldCode="AR" term="%22Shin+Okazaki%22">Shin Okazaki</searchLink><br /><searchLink fieldCode="AR" term="%22Toshihiro+Kato%22">Toshihiro Kato</searchLink><br /><searchLink fieldCode="AR" term="%22Toshio+Heike%22">Toshio Heike</searchLink><br /><searchLink fieldCode="AR" term="%22Masatoshi+Hagiwara%22">Masatoshi Hagiwara</searchLink><br /><searchLink fieldCode="AR" term="%22Takanori+Yamagata%22">Takanori Yamagata</searchLink><br /><searchLink fieldCode="AR" term="%22Kiyotaka+Tomiwa%22">Kiyotaka Tomiwa</searchLink><br /><searchLink fieldCode="AR" term="%22Ryo+Kimura%22">Ryo Kimura</searchLink>
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  Data: <searchLink fieldCode="SO" term="%22Journal+of+Autism+and+Developmental+Disorders%22"><i>Journal of Autism and Developmental Disorders</i></searchLink>. 2025 55(6):2092-2099.
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  Data: Springer. Available from: Springer Nature. One New York Plaza, Suite 4600, New York, NY 10004. Tel: 800-777-4643; Tel: 212-460-1500; Fax: 212-460-1700; e-mail: customerservice@springernature.com; Web site: https://link.springer.com/
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  Data: Y
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  Data: 8
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  Data: 2025
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  Label: Document Type
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  Data: Journal Articles<br />Reports - Research
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  Label: Descriptors
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  Data: <searchLink fieldCode="DE" term="%22Sensory+Experience%22">Sensory Experience</searchLink><br /><searchLink fieldCode="DE" term="%22Profiles%22">Profiles</searchLink><br /><searchLink fieldCode="DE" term="%22Autism+Spectrum+Disorders%22">Autism Spectrum Disorders</searchLink><br /><searchLink fieldCode="DE" term="%22Genetic+Disorders%22">Genetic Disorders</searchLink><br /><searchLink fieldCode="DE" term="%22Severity+%28of+Disability%29%22">Severity (of Disability)</searchLink><br /><searchLink fieldCode="DE" term="%22Symptoms+%28Individual+Disorders%29%22">Symptoms (Individual Disorders)</searchLink><br /><searchLink fieldCode="DE" term="%22Personality+Traits%22">Personality Traits</searchLink><br /><searchLink fieldCode="DE" term="%22Scores%22">Scores</searchLink>
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  Label: DOI
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  Data: 10.1007/s10803-023-06205-1
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  Data: 0162-3257<br />1573-3432
– Name: Abstract
  Label: Abstract
  Group: Ab
  Data: Purpose: With the current study, we aimed to reveal the similarities and differences in sensory profiles between Williams syndrome (WS) and autism spectrum disorder. Methods: Using the sensory profile questionnaire completed by the caregivers, we analyzed the WS (n = 60, 3.4-19.8 years) and autistic (n = 39, 4.2-14.0 years) groups. Results: The Severity Analysis revealed a significant group difference in Sensory Sensitivity but not in Low Registration, Sensation Seeking, and Sensation Avoiding subscales. Age can modulate the subscale scores differently across groups. For Sensation Seeking, the scores of both groups decreased with development. However, the scores of Sensory Sensitivity decreased with age in the autistic group but not in the WS group. Sensation Avoiding scores increased with development in the WS group but not in the autistic group. No significant developmental changes were observed in Low Registration. Conclusion: This study highlights the cross-syndrome similarities and differences in sensory profiles and developmental changes in autistic individuals and individuals with WS.
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  Data: 2025
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  Data: EJ1470827
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        Type: general
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      – SubjectFull: Autism Spectrum Disorders
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