Meta Analisis
Meta Analisis
Meta Analisis
https://doi.org/10.1007/s10803-019-04180-0
ORIGINAL PAPER
Abstract
This meta-analysis updated evidence regarding sensory over-responsivity (SOR), under-responsivity (SUR) and seeking
symptoms in individuals with autism spectrum disorders (ASDs) relative to typical controls and those with other conditions.
Fifty-five questionnaire studies included 4606 individuals with ASD. Moderators tested were age, IQ, male ratio, matching
group, and self-report. Compared to typical controls, effect size was large and significant for SOR, SUR, and Seeking but
heterogeneous. For Seeking, age, IQ and self-report were significant moderators. Compared with developmental disorders
(DDs) groups, effect size was significantly positive for SOR and Seeking; whereas compared with other clinical groups,
only SOR was significant. These findings highlight the core nature of sensory symptoms in ASD and particularly SOR.
Explanatory factors are yet to be revealed.
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Fig. 1 The number of articles between 1963 and 2017 with autism and sensory (filtered by humans) in PubMed
displayed atypical patterns of SOR, SUR, and Seeking. Of bridging inter-disciplinary methodologies towards sensory
the ASD–TD comparisons, mean effect size was high for all symptoms in ASD at the assessment and neurobiology lev-
types of sensory scores (d = 0.82–2.14), but larger for SUR. els. This includes a literature review by Hazen et al. (2014),
SOR and Seeking symptoms were highest in the 6–9 years a review integrating symptom and neural literature in this
old group and decreased thereafter. The three sensory pat- area (Schauder and Bennetto 2016), and a scoping review
terns showed different relations relative to severity of ASD about measures of sensory symptoms in adolescents and
and relative to whether they were compared to a mentally adults (DuBois et al. 2017). None of the recent reviews used
versus chronologically age matched control group support- a meta-analysis design.
ing their differentiation. The distinct relations of these three There has been increasing acknowledgement that these
sensory patterns with developmental, psychological, and sensory symptoms are neurobiologically grounded since
maturational indices in ASD was also supported in more our 2009 publication, with individuals with ASD showing
recent reviews (Glod et al. 2015; Schauder and Bennetto different neural processing compared to peers (Cascio et al.
2016). While the 2009 review contributed to our understand- 2012; Chang et al. 2012; Green et al. 2015, 2016b; Hazen
ing of sensory features, findings were limited by the small et al. 2014; Madsen et al. 2014; Marco et al. 2011). Review
number of available studies, with only four studies including of fMRI research points to abnormalities in the activation
a DD comparison group. patterns across sensory brain areas, and MEG evidence
Since our 2009 paper, there has been increasing recogni- indicates the presence of maturational and lateralization
tion of the prominence of sensory features of ASD. In 2013, atypicalities in sensory processing in ASD (Schauder and
DSM-5 added both sensory hypo-reactivity (referred to as Bennetto 2016). Nonetheless, the correspondence between
SUR hereafter) and sensory hyper-reactivity (referred to as observable sensory symptoms and neurobiological atypicali-
SOR) to the diagnostic criteria for ASD (American Psy- ties is yet to be determined.
chiatric Association 2013). Sensation seeking was already Evidence attests to the association of sensory symptoms
captured in DSM-IV within the rubric of unusual sensory with developmental and other facets of the disorder, as well
interests. Since the DSM-5 publication, research on sensory as the likelihood of co-morbid conditions. Sensory symp-
features in ASD has skyrocketed, as shown by a simple Pub- toms are associated with compromised adaptive perfor-
Med search (October 2018) using the terms sensory AND mance, participation (Dellapiazza et al. 2018; Tomchek et al.
autism, limited to humans (see Fig. 1). Several systematic 2015) and performance of activities of daily living (Dunn
reviews about sensory features and ASD have been pub- et al. 2016; Ismael et al. 2018), including eating (Cermak
lished recently, mostly highlighting correlates of sensory et al. 2010; Mazurek et al. 2013; Zobel-Lachiusa et al. 2015)
symptoms such as adaptive behavior and attention impair- and sleeping (Mazurek and Petroski 2015). Sensory symp-
ments (Dellapiazza et al. 2018), psychological symptoms toms have been associated with greater severity of restricted
(i.e., emotional, affective and behavioral symptoms; Glod and repetitive behaviors (Chen et al. 2009; Gabriels et al.
et al. 2015) and participation in daily functioning (Ismael 2008), and reduced social functioning (Glod et al. 2015;
et al. 2018). Additional recent literature reviews focused on Hilton et al. 2010). They are also associated with increased
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affective symptoms (Ben-Sasson et al. 2008; Glod et al. decrease with age due to neuro-hormonal changes and/or
2015), particularly anxiety (Green et al. 2012; Lane et al. the establishment of coping strategies. The inconsistency
2012). In addition, sensory symptoms have been shown to in findings calls for testing the moderating effects of age
interplay with communication and language deficits (Glod in explaining differences between groups.
et al. 2015; Tomchek et al. 2015) and attention competen- Gender may also play a moderating role as recent
cies (Dellapiazza et al. 2018; Glod et al. 2015). Further, research suggests gender differences in severity of repetitive
sensory symptoms negatively impact family life (Bagby and restricted behaviors (Antezana et al. 2019; Van Wijn-
et al. 2012; Kirby et al. 2015). It is possible that the inherent gaarden-Cremers et al. 2014). In a meta-analysis, females
involvement of sensory processing in every interaction and with ASD over the age of 6 years compared to males showed
learning process during development explains its pervasive a lower severity in the repetitive and restricted domain (Van
impact upon functional impairment and well-being. Given Wijngaarden-Cremers et al. 2014). Other evidence in ado-
that adaptive living skills contribute to a person’s ability lescents with ASD points to an increased likelihood of spe-
to participate successfully in the environment, it is critical cific types of repetitive and restricted behaviors in males and
to fully understand sensory processing and make it a high others in females. Given the DSM-5 inclusion of sensory
priority for research and practice. symptoms in ASD diagnotic criteria, we tested for gender
Sensory symptoms and specifically SOR also appear early differences in sensory symptoms.
in other types of developmental and psychopathological con- In a recent scoping review focused on 66 studies of
ditions such as ADHD (Cheung and Siu 2009), intellectual sensory processing of adolescents and adults with ASD,
disability (Gal 2006), Fragile X (Baranek et al. 2008), anxi- DuBois et al. (2017) reported that proxy reporting was the
ety disorders (Conelea et al. 2014), and obsessive compul- most prevalent (78.7%) method of measurement. The Ado-
sive disorder (OCD; Lewin et al. 2015). This raises questions lescent/Adult Sensory Profile was the most frequently used
regarding their distinct manifestation in ASD, both in mag- measure (71.1%) and was used as self-report and adapted
nitude and nature. Aside from their unique co-occurrence for proxy report. While observation tools and physiological
with social–communication symptoms in ASD, it is unclear measurement were included in a few of the recent studies,
how much these symptoms are related to developmental overall there was too much variability in the conceptualiza-
level. There is evidence associating higher mental capac- tion of the target behaviors for the quantitative demands of
ity with fewer sensory symptoms in young children with a meta-analysis. Thus, we decided to continue limiting the
ASD (Baranek et al. 2006) as well as adults (Kargas et al. current meta-analysis to questionnaire-based methodology.
2015). In our 2009 review there was not sufficient IQ data To summarize, 10 years have passed since the publication
to test this moderator across studies and sensory patterns. of the first meta-analysis examining the magnitude of sen-
We hypothesized that there would be fewer significant and sory symptoms in individuals with ASD. During this time,
smaller effect sizes between ASD and other clinical groups sensory symptoms have become part of the diagnostic crite-
compared to TD groups, as well as moderating effects ria for ASD and the number of new studies including clinical
related to developmental level. groups with and without ASD has increased tremendously
There is conflicting evidence regarding the course of (DuBois et al. 2017; Glod et al. 2015; Schauder and Ben-
sensory symptoms in ASD throughout the life span. Age netto 2016). Further, the new DSM-5 ASD criteria groups
related changes in sensory symptoms are important indica- all ASD subtypes together potentially impacting the nature
tors for determining their role in early identification, matu- of samples studied. As such, it is critical to examine current
rational mechanisms and for allocating services addressing research and update the meta-analysis to understand the fac-
sensory features. Some studies found a reduction in sen- tors that moderate sensory processing in individuals with
sory symptoms in adolescents and adults compared with ASD more fully. Some of our previous moderators were no
children with ASD (Kern et al. 2006; Leekam et al. 2007), longer relevant such as percent of autism diagnosis in sam-
some showed an increase in symptoms with age (Talay- ple (e.g., autism category omitted from DSM-5) and some
Ongan and Woods 2000), while others showed stability became relevant with more studies reporting them (e.g.,
throughout childhood (McCormick et al. 2016; Rogers male ratio, IQ). The objectives of the meta-analysis were:
et al. 2003). In our previous meta-analysis, we found a (1) to characterize the magnitude of sensory symptoms (i.e.,
non-linear course of symptom development, the 6–9-year- SOR, SUR, and Seeking) in ASD compared with typical and
old age group presented with greater SOR and Seeking clinical samples and (2) to identify the contribution of demo-
effect sizes compared to younger and older age groups. graphic and methodological moderators to the variability in
Note that in 2009 there were many more samples with findings across studies. We hypothesized that there would
mixed age groups (e.g., sample of children and adults) to be fewer significant and smaller effect sizes between ASD
rely upon, hence the recent evidence is of value for exam- and other clinical groups compared to effect sizes relative
ining distinct age related changes. Sensory symptoms may
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Methods
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Table 1 (continued)
Author Group N CA mean/median CA range years IQ (SD) Levelix % Boys Sensory questionnaire
(SD) years
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Table 1 (continued)
Author Group N CA mean/median CA range years IQ (SD) Levelix % Boys Sensory questionnaire
(SD) years
Chistol et al. (2018) ASD 53 6.6 (2.1) 3–11 85.7 (22)10,6 83 SPa
iFor these studies raw data was sent by authors for sensory scores and/or demographic information
iiThe reason for matching data is that it reflects the whole sample, no specific data for the separate groups was given for these variables
(only in sensory variables)
iiiDemographics are based on a larger sample size than the sensory data available
ivOne subgroup or one wave was selected for the meta-analysis based on sensory scores available or age group with the least studies
vIQ = DQ
ixLevel of functioning 1 = high functioning and 2 = low functioning. This was based on description of inclusion criteria and/or self-report meas-
ure applied and not on mean IQ scores
1
Mullen Scales of Early Learning (1995), 2Bayley Scales of Infant Development-Second Edition (1993), 3Wechsler Adult Intelligence Scale-
Third Edition (1997), 4Wechsler Intelligence Scale for Children-Third Edition (1991), 5Wechsler Intelligence Scale for Children-Fourth Edition
(2003), 6Differential Ability Scales-II (Elliot 2007), 7Leiter International Performance Scale-Revised (Roid and Miller 1997), 8Wechsler Pre-
school and Primary Scale of Intelligence-Third Edition (2002), 9Wechsler Preschool and Primary Scale of Intelligence-Revised (1989), 10Vine-
land Adaptive Behavior Scales (Sparrow et al. 1984), 11Wechsler Abbreviated Scale of Intelligence (2011), 12British Picture Vocabulary Scale
(Dunn et al. 1997), 13Preschool Language Scale. 4 (Zimmerman et al. 2002), 14Wechsler Intelligence Scale for Children-unspecified, 15Tanaka–
Binet Intelligence Scale-V (Matsubara et al. 2005), 16Kyoto Scale of Psychological Development (Ikuzawa et al. 2001), 17The Raven’s Standard
or Coloured progressive matrices (1990a, b), 18Parent Estimated Developmental Age (PEDA; Ausderau et al. 2014), 19Kaufman’s Brief Intel-
ligence Test-Second edition (Kaufman and Kaufman 2004), 20Stanford–Binet intelligence scales. 5 (Roid 2003)
a
Sensory Profile (Dunn and Westman 1995; Dunn 2002)
b
Sensory Sensitivity Questionnaire-Revised (Talay-Ongan and Wood 2000)
c
Infant/Toddler Sensory Profile (Dunn 2002)
d
Short Sensory Profile (McIntosh et al. 1999)
e
Sensory Experiences Questionnaire: Version 1 (Baranek et al. 2006)
f
Sensory Sensitivity Survey (60 items from The Sensory Profile, Dunn 1999; Liss et al. 1998)
g
Chinese Sensory Profile (Tseng and Cheng 2008)
h
Adolescent/Adult Sensory Profile (Brown and Dunn 2002)
i
Japanese Sensory Inventory-Revised (Ota et al. 2002)
j
The Sensory Processing Scales Inventory: Sensory Over-Responsivity subscale (Miller and Schoen 2012)
k
Sensory Experiences Questionnaire: Version 3.0 (Baranek 2009, Unpublished manuscript)
l
Sensory Perception Quotient (Tavassoli et al. 2014a, b)
m
Sensory Reactivity in Autism Spectrum (Elwin et al. 2016)
n
Composite based on some of the Short Sensory Profile factors and the Sensory Over-Responsivity subscale
o
The Sensory profile-2 (Dunn 2014)
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Final Set of Studies the SUR effect size. In 2009, we did not include this score as
it has limited items which are not cross-modality. However,
The meta-analysis included 55 studies, 14 from the 2009 in the current review the additional studies using the SSP
meta-analysis and 41 new studies. Table 1 presents the enabled us to test the moderating effect of using the SSP
detailed features of the studies and their samples. (most popular measure) upon the SUR effect size results.
In SSP studies (k = 4) for which we had the raw data, SOR,
Coding Process SUR, and Seeking scores were computed directly to include
a wider range of items under these patterns (personal com-
The 2009 meta-analysis coding guidelines were adapted to munication Dunn August 2002).
meet changes in diagnostic procedures and the availabil- For studies using any version of the Sensory Profile that
ity of more descriptive information in papers (e.g., ratio reported quadrant scores in addition to factor scores or
of Caucasian ethnicity). Two independent researchers (the instead of factor scores, we relied on the quadrant scores.
first and fourth authors) reviewed the coding guideline for This was because they are based on a wider range of items.
extracting background information and sensory data from For these studies, the SOR score was computed as a sum of
the publications, as well as coding examples from the 2009 Avoiding and Sensitivity quadrants following the manual
coding. Both researchers coded the same four papers and guideline (Dunn 2002).
reached 99.95% agreement. The researchers further refined
the guidelines and added variable categories as needed (e.g., Effect Size Computation
new types of sensory questionnaires not studied in 2009).
The papers were coded for: group size, publication year, type Cohen’s d effect size was computed i.e. the standardized
of comparison group, type of group matching, mean age, mean difference using the pooled standard deviation of the
age group, mental level, male ratio, Caucasian ratio, type of ASD and control groups with a bias correction (Hedges and
ASD, ASD diagnostic method and professional diagnosis, Olkin 1985). In two studies (Dunn 2002; Tomcheck and
recruitment setting, type of mental test, type of sensory test, Dunn 2007), only effect sizes were reported and not means
self-report, version of Sensory Profile, Short Sensory Profile and SDs. Therefore, we relied on the effect sizes reported.
(SSP), sensory quadrants reported, direction of the sensory If an eta-squared was the type of effect size reported, it was
scores (+ 1 for studies in which higher scores indicated transformed into a d value (Cooper 2015). When several
higher severity and − 1 vice versa). The remaining papers comparisons were reported leading to several effect sizes of
were coded separately by each of the researchers. Periodic the same study, we averaged them. Since the correlations of
review of coding (for approximately 30% of papers) and dis- these effect sizes were not reported, we estimated the effect
cussions of outstanding examples in studies were regularly size variance by the average of their estimated variances.
conducted. The justification for this is that all effect sizes should have
similar asymptotic variance (i.e. 1/n1 + 1/n2) and they may
be highly correlated.
Data Analysis The following variables were tested as moderators; sig-
nificant moderators were reported in results:
Comparison of Sensory Scores Across Studies
(1) Demographic variables for ASD group mean age
For each study (and group) an effect size was computed for in years, age group (five groups), ratio of males, IQ
the three sensory patterns, SOR, SUR and/or Seeking. As scores, level of functioning (high vs. low), type of
such, studies that reported sensory pattern subcomponents group matching (only chronological age vs. only men-
only (e.g., for SOR they reported sensory avoiding and sen- tal age).
sory sensitivity scores) required the computation of one (2) Methodological variables publication year, Sensory
effect size for each pattern. See “Appendix” for a concep- Profile based measure, SSP used, self-report.
tual comparison of scores between measures. This “Appen-
dix” details the types of scores for which one effect size Some moderators overlap conceptually. They were coded
was computed per pattern. In addition, when mean sensory to enable quantification for studies that had one overlapping
scores were reported for each ASD subgroup (e.g., autism moderator and not the other. For example, the categorical
and PDD-NOS) as opposed to the total ASD sample, com- moderator of level of functioning overlaps with the continu-
putation was weighted by the size of each subgroup. ous IQ variable. Studies that did not report IQ scores for the
The SSP does not provide a distinct Seeking score; hence, ASD group but stated that the ASD group included only
studies using this measure do not have a Seeking effect size. a certain level of functioning could be coded for the cat-
The SSP has a low energy score that was used to compute egorical variable. Self-report measures can be only applied
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in studies of adolescents/adults who are cognitively able to For 48 studies for which chronological age was reported,
complete them. As such, this variable also captures mental the average age of the ASD group was 10.85 years
level and is not solely an indication of biases of caregiver (SD = 9.26), of the typical group 10.9 (SD = 9.67), of the
versus self-report. The moderation of self-report was tested DD group 6.59 (SD = 3.98) and of the mixed clinical group
only among studies with individuals for whom self-report 8.90 (SD = 0.91). ASD age group data was available for all
measures were available (≥ 9 years). 55 studies: 10.9% (6 of 55) of the studies included children
Each study was included once in each of the nine models. under the age of 3 years, 12.7% (7 of 55) included children
Therefore, if a study had two DD groups, the larger was 3–6 years, 40.0% (22 of 55) included children 6–9 years old,
included in the meta-analysis. The only exception was in 21.8% (12 of 55) were 9–18, and 14.3% (8 of 55) were above
the case of testing the moderating effect of the type of group 18 years of age. For 26 of 55 studies with IQ information
matching on ASD and TD effect sizes. Here, an additional for the ASD group, the mean IQ for the ASD group was
group of 99 TD children from the same study (Ben-Sasson 83.95 (SD = 18.73). For the 15 of 47 studies with IQ for the
et al. 2007) matched on mental age was included, given the TD group, the mean was 110 (SD = 5.24), for 9 of 11 stud-
few mental age-matched studies. All effect size calculations ies with IQ data for the DD group, the average was 65.77
and moderator testing were done with the “metafor” package (SD = 8.39). For the two of seven studies with IQ data for
(Viechtbauer 2010) in the software (R Core Team 2018). the clinical group, the mean was 73.65 (SD = 40.8). Most
studies included higher functioning individuals with ASD
Comparison of Moderators Between Current (70.6% 27 of 34 studies based on study inclusion criteria or
and Previous Meta‑analysis mean IQ). The ASD samples from the 14 studies analyzed
in 2009 versus the 41 new ones, were younger (95.8 vs.
Changes in diagnostic practices (e.g., DSM-5 subtyping does 139.5 months respectively) and had lower average IQ scores
not include autism separately hence coding for percentage (66.97 vs. 83.95 respectively).
of autism diagnosis was no longer relevant) as well as more Forty-nine studies (of 55 studies, 89.1%) reported the
detailed descriptive information reported in research (e.g., gender of participants. The percentage of males in the ASD
with regards to raw IQ values) offered an opportunity for fine group ranged from 36.6 to 100%, with an average of 81.4%
grained coding which in 2009 was not possible. The only as compared to 69.6% in typical group, 75.3% in DD group
identical moderator tested in both meta analyses was type and 78.7% in the clinical group. The study participants were
of matching, i.e., chronological versus mental age matching. predominantly Caucasian (for k = 20, M = 72.6%). Three
Similar moderators but defined differently between meta- studies with large Asian samples appear in the current meta-
analyses were age group which we were able to expand to a analysis, representing a population that was not previously
wider age range in the current analysis, percentage of autism studied (Cheung and Siu 2009; Matsushima and Kato 2013;
diagnosis in 2009 and level of functioning in this analysis. Tseng and Cheng 2008; n = 189 with ASD compared to none
New moderators available only in the current analysis were: in the 2009 meta-analysis), and a Saudi Arabian sample not
IQ score, male ratio, Sensory Profile based measure, SSP previously represented (Al-Heizan et al. 2015).
used, self-report. Fifty-five studies reported the type of matching between
groups. For studies compared to a typical group, most (k = 33
of 47) matched on chronological age, five on mental age,
Results and nine on both. For studies with a DD comparison group,
most (k = 8 of 11) matched on mental and chronological age,
Characteristics of Studies two matched only on chronological age and three only on
mental age. Of studies with a different clinical comparison
The 55 studies included in the meta-analysis included 4606 group, six matched only on chronological age and one on
individuals with ASD, 5508 TD (k = 47 [with k denoting the both chronological and mental age.
number of studies and n the number of participants], 376
with DD [k = 11]), and 399 with other clinical conditions ASD Diagnostic Procedures
(k = 7, 5 ADHD comparison groups, 1 Learning Difficul-
ties, and 1 SPD). Nine of the 47 studies, had another com- The most common diagnostic method reported was meeting
parison group besides the typical group. For five studies this DSM or ICD criteria (k = 30 of 50 that reported diagnostic
was a DD group and for four another clinical group. Studies method) with or without another method. Other diagnostic
were published from 1998 to June 2018 (2009 studies years criteria were ADOS or ADOS-G (k = 21), having a current or
of publication were 1998–2007), with 21 of the 55 studies previous clinical diagnosis of ASD (k = 18), ADI or ADI-R
published after DSM-5 publication in 2013. See Table 1 for (k = 12), and CARS (k = 4). A total of 24 studies required
features of the individual studies. meeting at least two diagnostic methods. Nine studies of
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the 21 which required meeting ADOS cutoff required also moderators tested did not significantly explain the heteroge-
meeting ADI cutoff for ASD. Five of these nine studies also neity in effect sizes for SOR or SUR models.
required meeting DSM/ICD criteria. In one study, parents For Seeking effect sizes, several moderators signifi-
completed the sensory questionnaire prior to receiving the cantly explained differences between ASD and TD groups.
child’s diagnosis (Wiggins et al. 2009). Effect sizes significantly differed between age groups
Most studies described the professional required to diag- (Qw[df = 3] = 10.51, p = .02), Fig. 6 shows that effect size
nose ASD (k = 36 of 55). Of these, 26 professionals who increased up to Group 3, ages 6–9 years and decreased
diagnosed were an MD and/or psychologist (k = 26 of the 36 thereafter (see Fig. 6). IQ significantly moderated Seeking
studies), and 10 did not specify the profession required for (Qm[df = 1] = 4.71, estimate = − 0.04, p = 0.03), such that the
diagnosing but stated that they were trained professionals. lower the IQ, the larger the effect size. Self-report was also a
significant moderator (Qm[df = 1] = 9.16, estimate = − 1.98,
p = .003). Those who self-reported, had smaller effect sizes
Distribution of Questionnaires Used
than those whose caregivers reported for them.
Forty-one studies (of 55, 74.55%) used some version of the
Comparison of ASD and DD Groups
Sensory Profile or an adapted version of the Sensory Profile,
of which 16 (of 41) were the SSP, and for 19 (of 41) we had
Average effect size for the comparison of ASD and
quadrant scores (i.e., seeking, low registration, avoidance,
DD groups was significant for SOR 0.54 (k = 11, 95%
sensitivity). Three of the 55 studies used the Sensory Expe-
CI = [0.30–0.78]; see Table 2). Significant positive effect
rience Questionnaire, three of 55 used different versions of
sizes were found in six of the 11 studies (54%), ranging from
the SensOR scale (known today as SP3D). The remainder of
0.58 to 1.06. Average Seeking effect size was significant and
measures appeared once. Nine studies relied on self-report
positive but low, 0.49 (95% CI = [0.25–0.73]), with three of
(of 16 studies that could potentially use self-report based on
the six studies showing significant positive effect sizes.
their mean age of at least 11 years).
Average SUR effect size between ASD and DD groups was
not significant 0.22 (k = 11, 95% CI = [− 0.14 to 0.58]).
Comparison of ASD and Typical Groups Further testing showed significant heterogeneity for
SOR (Qw[df = 10] = 28.96, p = .0013) but not for Seek-
Average effect sizes across studies were large and signifi- ing (Qw[df = 5] = 9.50, p = .09). No significant moderators
cant for all types of sensory scores, SOR 1.28 (k = 47, 95% explained the SOR heterogeneity.
CI = [1.11–1.45]), SUR 1.38 (k = 43, 95% CI = [1.17–1.59]),
and Seeking 0.66 (k = 30, 95% CI = [0.25–1.07]: see Figs. 3, Comparison of ASD and Clinical Groups
4, 5). Forty-five of the 47 studies with SOR scores were
positive and significant (d = 0.47–2.57; with 84.4% with The average effect size for the seven studies comparing SOR
large effect sizes greater than 0.8). Two non-significant of ASD to another non-DD clinical group (i.e., ADHD,
studies were Gonthier et al. (2016) with a negative effect SMD, LD) was significant, 0.52 (95% CI = [0.19–0.85];
size and Van Etten et al. (2017) with a positive effect see Table 3). Four of the studies showed significant posi-
size. For SUR, 41 of the 43 studies were positive and sig- tive ds from 0.33 to 1.58. Testing showed significant het-
nificant (d = 0.43–2.82; with 85.4% greater than 0.8). One erogeneity among the SOR effect sizes (Qw[df = 6] = 22.93,
study had a significant negative SUR effect size (− 0.89, p = 0.0008); however, moderators with sufficient data were
95% CI = [− 1.13 to 0.66]). Only 1 study was not signifi- not significant. Average effect size was not significant for
cant (d = 0.06, 95% CI = [− 0.47 to 0.58]). For Seeking, 20 SUR, 0.31 (95% CI = [− 0.16 to 0.76]), and for Seeking
out of 30 studies had positive and significant effect sizes − 0.10 (95% CI = [− 0.41 to 0.21]).
(d = 0.30–2.15; with 75% greater than 0.80). Seven studies
had significant negative Seeking effect sizes. Three stud-
ies had a non-significant positive effect size with a CI that Discussion
crossed ‘0’.
While aggregated average effect sizes were significant, Since the 2009 meta-analysis and the inclusion of sensory
they were not homogeneous (SOR Qw[df = 46] = 486.43, symptoms in the DSM-5 ASD diagnostic criteria, there has
SUR Qw[df = 42] = 724.07, Seeking Qw[df = 29] = 1146.32, been a dramatic increase in research in this area. In addi-
p < .0001 for all). This suggests that studies varied in the tion to our goal to aggregate a larger body of evidence, the
magnitude of difference between groups. Therefore, potential current meta-analysis was able to expand inquiry to mod-
moderators were tested. Demographic and methodological erators for which previously we had limited data such as
age, male ratio, other clinical group comparisons, and type
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Fig. 3 Effect sizes (d) and 95% CI for SOR between ASD and typically developing groups
of questionnaire. The current meta-analysis focused on 55 groups other than DD/ID. Findings from this meta-analysis
studies comparing individuals with ASD to typical and strongly support the inclusion of sensory symptoms as part
clinical control groups using sensory processing question- of the diagnostic criteria of ASD, given the significant differ-
naires. Particular growth was observed in studying older age ences among most sensory comparisons and their robustness
groups, groups with higher functioning individuals, sam- resulting from their independence of the demographic and
ples from Asian countries, and inclusion of clinical control methodological variations among studies. The main findings
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Fig. 4 Effect sizes (d) and 95% CI for SUR between ASD and typically developing groups
showed how SOR uniquely differentiates individuals with by age, IQ and self-report. This meta-analysis indicates the
ASD, no matter what the comparison group and with no age, need to examine different types of sensory patterns sepa-
developmental, or methodological explanations. While SUR rately and to investigate factors underlying variability in
distinguished ASD from TD groups, differences diminished their expression.
when related to other clinical groups. Seeking was elevated SOR differences were the most prominent, as shown
in ASD compared to TD and DD groups and was moderated by their consistently positive differences across individual
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Fig. 5 Effect sizes (d) and 95% CI for Seeking between ASD and typically developing groups
studies and significance across all types of control groups. moderated by age, type of group matching, and whether over
The percentage of individual studies with significant effect 80% of the sample included people diagnosed with autism.
sizes was highest among comparisons to a TD group It is plausible that the current moderators estimating level
(95.74% for TD, 54.55% for DD, and 57% for clinical com- of functioning and development lost their power as a result
parison). TD comparison studies showed significant het- of the addition of older and higher functioning samples in
erogeneity; however, none of the moderators tested could this meta-analysis relative to the previous report. While
account for this. In our previous meta-analysis, SOR was at the individual study level moderators such as IQ were
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Journal of Autism and Developmental Disorders (2019) 49:4974–4996 4987
Table 3 Effect size of sensory symptoms of ASD compared to other clinical groups
Author SOR SUR Seeking
d CI d CI d CI
Ermer and Dunn (1998) 0.90 [0.5, 1.31] 0.6 [0.19, 1] − 0.51 [− 0.91, − 0.1]
Cheung and Siu (2009) 0.07 [− 0.22, 0.37] − 0.52 [− 0.81, − 0.22] − 0.27 [− 0.56, 0.03]
O’Brien et al. (2009) 0.60 [0.07, 0.14] 0.54 [0.01, 1.08] 0.63 [0.1, 1.17]
Mattard-Labrecque et al. (2013) 1.58 [0.86, 2.31] 1.32 [0.6, 2.05] 0.47 [− 0.26, 1.19]
Clince et al. (2016) 0.35 [− 0.18, 0.88] 0.26 [− 0.27, 0.78] − 0.62 [− 1.15, − 0.09]
Little et al. (2018) 0.22 [− 0.1, 0.53] − 0.03 [− 0.34, 0.29] − 0.21 [− 0.52, 0.11]
Tavassoli et al. (2018) 0.33 [0.01, 0.66] 0.58 [0.26, 0.9] 0.07 [− 0.25, 0.4]
Average 0.52 [0.19, 0.85] 0.35 [− 0.06, 0.76] − 0.10 [− 0.41, 0.21]
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4988 Journal of Autism and Developmental Disorders (2019) 49:4974–4996
in 2009, SUR of the SSP studies was not analyzed due to explained by age and IQ. Alternatively, the inconsistent
the limited number of items in this scale, resulting in very conceptualization of Seeking across studies might lead to
few studies included in the previously tested model. In the this variability (Schauder and Bennetto 2016). Part of this
current review, SUR of the SSP was included given the large inconsistency is that Seeking items in young children’s
number of SSP studies identified as well as several raw data- scales refer to intense sensory exploration, while in adult
sets which became available and enabled a richer computa- questionnaires, such as the AASP, they refer to personal
tion of SUR and Seeking for SSP studies. Even though SSP sensory preferences, which do not capture the repetitive
is very limited in measuring SUR, its use in a study did not circumscribed, idiosyncratic nature of Seeking in ASD
appear to moderate SUR differences. (Elwin et al. 2013).
Surprisingly, SUR average effect size was positive and
significant relative to TD controls but not relative to DD or Developmental Level and Sensory Symptoms in ASD
other clinical groups. The individual DD comparisons indi-
cate a diversity in the direction of the effect size, with two We approached the investigation of the role of developmen-
studies showing a negative effect size for very young chil- tal level in explaining sensory symptoms in ASD from dif-
dren (McCormick et al. 2016; Rogers et al. 2003). The lack ferent angles to capture as many studies as possible and to
of significant SUR effects is surprising as many of the sen- obtain a comprehensive definition of this construct. This
sory questionnaires ask within their SUR scale about social included coding IQ, level of functioning of ASD group, type
under-response which is characteristic of ASD. Hence, of matching (CA- vs. MA-based), and separating effect sizes
one would expect that SUR would be consistently different compared to typical versus DD groups. Nonetheless, the
across comparisons. Although developmental moderators various developmental level variables were not significant,
did not explain the SUR TD model, the lack of significant except in the Seeking TD model. This is consistent with
comparisons with DD supports the role of development in findings of lack of relation between sensory symptoms and
the manifestation of SUR symptoms in ASD. cognitive abilities (O’Donnell et al. 2012). Note that in the
Atypical sensory seeking difference, while smaller in 2009 meta-analysis, the moderation of developmental level
magnitude relative to the other sensory patterns, was the was estimated more crudely by the type of matching and
only pattern with significant moderating factors. Average the dichotomous severity of autism variable (> 80% autism
Seeking effect size was significant when compared with diagnosis). In the current meta-analysis, there was a trend
TD and DD groups (ASD group on average showed greater toward a relation with development level seen by lower aver-
Seeking) but not relative to other clinical groups. None- age d’s and fewer significant d’s when ASD was compared
theless, there was great variability in the direction of the to DD and other clinical groups versus TD comparisons.
effect size and in its significance among the individual In addition, SUR was only significant for TD comparison,
studies. Seven of the 30 studies with Seeking effect sizes, suggesting it is a symptom mostly accounted for by devel-
had significantly negative effect sizes (ASD group shows opmental level.
less Seeking), six of which included adolescents/adults, Conflicting findings for the association between sensory
and five of the six relied on the AASP. Three studies were symptoms and adaptive behaviors (i.e., proxy of develop-
not significant and the rest (k = 20) were significant and mental level) were also reported in a recent review of cor-
positive. The ASD versus TD Seeking effect size was relational research by Dellapiazza et al. (2018). The diver-
moderated by age, IQ and self-report. There was a non- sity in the operational definition of sensory patterns and of
linear relation with age group, with increases through the developmental level among studies requires caution in gen-
6–9 years’ age group and a decrease thereafter. This is erating far-reaching conclusions concerning the contribution
consistent with the non-linear relation we reported in 2009 of developmental level to the severity and type of sensory
for Seeking. Looking at individual studies, we saw stabil- symptoms.
ity in studies with a young narrow age range (McCormick
et al. 2016) versus a decrease in sensory symptoms when Sensory Symptoms in ASD Versus Non‑DD Clinical
looking across a wider age range (3–56 years, Kern et al. Disorders
2006; 3–11 years, Leekam et al. 2007). It seems that in
older samples, the atypicality is in the low level of seek- Compared to other clinical groups that did not include a
ing, which may be an outcome of avoidance. Higher IQ DD/ID, the SOR average d was the only significant pattern.
was associated with a smaller Seeking effect size. In addi- Nonetheless, the magnitude of d and the number of signifi-
tion, there was a smaller effect size for studies using a cant individual d’s was lower relative to the TD and DD
self-report measure; probably a factor of higher IQ. The SOR models. At the individual study level, three (Cheung
great heterogeneity in effect sizes among studies for Sen- and Siu 2009; Clince et al. 2016; Little et al. 2018) of
sory Seeking might be the reason it is the only pattern the five studies comparing ASD to an ADHD group had
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Journal of Autism and Developmental Disorders (2019) 49:4974–4996 4989
non-significant SOR d. Further understanding of sensory dif- Kern et al. 2007). The skewed distribution of age as a con-
ferences between ASD and ADHD is needed. These results tinuous variable supported the analysis of age groups. Age
regarding non-DD clinical disorders are based on a very few groups were defined by the mid-age range of the group, how-
studies (k = 7) which looked at clinical comparisons other ever at sometimes this could not fully capture the heteroge-
than DD (k = 5 ADHD, 1 SPD, 1 LD). None of the studies neous age range in the study.
compared ASD to a psychopathological group with anxiety We were limited in our ability to examine the effects of
disorder or OCD, which also present extreme sensory symp- gender upon sensory symptoms, hence its non-significant
toms. There was also no language disorder or social–com- moderation across models was not surprising. This was due
munication disorder control group, conditions important to to the narrow range and variance of the male ratio leading
study, particularly when thinking of the strong social com- to a non-normal distribution among the compared studies.
ponents of tactile and auditory stimuli. To fully examine this effect more sensory data for females
with ASD is needed.
Limitations From a measurement perspective, most measures are rely-
ing upon parental reports about the frequency of the behav-
Several limitations within this body of literature should be ior or the number of symptoms. A few studies included self-
acknowledged when interpreting the findings. Several stud- reports, which differ in that they provide a first-hand account
ies were excluded because they did not publish means or from individuals with high abilities. In addition, current
SDs for the sensory scores or did not report scores reflect- healthcare models (e.g., ICF) stress the need to assess limi-
ing one or more of the three distinct sensory pattern scores. tations based on the condition’s impact upon performing
For example, the SSP does not yield a distinct Seeking activities and participation. The degree of interference/
score, hence if authors did not send us means or raw data functional impairment associated with sensory symptoms
so we could compute distinct SUR and Seeking scores their in ASD need to be measured.
research could not be included in the SUR and Seeking mod- While conceptually, we aimed to examine comparable
els. This may have created bias among SSP studies. While constructs (see “Appendix”), variability in the actual meas-
many authors sent us these scores, others did not or used urement of the construct may have biased results beyond
other questionnaires that do not readily provide these type of what we could identify in this meta-analysis. First, some
scores (e.g., Sensory Processing Measure, Parham and Ecker of the studies modified the use of standard sensory ques-
2007). The publication of the Sensory Profile 2 versions will tionnaires. This included self-report rephrased for caregiver
reduce this limitation by providing the same three pattern report for individuals who are low functioning and living in
scores across age groups. a care center (Gonthier et al. 2016), reducing the number
ASD severity was not examined directly in this meta- of SP items for the Chinese SP to fit culturally (Cheung
analysis due to the studies relying on different metrics, dif- and Siu 2009), or using the SSP for a younger age range
ferent versions of the DSM, or not reporting severity at all. (Wiggins et al. 2009). Second, the number of items and
Studies varied in the homogeneity of the ASD group, with types of modalities covered by each questionnaire varied
some purposefully studying a unique type of ASD, such as between measures. For instance, SUR in the SSP was based
only HFASD (Hilton et al. 2010), LFASD (Gonthier et al. on two modality-specific items compared to 11 items across
2016), or those with ASD and ADHD (Mattard-Labrecque modalities in the ITSP. Third, even for the same measure,
et al. 2013). Our previous moderator testing of percent of scores may have reflected a different set of items, such as,
autism diagnosis was no longer relevant given changes in researchers who used the SSP but added a few factors from
DSM-5 classification. We partially captured ASD severity the SP (O’Brien et al. 2009). The recent publication of one
by examining developmental level. scoring system across versions of Sensory Profile will assist
It is worth noting that IQ was measured in different ways in comparing results across studies.
across studies. Only non-verbal testing was used in some Finally, for studies with multiple scores, we averaged
studies, while full-scale IQ was used in others. Crane et al. their effect size into a composite effect size. Nonetheless, in
(2009) demonstrate how different types of IQ scores yield the absence of information regarding the shared covariance
different correlations with SOR, SUR and Seeking. Set- between the subscores, there is no correct way to estimate
ting unified guidelines for phenotyping in research would the standard error of this composite effect size. Although
strengthen the ability to conduct wide-scale, cross-site the method we used is common practice in meta-analytic
research, as well as large meta-analyses. research (Lipsey and Wilson 2001), its limitations should
The age range within some samples was very wide, with be acknowledged.
some studies including both adolescents and adults (e.g.,
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4990 Journal of Autism and Developmental Disorders (2019) 49:4974–4996
Appendix
See Table 4.
13
Table 4 Comparison of sensory scores across studies
Over-responsivity Under-responsivity Seeking Comment
Sensory Profile (Dunn and Westman 1995; Dunn Sensitivity, oral sensitivity Poor registration, Seeking In 2002 a scoring of the Sensory Profile items by
2002) low endurance/ quadrants was published (Dunn 2002) hence later
tone studies report these scores for the Sensory Profile
Sensory Profile 2 (Dunn 2014) Sensitivity, avoiding Low registration Seeking
Short Sensory Profile (SSP)b (McIntosh et al. Auditory/visual sensitivity, tactile sensitivity, Low energy Auditory filtering, and underresponsive/seeking
1999) taste/smell sensitivity, movement sensitivity scores do not correspond to one pattern
Infant/Toddler Sensory Profile (ITSP) (Dunn Sensitivity, avoiding Low registration Seeking
2002)
Adult/Adolescent Sensory Profile (AASP) (Brown Sensitivity, avoiding Low registration Seeking
and Dunn 2002)
Sensory Experience Questionnaire (SEQ v1 and Hyper-responsive Hypo-responsive Seeking The authors provided us with hypo-responsive sepa-
Journal of Autism and Developmental Disorders (2019) 49:4974–4996
v3) (Baranek et al. 2006; Baranek 2009) rated from Seeking scores
Sensory Sensitivity Questionnaire-Revised (Talay- Visual, auditory, taste, smell, tactile, vestibular
Ongan and Wood 2000) sensitivity
Sensory Sensitivity Survey (Liss et al. 1998) Over-reactivity Under-reactivity Seeking 104 Items but based on 60 items from the Sensory
Profile
Japanese Sensory Inventory Revised (Ota et al. Over-reactivity Under-reactivity Seeking Scores reported by modality. Therefore items were
2002) classified into patterns as part of this meta-analysis
using experts’ inter-rater agreement
The Sensory Processing Scales Inventory: Sensory SOR The measure also has SUR and Seeking scores but
Over-Responsivity Subscale (Miller and Schoen not for the studies included
2012)
Sensory Processing Quotient (SPQ) (Tavassoli SOR
et al. 2014a, b)
Sensory Reactivity in Autism Spectrum (SR-AS; Hyper-reactivity Hypo-reactivity Strong
Elwin et al. 2016) Sensory
Interests
Chinese Sensory Profile (CSP; Tseng and Cheng Adapted Sensory Profile. Scores reported by modal-
2008) ity. Therefore, items were classified into patterns
as part of this meta-analysis using experts’ inter-
rater agreement
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