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Journal of Autism and Developmental Disorders

https://doi.org/10.1007/s10803-019-04159-x

ORIGINAL PAPER

Effects of Diagnostic Severity upon Sex Differences in Behavioural


Profiles of Young Males and Females with Autism Spectrum Disorder
Vicki Bitsika1 · Christopher F. Sharpley1

© Springer Science+Business Media, LLC, part of Springer Nature 2019

Abstract
To determine if diagnostic severity of Autistic Spectrum Disorder (ASD) affected sex differences in the detailed ASD-related
behavioural profiles of young males and female, two samples of males and females with a diagnosis of ASD, aged between
6 and 17 years, were compared across the 65 items of the Social Responsiveness Scale (2nd ed.). Results are reported for a
sample of males and females matched on age and IQ (n = 51 pairs) and a smaller sample matched on age, IQ and ADOS-2
severity (n = 32 pairs). ASD-related behaviours from the SRS-2 that were significantly and meaningfully different across
sexes were identified for both samples. ADOS-2 diagnostic severity was associated with different sets of sex-based differ-
ences in SRS-2 item scores.

Keywords Autism · Sex differences · Diagnostic severity · Behavioural profiles · Social communication

It has been noted for some time that there is a difference were not actually interacting with them, while ASD boys
in the prevalence of Autism Spectrum Disorder (ASD) tended to play alone (Dean et al. 2017).
between males and females (Wing 1981). Although that This suggestion that females may camouflage certain
difference is often quoted as 4:1 (APA 2013), the reported aspects of their ASD-related behaviour raises the issue of
prevalence ratio has been found to be between 2:1 and 7:1 sex differences in symptom profiles as a possible contribu-
males:females, depending on the sample (Halliday et al. tor to the prevalence of overall diagnosis, but there is also
2015), with some recent evidence from a meta-analysis of some lack of consistency in findings regarding sex dif-
54 studies of more than 13 million participants indicating ferences in the profile of symptoms across studies (Ferri
that the actual ratio was closer to 3:1 (Loomes et al. 2017). et al. 2018). For example, some papers have reported more
Various explanations have been given for this difference in severe ASD core symptom deficits for males (Rynkiewicz
prevalence, including genetic factors (Ferri et al. 2018), foe- et al. 2016) and some have found the opposite (Frazier et al.
tal hormonal influences (Baron-Cohen et al. 2014), neuro- 2014). There is also evidence of differentiation in prevalence
transmitter effects (Shuffrey et al. 2017) and immunological within the ASD core symptoms themselves, as shown in van
expression (Hanamsagar et al. 2017). There is also a sug- Wijngaarden-Cremers et al. (2014) meta-analysis of 22 stud-
gestion that females do not actually have a lower prevalence ies which noted that males tended to show more evidence
of ASD but are underdiagnosed because they exhibit ‘cam- of restricted and repetitive behaviours than females but that
ouflage’ behaviour that makes certain ASD symptoms less there were no significant differences in social behaviour and
noticeable (Hull et al. 2017; Kreiser and White 2014). Some communication. Those authors also commented that there
data to support this suggestion come from an observational was a potential for differences in IQ to confound these sex-
study of social behaviours among ASD and non-ASD chil- differences in symptom profiles. That suggestion is sup-
dren of both sexes that found that ASD girls used ‘compen- ported by evidence from a recent PRISMA review of the
satory’ behaviours of staying near peers even though they literature on female camouflaging in ASD (Allely 2018) that
noted that the ability to effectively imitate socially accept-
* Christopher F. Sharpley able behaviour was particularly likely to occur in females
csharpl3@une.edu.au; csharpley@onthenet.com.au with intelligence within the normal range.
The issue of assessment and participant selection method-
1
Brain‑Behaviour Research Group, University of New ology in studies of ASD sex differences is therefore relevant
England, Armidale, NSW 2350, Australia

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Vol.:(0123456789)
Journal of Autism and Developmental Disorders

for consideration. Two of the most pertinent aspects of mainstream schools and are able to self-care, increases
that methodology are the instrument used and the source the likelihood of including females who may be adept at
of information collected. According to van Wijngaarden- camouflaging, and therefore could provide a basis for com-
Cremers et al. (2014), the most common instrument used paring their ASD-related behavioural profile with males
in sex difference studies in ASD is the ADOS (Lord et al. with the same level of cognitive and self-care skill. Fourth,
2012). Although the ADOS is an excellent instrument for the sampling from the age ranges during which children usu-
purpose of making a diagnosis of ASD, it is firstly designed ally attend school (i.e., 6 years to 18 years) and where pre-
to identify the presence of ASD rather than the detailed pro- vious researchers have observed camouflaging behaviour
file of ASD-related behaviour. Investigation of the detailed (Dean et al. 2017) would allow for the testing of effects
nature of any differences in the profile of ASD-related due to age and maturity associated with greater length of
behaviour across males and females would benefit from use time at school and the likelihood of learning social cam-
of an instrument that can provide a fine-grained account ouflaging skills there.
of the ASD-related observable behaviours of those males An additional aspect of methodology which has
and females rather than overall subscale or total scores, as received scant attention is the effect of ASD severity upon
are obtained from the ADOS. Similarly, the use of a wide specific symptom profiles of male and female samples.
scale of severity for responses can also provide valuable data That is, most previous studies have focussed upon (a)
when compiling behaviour-severity profiles. applying an instrument such as the ADOS or ADIR, and
In terms of source of information about a child’s ASD, (b) testing for the presence of significant differences in
although clinicians decide on the presence of ASD from scores from these instruments across males and females
their observations during assessment procedures, a major (van Wijngaarden-Cremers et al. 2014). This is an appro-
source of data regarding the everyday functioning of a child priate methodology for the early stage of sex difference
with ASD is the child’s primary caregivers, usually its par- investigations, but does not allow for the more detailed
ents, because they are most familiar with the child’s behav- comparison of ASD-related behaviours across males and
iour. One instrument designed to be completed by caregiv- females within the same ASD severity range. That is, while
ers, which collects data on 65 behaviours related to ASD, the severity of ASD symptomatology per se via the ADOS
and which compiles scores on a four-point Likert scale, is or ADIR has been the research target of most previous
the Social Responsiveness Scale (2nd ed.) (SRS-2) (Con- studies, the examination of the profile of detailed ASD-
stantino and Gruber 2012), which has been widely used in related behaviours across males and females with matched
studies of people with ASD. Use of such a detailed measure age, IQ and ASD severity would provide an opportunity to
of ASD symptoms and related behaviours, collected from compare males and females at a greater level of detail and
the child’s parent, may provide greater depth and validity of with three major potential sources of confound controlled
data regarding everyday ASD-related behaviour than clini- for by matching of male–female pairs. The inclusion of
cians’ observations within a limited timeframe. ASD severity (as determined by the ADOS-2) rather than
These findings, suggestions, and comments argue for just age and IQ would clarify any sex differences in the
the further examination of sex differences in the behaviour wider set of ASD-related behaviours that may be attribut-
of young people with ASD, but with several methodologi- able to the overall severity of the ASD per se (i.e., via the
cal provisos that may help clarify how any sex differences ADOS-2 Total Score) rather than simply sex or IQ.
in this ASD-related behaviour manifest themselves. First, Therefore, this study aimed to investigate the detailed
although the ADOS is the most commonly-used instru- ASD-related parent-reported behavioural profiles of a
ment in studies of sex differences in ASD (van Wijn- sample of school-age males and females who had firstly
gaarden-Cremers et al. 2014), the sourcing of information been matched on age and IQ (as in most previous research
from parents may provide a greater insight into the kinds on sex differences) and then a subsample that had also
of behaviours exhibited by children with ASD when they been matched on ASD severity. This latter step offers the
are not in structured situations such as clinical assessment opportunity to test for the effects of ADOS-2 diagnos-
settings. Second, the use of a more detailed instrument tic severity upon differences in ASD-related behavioural
(such as the SRS-2) which asks parents to rate their child profiles within an age- and IQ-matched sample of young
on a four-point Likert scale for severity of ASD-related males and females.
behaviours, could provide finer-grained data regarding
sex differences across a wide range of these behaviours
and allow for profile comparisons that are not possible
in studies of the presence/absence of ASD as defined by
the ADOS cutoff. Third, constraining the samples of chil-
dren with ASD to those with an IQ > 70, and who attend

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Journal of Autism and Developmental Disorders

Methods males had been diagnosed with Attention-Deficit/hyperac-


tivity Disorder (ADHD), two with Obsessive–Compulsive
Participants Disorder (OCD) and 3 with Oppositional Defiant Disorder
(ODD), plus 12 females with ADHD, 2 with Generalised
Total Sample Anxiety Disorder (GAD), 3 with OCD and 7 with unspeci-
fied “other” disorders, according to the parents of these
Fifty-one males and 51 females with ASD and one of their children. According to their parents, 14 of the females
parents (95 mothers) were recruited from a local parent had reached menarche, but no comparative data regarding
support group and ASD service organizations in the Gold puberty were able to be obtained from the males’ parents.
Coast, Queensland, Australia, for a study “about ASD”. As Further biological tests for puberty were declined by the
such, the sample may be described as “community” rather parents of both the males and females. There were no sig-
than “clinical”. The mean age of the 51 males with ASD nificant main effects for ADOS-2, WASI-II Full Scale IQ
was 10.2 years (SD = 2.8 years, range 6 to 17 years) and for or any of the five SRS-5 subscales according to the puber-
the 51 females with ASD it was 10.1 years, SD = 2.7 years, tal status of the females F(7,44) = 1.457, p = .208, and so
range 6 to 17 years). The males and females were formed the females’ data were analysed as a combined sample,
into age- and IQ-matched pairs on a one-to-one basis so with age included in the analyses (explained below).
that each male was matched with a female whose age was
within 6 months of his, and whose WASI-II Full Scale ASD‑Severity Matched Sample
IQ score was within five points of his, so that there were
no significant differences in either age (F(1,101) = .020, The subsample that was matched on the ADOS-2 Total
p = .887) or IQ (males: M = 97.9, SD = 12.0, range Scores as well as their age and IQ consisted of 32 males and
76–125; females: M = 98.2, SD = 13.1, range 77–128: females drawn from the original sample (not all participants
F(1,101) = .006, p = .937) between the males’ and females’ had scores that enabled them to be matched with another
samples on these two variables. All the males and females participant of the opposite sex). Table 1 shows the age, IQ
had received a Total Score of > 7 on the ADOS-2 (Classi- and ADOS-2 data for this smaller sample, plus the results of
fication and Severity scores were also calculated to ensure the MANOVA on these variables, using sex as the independ-
that each participant met the accepted criteria for a diag- ent variable. Although the statistical power of this reduced
nosis of ASD as set out in the ADOS-2 Manual), adminis- sample was also reduced compared to the original (n = 51)
tered during intake for this study by a clinically-competent sample, it was great enough to detect a large effect at p < .05
research assistant who had been trained in the use of the level with power of almost .80 (Cohen 1988).
ADOS-2, as specified by the ADOS-2 Training Protocols
and Manuals developed by the ADOS-2 authors for the Age
purposes of valid administration of this scale. This sample
had been used in a previous study of sex differences in As explained above, in the absence of any biological data
Sensory Features in ASD (Bitsika et al. 2018). for both males and females, a simple dichotomisation based
All these participants had been originally diagnosed upon the presumed age of puberty may be inaccurate due
from a 2-h clinical interview conducted by either a regis- to variable maturation rates. In addition, dichotomisation
tered paediatrician or psychiatrist with their parents using of what is actually a continuous variable can have limiting
the relevant DSM criteria for ASD, plus family history. effects upon the reliability and power of statistical proce-
These diagnoses were confirmed by a suitably qualified dures (Chen et al. 2007; Cohen 1983). Therefore, testing
and registered clinical psychologist with expertise in for age effects was done via correlational procedures rather
ASD and were again confirmed during the recruitment than ANOVA models.
process for the current study by the ADOS-2 (Lord et al.
2012) as described above. In addition, all these partici- Table 1  Age, IQ and AOS-2 Scores for males versus females
pants had their original WISC-IV Full Scale IQ of > 70 matched on age, WASI-II IQ, and ADOS-2 Total Score
confirmed by the WASI-II during recruitment, and all
Variable Males (n = 32) Females (n = 32) F p
attended mainstream schools. According to their parents,
who were asked to report any clinical diagnosis previously Age (years) M = 10.2 (2.8) M = 10.1 (2.7) 0.072 .789
given by the psychiatrist or psychologist who was respon- Range 6 to 17 Range 6 to 17
sible for the care of their child, twenty-nine (56.9%) of the WASI-II Full Scale M = 95.8 (13.7) M = 99.9 (12.8) 1.593 .292
IQ Range 74 to 125 Range 79 to 128
males and 29 of the females had no concurrent diagnosis
ADOS-2 Total M = 11.8 (2.2) M = 12.4 (2.6) 0.858 .358
of another psychiatric or developmental disorder, but 17
Score Range 7 to 17 Range 7 to 17

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Journal of Autism and Developmental Disorders

Instruments that reflect a range of severities rather than dichotomous


classifications, and also to tap a wide range of ASD-related
The Autism Diagnostic Observation Schedule Second behaviours (Constantino and Gruber 2012).
Edition (ADOS‑2)
Procedure
The ADOS-2 (Lord et al. 2012) is recommended in several
Best Practice Guidelines as an appropriate standardized Data-collection was conducted in the homes of all partici-
diagnostic observation tool for ASD (Filipek et al. 1999; pants in order to avoid stress upon them by attending the
National Research Council 2001). It presents a series of researchers’ laboratory. The parents were given written
standardized interactive activities that focus upon social instructions on how to complete the SRS-2. The WASI-II
interactions, communication and repetitive behaviours, pro- and ADOS-2 assessments were conducted with participants
viding data for a diagnosis of ASD. in their homes within 1 week of the collection of SRS-2 data.
The study was approved by the Bond University Human
The Wechsler Abbreviated Scale for Intelligence (WASI‑II) Research Ethics Committee approval no. RO 1516 and all
procedures complied with the 1964 Helsinki declaration
The WASI-II contains four subtests with average reliability and its later amendments. Written informed consent was
coefficients of between .92 and .96, and has strong validity obtained from all parents included in the study and their
with the WISC-IV when used with high-functioning people children gave their assent to the study.
with ASD (Minshew et al. 2005). Results from the WASI-II
provide an accurate estimate of intelligence that is useful Statistical Analysis
for research screening purposes and produces results on two
composite scales (Verbal Comprehension, Perceptual Rea- Data were analysed via IBM SPSS 24 to obtain mean and
soning), plus a Full Scale IQ based on those two composites. 5% trimmed mean values (as an indicator of the effects of
The Full Scale IQ was used as the measure of cognitive outliers), SD, and ranges. Normality was assessed by the
ability in this study. Kolmogorov–Smirnov statistic. Pearson correlation analy-
sis tested for any significant association between SRS-2
Social Responsiveness Scale (2nd Ed) (SRS‑2 Total Scores, items and age. MANOVA was used to test
for the presence of significant differences in males’ vs
The Social Responsiveness Scale (2nd ed.) (SRS-2) (Con- females’ ages, WASI-II Full Scale IQ scores, and ADOS-2
stantino and Gruber 2012) consists of 65 items that may be Total Scores, and for the effects of menarche upon females’
answered by a parent or other caregiver about a child with scores on these instruments. MANOVA tested for any sig-
ASD. These items describe autistic traits (i.e., interpersonal nificant main effects on SRS-2 Total Scores according to
behaviour, social communication, and repetitive/stereotypic sex. MANOVA main effects were further investigated via
behaviour) in respondents aged 4 to 18 years of age. The univariate analysis of the 65 SRS-2 items. Wilks’ Lambda
SRS-2 uses a four-point Likert scale (not true = 0, some- was used as the determining outcome statistic for all mul-
times true = 1, often true = 2, almost always true = 3), provid- tivariate effects, with appropriate Bonferroni correction for
ing a total score ranging from 0 to 195 so that continuous family-wise error rate.
scores of Social Awareness, Social Cognition, Social Com-
munication, Social Motivation, and Restricted and Repeti-
tive Behaviours, as well as a Total score, can be calculated Results
(Constantino and Gruber 2012).
The SRS-2 has strong agreement with the ADOS (Lord Overall Data
et al. 2012) and the Developmental, Dimensional and Diag-
nostic Interview 3Di (Duvekot et al. 2014; Skuse et al. The SRS-2 Total Scores were within acceptable normality
2004). As well as raw scores, sex-specific norms are avail- limits as assessed by the presence of nonsignificant Kol-
able to convert SRS-2 raw scores to Treatment-scores for mogorov–Smirnov statistics of .056 for the 51 males, and
individualised treatment planning, although raw scores are .065 for the 51 females (both p > .05); similar results were
most commonly used in research studies (Constantino and found for the 32 males (K–S statistic = .087) and 32 females
Gruber 2012) and were used here. Cronbach’s alpha for the (K–S = .113), and neither data set required transformation.
SRS is in excess of .90 for the Total score and from .76 to The 5% trimmed means were within < 1% of the actual
.91 (median = .85) for the five subscales (Constantino and means for both sexes within both samples, indicating only
Gruber 2012). The authors of the SRS-2 argued that one of negligible effects from outliers. Because multicollinearity is
its major strengths is its ability to provide scores about ASD a potential source of error in MANOVA calculations when

13
Journal of Autism and Developmental Disorders

many items from the same scale are analysed as individual conducted in groups of 20 to 25 SRS-2 items. The Bon-
dependent variables, Pearson correlations were run for all ferroni-adjusted level of statistical significance for sex dif-
65 SRS-2 items but none of the values were greater than ferences was 05/65 = .0007, which may have been a poten-
r = .67, which is less than the level of .9 recommended as tial source of Type II error. Therefore, a two-step selection
the indicator for violation of this assumption (Tabachnik procedure was undertaken to identify SRS-2 items which
and Fidell 2013). showed meaningful differences across the sexes as indicated
by (a) p < .001, plus (b) an effect size (partial eta squared)
Age Effects of at least .14, that represents a ‘large’ effect (Cohen 1988).
Both of these steps are conservative in that p < .001 repre-
There were no significant correlations between age and sents a chance of a Type I error that is < 1 in 1000, and an
WASI-II Full scale IQ, ADOS-2 Total Score or the SRS-2 effect size of .14 is well-recognised as only being present
Total Scores or any of the five SRS-2 Subscale Scores, for when the associations being tested are very robust (Tabach-
either the larger (n = 51) or reduced (n = 32) male and female nik and Fidell 2013). Tables 3, 4 show the SRS-2 items that
samples. met these dual criteria in bold, with the mean (SD) values
for males and females, for both samples (i.e., Table 3: n = 51
Sex Differences males and females; Table 4: n = 32 males and females).
Two sets of information can be drawn from Tables 3,
SRS‑2 Total Score 4, firstly about the differences in frequency of occurrence
across males and females, and secondly, in terms of the
Table 2 shows the mean SRS-2 Total Scores for males and actual content (i.e., behavioural differences) that is repre-
females for each of the samples, with ANOVA results. sented by these items. First, in terms of numerical differ-
Although there was no significant sex difference in these ences alone, Table 4 table represents those SRS-2 items
SRS-2 Total Scores, the focus of this study was upon indi- on which sex differences existed but which were free from
vidual ASD-related behaviours, and so the remaining analy- the effects of a possible confound due to ASD severity as
ses for sex differences were undertaken at the individual measured by the ADOS-2. There were 27 SRS-2 items with
SRS-2 item level. This is congruent with the suggestion meaningful differences (i.e., both p < .001, also a large effect
that, although the main effect from MANOVA analyses size). These 27 ASD-related behaviours represent the most
may be nonsignificant, exploration of the univariate effects comprehensive account of sex differences across the 6 years
may be undertaken because “the multivariate F is often not to 18 years age group (with no significant age effects) in a
as powerful as univariate or stepdown F and significance sample that was controlled for age, IQ and ADOS-2 severity.
can be lost” (Tabachnik and Fidell 2013, p. 348). Although Females had meaningfully higher scores on each of these 27
MANOVA was not used at this stage, the analogy holds for ASD-related behaviours.
the relevance of this ANOVA on SRS-2 Total Scores. Table 5 shows these 27 items classified into the five Treat-
ment Subscales defined by the SRS-2, and indicates that the
SRS‑2 Item Data most common sex differences were found for Social Com-
munication and Social Motivation, and the least common sex
To reduce the likelihood of an error due to violation of one differences were found for Social Awareness and Restricted
of the key assumptions underlying MANOVA (i.e., sam- Interests and Repetitive Behaviour. When examined for their
ple size:dependent variable ratio), these analyses were Spearman correlation coefficients with sex, all of these 27
items were statistically significant at the p < .001 level, and
10 of them met the criteria for a ‘large’ effect size of .50 or
Table 2  Males’ and females’ SRS-2 Total Scores and ANOVA results
greater (Cohen 1988), shown in bold in Table 5. These may
Sample M (SD) M (SD) F p Partial be identified as the SRS-2 items which were most strongly
Males Females eta
associated with sex differences in this sample of males and
squared
females matched for their ASD severity.
N = 51a Second, in terms of the content of these items, com-
SRS-2 Total 97.15 (28.91) 99.35 (24.82) 0.667 .409 .007 parison of Tables 3, 4 allows for the evaluation of possible
Score ASD severity effects upon sex differences by identifying
N = 32b the SRS-2 items which were found to be meaningfully sig-
SRS-2 Total 99.25 (30.41) 95.46 (26.46) 2.861 .096 .000 nificant in Table 3 but not in Table 4. There were 20 SRS-2
Score
items which showed meaningful significance in Table 3 (no
a
df = 1,101 ADOS-2 matching for severity) but which did not show
b
df = 1,63 similarly meaningful differences in Table 4 (matched for

13
Journal of Autism and Developmental Disorders

Table 3  Mean (SD) scores for N = 51 males and females Males Females F Partial
selected SRS-2 items showing eta
meaningful sex differences in SRS-2 item (abbreviated; reverse scoring removed) M (SD) M (SD)
squared
bold for total sample (51 males,
females) 1. More fidgety in social situations 2.92 (0.82) 3.64 (0.55) 27.206* .214
2. Facial expressions don’t match what he/she is saying 2.04 (0.87) 3.01 (0.73) 37.764* .274
3. Is not self-confident when interacting 2.66 (1.01) 3.02 (0.78) 3.859 .037
4. When under stress, shows inflexible behaviour 2.82 (0.86) 3.51 (0.62) 21.388* .176
5. Doesn’t recognise when others are taking advantage 2.86 (0.98) 3.71 (0.51) 29.900* .230
6. Would rather be alone than with others 2.27 (0.91) 3.17 (0.68) 31.639* .240
7. Is unaware of what others are thinking or feeling 3.00 (0.82) 2.88 (0.62) 0.638 .006
8. Behaves in ways that seem strange or bizarre 2.15 (0.86) 3.25 (0.65) 52.617* .345
9. Clings to adults, seems too dependent on them 2.09 (1.060 3.13 (0.80) 31.101* .237
10. Takes things too literally 2.92 (0.74) 3.61 (0.56) 27.393* .215
11. Has poor self-confidence 2.65 (0.99) 2.94 (0.86) 2.551 .025
12. Is unable to communicate his/her feelings to others 2.68 (0.88) 2.96 (0.66) 3.155 .031
13. Is awkward in turn-taking interactions 2.62 (0.94) 3.39 (0.75) 21.692* .178
14. Is not well coordinated 2.25 (1.02) 3.07 (0.82) 20.257* .168
15. Able to understand meaning of others’ voice and face 2.80 (0.80) 3.09 (0.67) 4.044 .039
16. Avoids eye contact or has unusual eye contact 2.41 (0.92) 3.19 (0.75) 22.284* .182
17. Does not recognise when something is unfair 2.12 (0.95) 3.39 (0.66) 61.410* .380
18. Has difficulty making friends 2.76 (1.01) 3.41 (0.73) 13.771* .121
19. Gets frustrated trying to get ideas across 2.94 (0.88) 3.67 (0.52) 25.733* .205
20. Shows unusual sensory interests 2.08 (1.07) 3.20 (0.83) 34.726* .258
21. Unable to imitate others’ actions 2.39 (0.85) 3.43 (0.67) 46.942* .319
22. Plays appropriately with children his/her age 2.51 (0.76) 3.22 (.76) 22.146* .181
23. Does not join group activities unless told to 2.49 (0.99) 3.06 (0.66) 11.521 .103
24. Has difficulty with changes in routine 3.08 (0.840 3.55 (0.64) 10.028 .091
25. Doesn’t mind being out of step with others 2.69 (0.88) 3.22 (0.83) 9.710 .089
26. Offers comfort to others when they are sad 2.67 (0.95) 3.27 (0.75) 12.820 .114
27. Avoids starting social interactions 2.20 (0.98) 3.04 (0.75) 23.864* .193
28. Thinks or talks about the same thing over and over 2.94 (0.99) 3.53 (0.64) 12.690 .113
29. Is regarded by other children as odd or weird 2.72 (0.90) 3.59 (0.57) 33.588* .251
30. Becomes upset in a situation with lots going on 2.94 (0.90) 3.69 (0.51) 26.312* .208
31. Can’t get his/her mind off something 3.20 (0.89) 3.80 (0.45) 18.814* .158
32. Does not have good personal hygiene 2.45 (0.97) 3.25 (0.77) 21.596* .178
33. Is socially awkward 2.59 (0.90) 3.47 (0.61) 33.616* .252
34. Avoids people who want to be emotionally close to him 2.02 (0.81) 2.90 (0.76) 32.286* .244
35. Has trouble keeping up with the flow of conversation 2.63 (0.82) 3.31 (0.62) 22.702* .185
36. Has difficulty relating to adults 1.82 (0.74) 2.73 (0.63) 43.611* .304
37. Has difficulty relating to peers 2.71 (0.81) 3.49 (0.64) 29.412* .227
38. Responds inappropriately to mood changes in others 2.92 (0.82) 2.94 (0.70) 0.017 .000
39. Has an unusually narrow range of interests 2.86 (1.00) 3.22 (0.76) 4.038 .039
40. Is unimaginative, poor at pretending 2.67 (0.99) 3.31 (0.79) 13.293* .117
41. Wanders aimlessly from one activity to another 1.90 (0.83) 3.00 (0.75) 49.184* .330
42. Seems overly sensitive to sounds, textures, or smells 2.78 (1.08) 3.55 (0.64) 18.815* .158
43. Does not separate easily from caregiver 2.37 (1.02) 3.23 (0.76) 23.404* .190
44. Doesn’t understand how events relate to one another 2.49 (1.03) 3.37 (0.69) 25.895* .206
45. Does not focus attention to where others are looking 2.61 (0.90) 3.20 (0.66) 14.187* .124
46. Has overly serious facial expressions 2.29 (1.03) 3.10 (0.81) 19.366* .162
47. Is too silly or laughs inappropriately 2.33 (0.97) 3.35 (0.72) 36.325* .266
48. Has no sense of humour, does not understand jokes 2.31 (0.910 3.27 (0.70) 36.138* .265
49. Does extremely well at a few tasks 2.76 (0.97) 3.39 (0.70) 14.074* .123

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Journal of Autism and Developmental Disorders

Table 3  (continued) N = 51 males and females Males Females F Partial


SRS-2 item (abbreviated; reverse scoring removed) M (SD) M (SD) eta
squared

50. Has repetitive, odd behaviours such as hand flapping 1.88 (1.05) 2.84 (0.81) 26.737* .211
51. Has difficulty answering questions directly 2.33 (0.86) 3.35 (.69) 43.473* .303
52. Does not know when he/she is talking too loudly 3.27 (0.94) 2.67 (0.71) 13.558* .119
53. Talks to people with an unusual tone of voice 1.92 (1.02) 2.92 (0.84) 29.185* .226
54. Seems to react to people as if they are objects 1.65 (0.82) 2.57 (0.70) 37.239* .271
55. Does not know when too close to others 3.13 (0.96) 2.86 (0.83) 2.400 .023
56. Walks between two people talking 2.76 (1.03) 3.41 (0.75) 13.095* .116
57. Gets teased a lot 2.49 (0.950 3.29 (0.81) 21.311* .176
58. Concentrates too much on parts of things 2.41 (0.92) 3.39 (0.78) 33.802* .253
59. Is overly suspicious 1.98 (0.91) 2.75 (0.82) 19.971* .166
60. Is emotionally distant 2.00 (0.80) 2.90 (0.85) 30.280* .232
61. Is inflexible, difficult to change his mind 2.84 (0.83) 3.61 (0.60) 26.188* .220
62. Gives unusual or illogical reasons for doing things 2.33 (0.93) 3.25 (0.69) 32.314* .244
63. Touches others in an unusual way 1.63 (0.87) 2.71 (0.83) 40.900* .290
64. Is too tense in social situations 2.31 (0.88) 3.35 (0.72) 42.612* .299
65. Stares or gazes off into space 1.75 (0.72) 3.11 (0.71) 94.231* .485

*p < .001

ADOS-2 severity). There was one SRS-2 item which showed social behaviour. There were also some differences in being
a meaningful difference in Table 4 but not in Table 3 (SRS-2 aware of social cues, being motivated to engage in social
item 26: Does not offer comfort to others when they are sad) interaction, and behaving in ways that appear to be bizarre.
which is underlined in Table 6. These 21 items are shown These widespread differences (i.e., across all five SRS-2
in Table 6, and indicate that the most common sex differ- Treatment Subscales) argue for a general sex difference
ences fell into the SRS-2 Treatment Scale for Social Com- across the range of ASD traits. The finding that females
prehension, followed by Social Cognition and Restricted and consistently had higher (i.e., worse) scores than males, and
Repetitive Behaviours. These 21 SRS-2 items represent the that there was no significant effect due to age, suggests that
change in sex differences that were due to ASD severity females’ camouflaging of their ASD behaviour may com-
as assessed via the ADOS-2. All sex differences in either mence before school age, so that, in order to be identified
set of participants were in the direction of females showing as ASD, females might usually exhibit the wider range of
greater severity than males on the SRS-2 scores given by ASD-related behaviours more severely than males, despite
their parents. being matched on a standardised screening instrument (i.e.,
the ADOS-2). The latter instrument is applied by clini-
cians, whereas the SRS-2 is completed by parents, and it
Discussion may be that the lack of agreement between the matching via
ADOS-2 Scores versus the SRS-2 Scores could be a func-
Few previous studies have provided a direct comparison of tion of the source of information, as well as the greater detail
the sex differences in detailed ASD-related behaviour when provided by the SRS-2. Instrumentation was not a focus of
males and females were matched for ASD severity as well as this study, but these data suggest that it might repay future
for age and IQ. The current results identified those specific research. It should also be mentioned that parents may have
ASD-related behaviours that were found to be significantly expectations of greater levels of social interactive behaviour
and meaningfully more severe in females than in males, from their female offspring than from their male offspring,
as rated by their parents. Those SRS-2 items represented and this may have resulted in parents rating their daughters
difficulties in expressive social communication aspects of more severely than parents rated their sons on the SRS-2.
reciprocal social behaviour plus the individual’s willingness The finding of 21 SRS-2 items that exhibited sex dif-
to engage in social communication. (Constantino and Gru- ferences when age and IQ were matched across males and
ber 2012, p. 77). However, when examined for their rela- females but not when ADOS-2 Scores were also matched,
tive power in defining sex differences, the strongest (largest plus one SRS-2 item that showed sex differences when
effect size) differences were found across ability to inter- samples were matched on age, IQ and the ADOS-2 but not
pret social cues once they had been noticed, plus reciprocal when they were matched solely on age and IQ (shown in

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Journal of Autism and Developmental Disorders

Table 4  Mean (SD) scores for N = 32 males and females Males Females F Partial
selected SRS-2 items showing eta
meaningful sex differences in SRS-2 item (abbreviated; reverse scoring removed) M (SD) M (SD)
squared
bold for subsample (32 males,
32 females) 1. More fidgety in social situations 2.66 (0.97) 3.69 (0.59) 26.313* .298
2. Facial expressions don’t match what he/she is saying 2.03 (0.82) 2.91 (0.73) 20.153* .245
3. Is not self-confident when interacting 2.56 (0.95) 2.94 (0.76) 3.049 .047
4. When under stress, shows inflexible behaviour 2.75 (0.98) 3.59 (0.61) 16.928* .214
5. Doesn’t recognise when others are taking advantage 3.28 (0.85) 3.69 (0.540 5.223 .078
6. Would rather be alone than with others 2.16 (0.92) 3.13 (0.71) 22.315* .265
7. Is unaware of what others are thinking or feeling 3.03 (0.970 3.00 (0.72) 0.022 .000
8. Behaves in ways that seem strange or bizarre 2.34 (0.79) 3.22 (0.71) 21.895* .261
9. Clings to adults, seems too dependent on them 2.25 (1.16) 3.19 (0.860 13.439 .178
10. Takes things too literally 2.84 (0.88) 3.56 (0.62) 14.198* .186
11. Has poor self-confidence 2.75 (0.92) 2.97 (0.86) 0.969 .015
12. Is unable to communicate his/her feelings to others 2.59 (0.87) 3.09 (0.64) 6.806 .099
13. Is awkward in turn-taking interactions 2.59 (0.91) 3.31 (0.78) 11.492 .156
14. Is not well coordinated 2.44 (0.91) 2.97 (0.86) 5.732 .085
15. Unable to understand meaning of others’ voice and face 2.97 (0.78) 3.19 (0.64) 1.491 .023
16. Avoids eye contact or has unusual eye contact 2.50 (1.05) 3.16 (0.81) 7.880 .113
17. Does not recognise when something is unfair 2.25 (0.95) 3.31 (0.69) 26.120* .296
18. Has difficulty making friends 2.72 (1.02) 3.38 (0.71) 8.906 .126
19. Gets frustrated trying to get ideas across 2.84 (0.88) 3.69 (0.54) 21.340* .256
20. Shows unusual sensory interests 2.03 (1.06) 3.06 (0.84) 18.559* .230
21. Unable to imitate others’ actions 2.47 (0.76) 3.41 (0.71) 25.881* .295
22. Plays inappropriately with children his/her age 2.63 (0.87) 3.19 (0.74) 7.774 .111
23. Does not join group activities unless told to 2.66 (0.90) 3.06 (0.76) 3.799 .058
24. Has difficulty with changes in routine 3.03 (0.97) 3.63 (0.61) 8.642 .122
25. Doesn’t mind being out of step with others 2.53 (0.95) 3.16 (0.85) 7.721 .111
26. Does not offer comfort to others when they are sad 2.69 (0.86) 3.47 (0.72) 15.587* .201
27. Avoids starting social interactions 2.22 (0.94) 3.13 (0.75) 18.117* .226
28. Thinks or talks about the same thing over and over 3.16 (0.88) 3.41 (0.71) 1.552 .024
29. Is regarded by other children as odd or weird 3.13 (0.87) 3.56 (0.62) 5.367 .080
30. Becomes upset in a situation with lots going on 2.97 (1.09) 3.72 (0.52) 12.281 .165
31. Can’t get his/her mind off something 3.28 (0.85) 3.81 (0.470 9.541 .133
32. Does not have good personal hygiene 2.13 (0.87) 3.34 (0.75) 36.186* .369
33. Is socially awkward 2.84 (0.81) 3.38 (0.66) 8.303 .118
34. Avoids people who want to be emotionally close 2.00 (0.92) 2.81 (0.74) 15.274* .198
35. Has trouble keeping up with the flow of conversation 2.59 (0.98) 3.19 (0.69) 7.842 .112
36. Has difficulty relating to adults 1.91 (0.89) 2.66 (0.65) 14.708* .192
37. Has difficulty relating to peers 2.63 (0.87) 3.50 (0.57) 22.672* .268
38. Responds inappropriately to mood changes in others 2.63 (0.91) 2.84 (0.68) 1.195 .019
39. Has an unusually narrow range of interests 2.94 (1.01) 3.06 (0.80) 0.300 .005
40. Is unimaginative, poor at pretending 2.47 (1.02) 3.38 (0.75) 16.469* .210
41. Wanders aimlessly from one activity to another 1.97 (0.86) 2.72 (0.73) 14.149* .186
42. Seems overly sensitive to sounds, textures, or smells 2.72 (1.11) 3.56 (0.67) 13.492 .179
43. Does not separate easily from caregiver 2.31 (0.93) 3.13 (0.79) 14.121* .186
44. Doesn’t understand how events relate to one another 2.63 (0.98) 3.25 (0.80) 7.828 .112
45. Does not focus attention to where others are looking 2.72 (0.81) 3.31 (0.64) 10.488 .145
46. Has overly serious facial expressions 2.19 (0.99) 2.97 (0.82) 11.679 .159
47. Is too silly or laughs inappropriately 2.22 (1.10) 3.31 (0.74) 21.837* .260
48. Has no sense of humour, does not understand jokes 2.16 (0.85) 3.41 (0.67) 43.130* .410
49. Does extremely well at a few tasks 3.03 (0.78) 3.34 (0.79) 2.537 .039

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Journal of Autism and Developmental Disorders

Table 4  (continued) N = 32 males and females Males Females F Partial


SRS-2 item (abbreviated; reverse scoring removed) M (SD) M (SD) eta
squared

50. Has repetitive, odd behaviours such as hand flapping 2.03 (1.09) 2.72 (0.81) 8.163 .116
51. Has difficulty answering questions directly 2.56 (0.84) 3.22 (0.79) 10.333 .143
52. Does not know when he/she is talking too loudly 3.19 (0.99) 2.78 (0.75) 3.387 .052
53. Talks to people with an unusual tone of voice 2.28 (1.05) 2.75 (0.80) 4.002 .061
54. Seems to react to people as if they are objects 1.84 (1.05) 2.44 (0.67) 7.272 .105
55. Does not know when too close to others 3.25 (0.98) 2.97 (0.86) 1.481 .023
56. Walks between two people talking 2.69 (1.15) 3.34 (0.79) 7.109 .103
57. Gets teased a lot 2.53 (0.98) 3.25 (0.760 10.683 .147
58. Concentrates too much on parts of things 2.44 (0.91) 3.28 (0.81) 15.239* .197
59. Is overly suspicious 1.78 (0.79) 2.72 (0.81) 21.831* .260
60. Is emotionally distant 1.94 (0.80) 2.88 (0.87) 20.101* .245
61. Is inflexible, difficult to change his mind 2.72 (1.050 3.66 (0.60) 19.083* .235
62. Gives unusual or illogical reasons for doing things 2.34 (1.00) 3.13 (0.71) 12.960 .173
63. Touches others in an unusual way 1.81 (1.03) 2.56 (0.80) 10.578 .146
64. Is too tense in social situations 2.28 (0.92) 3.38 (0.66) 29.691* .324
65. Stares or gazes off into space 2.00 (0.84) 2.94 (0.80) 20.821* .251

*p < .001

Table 5  Twenty-seven SRS-2 items (and their SRS-2 Treatment Subscale) that showed meaningful sex differences for males and females
matched for age, IQ and ASD severity
SRS-2 Treatment Social Awareness Social Cognition Social Communica- Social Motivation Restricted and Repeti-
Subscale N=2 N=6 tion N=8 tive Behaviours
N=8 N=3

Items showing mean- Expressions on face Taking things literally Gets frustrated More fidgety Shows inflexible behav-
ingful sex differ- don’t match state- Recognises unfair- Unable to imitate Would rather be iour under stress
ences ments ness others alone Behaves strangely or
Poor personal Unimaginative Doesn’t offer comfort Clings to adults bizarrely
hygiene No sense of humour Difficulty relating to Avoids starting social Shows unusual sensory
Concentrates on parts adults interactions interests
Overly suspicious Difficulty relating to Avoids emotional
peers closeness
Wanders aimlessly Does not separate
Is too silly from caregiver
Emotionally distant Is too tense in social
Inflexible settings
Gazes into space

Items with a large effect size are in bold

Table 6), provides some evidence for the hypothesis that might also provide a similar confound when testing for sex
ASD severity influences sex difference data, and comple- differences in ASD-related behaviours.
ments previous studies that used only age and IQ as match- The finding that the females in the current sample had
ing variables. Although the general direction of sex differ- significantly higher ASD-related behaviour scores on the
ence was consistent across the two samples (i.e., females SRS-2 than the males is in contradiction of some of the
had more severe selected SRS-2 item scores), the specific previous studies in this field which found more severe
ASD-related behaviours identified within each sample ASD traits for males (e.g., Rynkiewicz et al. 2016) (which
were different. The comment by van Wijngaarden-Cremers did not match males and females on ADOS-2 Scores) but
et al. (2014) from their meta-analysis of 22 sex difference does confirm previous studies reporting that females have
studies, that IQ might confound sex difference findings, more severe ASD traits than males (e.g., Frazier et al.
could now be augmented by suggesting that ASD severity 2014) (in which males and females did not have significant

13
Journal of Autism and Developmental Disorders

Table 6  Twenty-one SRS-2 items (and their SRS-2 Treatment Subscale) showing change in meaningful sex differences according to ASD-sever-
ity
SRS-2 Treatment Social Awareness Social Cognition N = 6 Social Communica- Social Motivation Restricted and
Subscale N=1 tion N = 9 N=0 Repetitive Behav-
iours N = 5

Items showing mean- Reacts to people as Doesn’t recognise Awkward in turn Not well coordinated
ingful sex differ- objects when others are try- taking Regarded as odd or
ences ing to take advantage Difficulty making weird
Becomes upset in a friends Can’t get his or her
complex situation Socially awkward mind off something
Overly sensitive to Has trouble keeping Repetitive, odd
sounds, textures, up with conversation behaviours
smells Overly serious facial Touches others in an
Doesn’t understand expressions unusual way
how events relate Difficulty answering
Gives unusual or questions directly
illogical reasons for Talks to people with
doing things an unusual tone of
Plays appropriately voice
with others Gets teased a lot
Doesn’t offer comfort

differences on ADOS-2 diagnostic criteria scores). The sex differences may be considered to be more robust that
current results also do not agree with the overall findings if ADOS-2 matching had not been undertaken.
from van Wijngaarden-Cremers et al. (2014) meta-analysis These findings hold implications for clinical settings,
of 22 studies in which males tended to show more evi- where the identification and assessment of females with
dence of restricted and repetitive behaviours than females ASD may be relatively difficult compared to males, princi-
but there were no significant differences in social behav- pally because of the females’ ability to disguise their ASD
iour and communication. Instead, the sex differences found behaviour by learning socially-appropriate behaviour despite
here when ASD severity was matched were across social not understanding or actually engaging in reciprocal social
behaviour and communication and also RRB. However, interaction at a level more than males. It may be that, as has
as noted above, van Wijngaarden-Cremers et al. (2014) been suggested by others, the 4:1 male:female prevalence
pointed to the potential for differences in IQ to confound ratio is a function of females’ ability to behave in ways that
these sex-differences in symptom profiles, and it may be appear neurotypical. If this is the case, then greater atten-
that the process of controlling for IQ and ASD severity tion must be paid to those females who may exhibit some
was the underlying factor in these different results between behaviours that cause concern to those who live beside them.
the current study and some previous reports. The current
findings do agree with those reported by Allely (2018)
from her review about camouflaging, in that the females Limitations
in this study did not show significant differences in the
formal ASD diagnosis process via the ADOS-2 but did There are several limitations to the generalisability of the
exhibit greater severity in ASD traits when SRS-2 data current findings, including age, geographic and cultural
from their parents were analysed. That is, the females in source of the participants, the collection of SRS-2 data at
this sample may have only been identified as ASD when a single point in time, and the use of the SRS-2 rather than
they had a higher level of ASD-related behaviour than the another scale of ASD-related behaviour. While none of these
males because they had effectively camouflaged it from are sources of invalidity, they limit the applicability of these
observations by the clinicians who assessed them during findings to data from other populations, times, and instru-
intake but not from their parents who observed them on ments. Although there were no significant associations found
a daily basis. Finally, it has been suggested that matching for menarche or the presence of other psychiatric conditions,
males and females on ASD severity may also mask sex dif- the possibility of a confound between these factors and sex
ferences in ASD symptomatology (Postorino et al. 2015), differences in other populations must always be considered.
but that was not found here. Instead, matching males and Strengths of this study include its statistical power, the use
females on the standard assessment instrument allowed of a conservative yardstick for meaningful effects in the
for identification of those ASD-related behaviours that are form of the two-step (p < .001, plus a large ES) procedure
differently expressed by males and females. As such, these which effectively reduced the likelihood of a Type I error,

13
Journal of Autism and Developmental Disorders

a well-established measure of ASD-related behaviours, and Ethical Approval All procedures performed in studies involving human
matching for age, IQ, and ASD severity. As in all research, participants were in accordance with the ethical standards 1964 Hel-
sinki declaration and its later amendments or comparable ethical stand-
replication of these findings in other, and larger, samples is ards.
needed for firm conclusions to be drawn. Similarly, although
the ADOS-2 is widely used for diagnosis, and therefore is Informed Consent Informed consent was obtained from all individual
the relevant instrument here for the process of identifying participants included in the study.
the presence of ASD, the Autism Diagnostic Interview
(revised) might provide alternative valuable adjunct data.

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