Journal of Autism and Developmental Disorders (2019) 49:1570–1585

https://doi.org/10.1007/s10803-018-3843-z

ORIGINAL PAPER

Gender Variance and the Autism Spectrum: An Examination

of Children Ages 6–12 Years
A. Natisha Nabbijohn1 · Anna I. R. van der Miesen2 · Alanna Santarossa1,7 · Diana Peragine1 · Annelou L. C. de Vries2 ·
Arne Popma2 · Meng‑Chuan Lai1,3,4,5 · Doug P. VanderLaan1,6

Published online: 13 December 2018

© Springer Science+Business Media, LLC, part of Springer Nature 2018

Abstract
Gender variance (GV) and autism spectrum disorder (ASD) frequently co-occur in clinical populations. We investigated GV
in association with ASD characteristics in nonclinical children and in children with developmental/mental health diagnoses.
In 6–12-year-olds (N = 2445; 51% birth-assigned boys), the Gender Identity Questionnaire for Children measured GV and
the Children’s Social Behavior Questionnaire measured six subdomains of ASD characteristics. Among nonclinical children,
GV was associated with parent-reported difficulties orienting socially and stereotyped behaviors. GV was also associated
with parent-reported clinical diagnoses of ASD, sensory processing disorder, and oppositional defiant disorder. These find-
ings suggest associations between specific ASD characteristics and GV in nonclinical children. Also, childhood GV should
be further examined in a range of clinical populations, including ASD individuals.

Keywords Gender variance · Gender dysphoria · Autism spectrum disorder · Sensory processing disorder · Oppositional
defiant disorder

In recent years, there has been strong clinical and research autism spectrum disorder (ASD) (for review, see van der
interest in the overlap between gender variance (GV) and Miesen et al. 2016). GV is a term that is often used in the
current literature to describe an individual’s variation in gen-
der role behaviors, deviating from culturally specific gender
* Doug P. VanderLaan norms (Adelson 2012). GV does not imply having distress
doug.vanderlaan@utoronto.ca and, thus, is different from gender dysphoria (GD; Adelson
A. Natisha Nabbijohn 2012). In the fifth edition of the Diagnostic and Statistical
aysha.nabbijohn@mail.utoronto.ca Manual of Mental Disorders (DSM-5), GD refers to sig-
1
Department of Psychology, University of Toronto nificant levels of distress associated with the incongruence
Mississauga, Deerfield Hall, 3359 Mississauga Road, Office between one’s birth-assigned gender (i.e. male or female)
4098, Mississauga, ON L5L 1C6, Canada and one’s experienced gender (American Psychiatric Asso-
2
Department of Child and Adolescent Psychiatry, Center ciation; APA 2013). Although a number of reports suggest
of Expertise on Gender Dysphoria, VU University Medical that childhood GV does not always persist as GD later in
Center, Amsterdam, The Netherlands life (de Vries et al. 2010; Steensma et al. 2011; Wallien and
3
Department of Psychiatry, Centre for Addiction and Mental Cohen-Kettnis 2008), high levels of GV are often predictive
Health and The Hospital for Sick Children, University of a potential GD diagnosis (Coleman et al. 2012). In the
of Toronto, Toronto, ON, Canada
DSM-5 (APA 2013), ASD is classified as a neurodevelop-
4
Department of Psychiatry, Autism Research Centre, mental condition. The DSM-5 criteria for an ASD diagnosis
University of Cambridge, Cambridge, UK
includes significant impairments in social communication
5
Department of Psychiatry, National Taiwan University and interaction (i.e. difficulties in social-emotional reciproc-
Hospital and College of Medicine, Taipei, Taiwan
ity, nonverbal expression and understanding, understanding
6
Child and Youth Psychiatry, Centre for Addiction and Mental and maintaining social relationships) as well as restricted,
Health, Toronto, Canada
repetitive patterns of behaviors, specific interests, and idi-
7
Present Address: Faculty of Medicine, University of Toronto osyncratic sensory experiences (APA 2013).
Mississauga, Mississauga, ON, Canada

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Journal of Autism and Developmental Disorders (2019) 49:1570–1585 1571

Evidence suggests the prevalence of GD and ASD have and 27.1% scored in the severe range (Skagerberg et al. 2015).
been increasing independently in the general population Another study of children less than or equal to 12 years of age
(Lai et al. 2014; Loomes et al. 2017; Meerwijk and Seve- reported that 44.9% fell within the clinical range (VanderLaan
lius 2017; Zucker 2005, 2017). Recent prevalence rates of et al. 2015).
GD, transgender identity, or GV, among adolescent and A recent clinical study used the Children’s Social Behavior
adult populations are estimated to be 0.2–1.1% (Flores Questionnaire (CSBQ; Hartman et al. 2006) to assess various
et al. 2016; Kuyper and Wijsen 2014; Meerwijk and Seve- domains of ASD characteristics in a sample of children and
lius 2017); however, the prevalence of individuals referred adolescents with GD in comparison to a typically develop-
to or undergoing treatment at specialty gender identity clin- ing control group and a group diagnosed with ASD (van der
ics is approximately 1:14,705 in birth-assigned males and Miesen et al. 2018a). The domains examined included: (1)
1:38,461 in birth-assigned females (Arcelus et al. 2015; the extent of situation-appropriate behavior and emotions, (2)
Zucker 2017). With regards to ASD, recent studies esti- responses to social situations, (3) orientation in time and place,
mated a prevalence rate of approximately 1–1.5% in the (4) understanding social information, (5) stereotyped move-
general population, with males being 3–4 times more likely ments and atypical responses from these senses, and (6) resist-
to be diagnosed with ASD (Christensen et al. 2016; Loomes ance to change. Overall, children and adolescents with GD
et al. 2017; Ofner et al. 2018). Interestingly, although GD had more ASD characteristics across all domains compared
and ASD are two distinct conditions and appear to reflect to typically developing children and adolescents but less than
a minority of the general population, various quantitative children and adolescents with an ASD diagnosis.
studies reported elevated ASD characteristics among those While the aforementioned studies focused on individuals
clinically referred for GD as well as elevated GV among with GD, other studies have examined whether individuals
individuals diagnosed with ASD (for review, see van der diagnosed with ASD exhibit increased GV. To date, three
Miesen et al. 2016). quantitative studies examined GV in clinical samples of
children and adolescents 6–18 years of age with ASD using
parental endorsement of the “wishes to be of the opposite sex”
Gender Variance and ASD Characteristics item (Item 110) in the most recent version of Child Behav-
in Prior Clinical Studies ior Checklist (CBCL; Achenbach and Rescorla 2001). Strang
et al. (2014) compared children and adolescents with ASD to
Among recent quantitative studies, de Vries et al. (2010) those diagnosed with attention-deficit/hyperactivity disorder
conducted the only systematic study on the prevalence of (ADHD), and other neurodevelopmental disorders as well as
ASD diagnoses among children (i.e. under the age of 12) two typically developing groups. Significantly higher rates of
and adolescents (i.e. between 12 and 18 years of age) with GV were found among children with ASD (5.4%) and ADHD
GD. Using the Dutch version of the Diagnostic Interview (4.8%) compared to two typically developing groups (0–0.7%).
for Social and Communication Disorders-10th revision No significant differences were found between the other neu-
(DISCO-10; Wing 1999) to assess ASD diagnoses, this rodevelopmental group (1.7%) and the typically developing
study reported an ASD rate of 6.4% and 9.4% for the pre- groups. Janssen et al. (2016) found that 5.1% of children and
pubescent child and the pubescent adolescent samples, adolescents diagnosed with ASD reported more GV compared
respectively. These rates were considered higher than the to 0.7% of the typically developing sample. A more recent
estimated 1–1.5% population prevalence rate (Christensen study also found a significantly higher rate of GV among chil-
et al. 2016; de Vries et al. 2010; Ofner et al. 2018). dren and adolescents diagnosed with ASD (4.0%) compared to
Another approach has been to examine the presence of a typically developing group (0.7%), but not compared to those
ASD-related traits (i.e. ASD characteristics or autistic(-like) clinically referred for other mental health concerns (4.0%; May
traits) among individuals with GD. Using the Autism-Spec- et al. 2017). These findings are paralleled by adolescent and
trum Quotient (AQ; Baron-Cohen et al. 2001), two adult stud- adult studies showing greater gender and sexual orientation
ies found elevated ASD characteristics among individuals diversity among samples of individuals with ASD (Dewinter
with GD relative to typically developing adults (Jones et al. et al. 2017; George and Stokes 2017, 2018a, b; van der Miesen
2012; Pasterski et al. 2014), and one of these studies reported et al. 2018b).
elevated ASD characteristics among birth-assigned females
with GD compared to birth-assigned males with GD (Jones
et al. 2012). Among children and adolescents with GD, stud-
ies examined clinical-range (mild/moderate or severe) ASD
scores on the Social Responsiveness Scale (SRS; Constantino
and Gruber 2005). In one sample of children and adolescents
5–18 years of age, 27.1% scored in the mild/moderate range

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1572 Journal of Autism and Developmental Disorders (2019) 49:1570–1585

Gender Variance and ASD Characteristics e.g. Baams et al. 2013; Landolt et al. 2004) that result in
in Nonclinical Studies elevated scores on measures of social impairment. In addi-
tion, a small number of case-reports and quantitative stud-
Several community studies suggested that, independently, ies point to other clinical populations in which GV may be
both GV and ASD characteristics are not uncommon in elevated. For instance, Strang et al. (2014) explored rates
nonclinical populations. Nevertheless, no study has of GV as measured by parental endorsement of cross-sex
investigated the relation between GV and ASD charac- wishes on the CBCL in children with ASD, ADHD, or
teristics in nonclinical, community samples to date. ASD other neurodevelopmental conditions. Childhood GV was
characteristics are continuously distributed in the general associated with ASD and ADHD; but, the latter finding
population (Baron-Cohen et al. 2014; Constantino and has not been replicated.
Todd 2003; Hoekstra et al. 2007; Hurst et al. 2007). For Further, based on past literature, one might expect an
example, Constantino and Todd (2003) used the SRS to over-representation of gender-variant individuals among
quantitatively measure ASD characteristics in twins aged other clinical populations, especially those who experience
7–15 years old from the general population and found internalizing conditions (e.g. anxiety or depression). Previ-
ASD characteristics to be continuously distributed, with ous studies suggest children who exhibit GV are more likely
1.4% of boys and 0.3% of girls scoring within the clinical to experience peer difficulties, including ridicule, correc-
range for ASD. With regard to GV, van Beijsterveldt et al. tions, and rejection (e.g. Berndt and Heller 1986; Carter
(2006) used the earlier version of the CBCL (Achenbach and McCloskey 1984; Wallien et al. 2010; Zucker et al.
1991) to measure parent-reported endorsement of Item 5 1995; Zucker 2005). Indeed, there is evidence to suggest
(i.e. “behaves like the opposite sex”) and Item 110 among that experienced social stigma was associated with poorer
a nonclinical population of children. They reported 3–5% psychological well-being, including distress, depression and
of the girls and 2–3% of the boys behaved like the opposite anxiety among transgender and/or gender-variant individu-
sex, and approximately 1% of boys and girls wished to be als (Baams et al. 2013; Bockting et al. 2013; Coolidge et al.
of the opposite sex. Other child- and parent-report studies 2002; de Vries et al. 2016; Landolt et al. 2004; Li et al. 2016;
also provided prevalence estimates ranging between 2.3 Lippa 2008; Shiffman et al. 2016; Toomey et al. 2010). Cor-
and 6% for GV in the community (Coolidge et al. 2002; respondingly, several studies found that associations between
Martin et al. 2017). Using a similar approach in a young GD/GV and internalizing challenges in youth were medi-
adult university-based sample, Lai et al. (2010) reported ated by poor peer relationships (e.g. Coolidge et al. 2002; de
7.3% of birth-assigned females and 1.9% of birth-assigned Vries et al. 2016; Landolt et al. 2004; Shiffman et al. 2016;
males reported “I wish I was the opposite sex.” Taken van Beijsterveldt et al. 2006). Given these patterns, it may
together, both GV and ASD characteristics are not spe- be relatively more common for individuals who express GV
cific to only clinical populations. As such, GV and ASD to be at risk for stigma-related psychological distress and,
characteristics may be associated more generally, rather therefore, to have received a mental health diagnosis such
than simply within the clinical contexts in which they have as anxiety or depression. Based on small clinic samples and
been studied to date. individual cases, it has also been hypothesized that conduct
disorders, mood/anxiety disorders, obsessive–compulsive
disorder, heightened sensory sensitivities, and learning dis-
abilities may be associated with increased GV (e.g. Kalti-
Gender Variance and Clinical Diagnoses ala-Heino et al. 2015; Landen and Rasmussen 1997; Tateno
Other than ASD et al. 2008; Williams et al. 1996; Wood and Halder 2014);
however, these hypotheses are best regarded as highly tenta-
While a number of studies support the potential associa- tive in the absence of more rigorous empirical data.
tion between GD/GV and ASD/ASD characteristics, Tur-
ban and van Schalkwyk (2018) note that the specificity of
this correlation has not been sufficiently established as few The Present Study
studies have examined GV among other clinical popula-
tions. As such, there is insufficient evidence to suggest that The overall goal of the present study was to advance our
elevated GD/GV is specific to ASD. For example, there understanding of the association between GV and develop-
might be other emotional challenges (e.g. body dissatis- mental/mental health conditions, with particular attention to
faction; van de Grift et al. 2017) and environmental fac- ASD. In comparison to previous studies examining the GV-
tors associated with GV such as those relating to minority ASD link, this study focused on parent-reported responses
stress (e.g. poor peer relations, familial non-acceptance; for a relatively younger sample of children between 6 and
12 years of age (N = 2445). The use of online recruitment

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Journal of Autism and Developmental Disorders (2019) 49:1570–1585 1573

also allowed for obtaining a large community-based sam- Parent-reported responses were collected for a total
ple that provided considerably higher statistical power than sample of 3,097 children; however, 652 participants were
previous studies in this field. Another novel aspect of the excluded because they did not provide adequate information
present study included the use of more comprehensive meas- on the focal measure of GV investigated in this study and/
ures for both GV and ASD. In particular, whereas previous or key demographic variables such as the child’s age and
studies examining GV in relation to ASD relied on a single- birth-assigned gender. Thus, the final sample included par-
item measure of cross-sex wishes (e.g. Janssen et al. 2016; ent-reports on 2445 children. Of the total responses included
May et al. 2017; Strang et al. 2014), this study employed the in this study, 96% of the data were obtained from maternal
Gender Identity Questionnaire for Children (GIQC; Johnson reports (n = 2347) and the remaining 4% (n = 98) were from
et al. 2004)—a well-validated and highly sensitive measure paternal reports; and 5.4% of the children (n = 132) lived in
of GV that relates to the DSM criteria for GD and assesses the same household (e.g. siblings).
several gender-typed domains relevant to children (e.g. toy, Parents were asked whether their child had received a
playmate, activity, clothing and hairstyle preferences). Like- developmental/mental health diagnosis from a healthcare
wise, the CSBQ was employed to examine potential associa- professional, and if they indicated “yes” then they were
tions between distinct domains of parent-reported ASD char- asked to list all such diagnoses. Children (Mean age = 8.83
acteristics and GV. In addition, the present study reports on years, SD = 1.99) were categorized as either typically devel-
levels of GV in relation to a variety of developmental/mental oping (i.e. from the nonclinical subgroup; n = 2004; 1022
health diagnoses to provide insight regarding the specificity girls, 982 boys) or as part of a clinical subgroup of the popu-
of the association between GV and ASD. lation meaning that they had (according to parent-report) at
least one of eight categories of developmental/mental health
diagnoses (n = 441; 165 girls, 276 boys): autism spectrum
Method disorder (ASD; n = 80; 23 girls, 57 boys), attention-def-
icit/hyperactivity disorder (ADHD; n = 247; 81 girls, 166
Participants and Procedure boys), oppositional defiant disorder (ODD; n = 39; 16 girls,
23 boys), obsessive–compulsive disorder (OCD; n = 17; 4
Parents of children (6–12 years of age) were recruited to girls, 13 boys), sensory processing disorder1 (SPD; n = 28;
participate in a parent-report online questionnaire from June 8 girls, 20 boys), mood and anxiety disorders (n = 140; 64
to December 2016. To participate in this study, parents were girls, 76 boys), learning disabilities (n = 51; 25 girls, 26
required to be a minimum age of 18 years, to be proficient in boys) and other neurodevelopmental conditions for which
English, and to have at least one child aged 6–12 years. Par- participant numbers were relatively small (n = 22; 6 girls,
ticipant recruitment was conducted online using Facebook 19 boys). This latter category included Tourette syndrome
and Kijiji, as well as by contacting Canadian organizations (n = 8; 2 girls, 6 boys), developmental coordination disorder
and facilities (e.g. recreational community centers). As part (n = 2; 2 boys), developmental delay disorder (n = 2; 2 boys),
of a larger study on gender expression and psychological speech sound disorder (n = 1; 1 boy), fetal-alcohol spectrum
well-being, advertisements used to recruit parents for the disorder (n = 2; 1 girl, 1 boy), executive functioning disorder
study included the tag-line “Survey on Child Well-Being.” (n = 3; 3 boys), and auditory processing disorder (n = 4; 2
A majority of participants who filled out the questionnaire girls, 2 boys).
reported that they were recruited via Facebook (79.3%)
where study advertisements were viewed by 182,887 people,
and the survey link was clicked 3119 times. Of the remaining
participants who completed the questionnaire, 68 reported
they were recruited from Kijiji (1.1%) or other sources (i.e.
word of mouth, community organizations, local advertise- 1
Although SPD is not included in the DSM, it is included here for
ments; 1.1%), and 573 participants (18.5%) did not report two reasons: (a) it has been conceptualized in the child development
where they learned about the study. Participants were given literature as a complex condition involving hyper- or hypo-sensitivity
a link to an anonymous survey that was administered using to sensory stimuli, a heightened drive for sensory experiences, dif-
Qualtrics Survey Software and were required to provide ficulty discriminating sensations, and atypical responses to sensory
stimuli that, together, impact daily functioning (Miller and Schaaf
informed consent before completing the survey online. Once 2008), and (b) previous authors have forwarded the hypothesis that
the survey was complete, participants were compensated for elevated GV might be associated with altered sensitivity towards cer-
their time by being given the opportunity to enter a prize tain sensory input (e.g. tactile sensations) that intersects with gender
draw with a 1-in-100 chance of winning a $100 e-gift card of expression (de Vries et al. 2010; Tateno et al. 2008; Williams et al.
1996). This latter consideration is particularly relevant to the inclu-
their choice (e.g., Amazon). The present study was approved sion of SPD in the present study because it allows for a quantitative
by the University of Toronto Research Ethics Board. assessment of this previously proposed hypothesis.

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1574 Journal of Autism and Developmental Disorders (2019) 49:1570–1585

Measures questionnaire used to assess characteristics of ASD (Hart-

man et al. 2006). Parents were asked to rate their child’s
Demographic Information behaviors during the preceding 2 months on a 3-point Lik-
ert scale ranging from 0 (does not apply) to 2 (clearly or
Parents responded to questions regarding their own and often applies). The CSBQ has six subscales measuring
their child’s demographic characteristics. The demo- different domains:
graphic variables of interest included the child’s birth-
assigned gender (coded as females/girls = 0 and males/ 1. The not tuned subscale describes a child’s difficulty
boys = 1), age, average school performance (based on letter adapting their emotional and behavioral reactions appro-
grades coded as 1 = D, 2 = C, 3 = B, 4 = A), location (i.e. priately to social context. An example item is “quickly
Western Canada, Eastern Canada, Quebec, Ontario), area gets angry.”
of residence (i.e. rural, urban, suburban), parent marital 2. The social subscale included items characterizing
status, ethnicity, annual household income in Canadian responses to social contact, social needs, and initiation
dollars, and predominant religious background. of contact. A sample item is “does not begin to play with
Table 1 presents the demographic characteristics for other children.”
boys and girls separately for the clinical and nonclini- 3. The orientation subscale reflects the inability for a child
cal subgroups, respectively. Overall, girls had a higher to adequately orient self in time, place or in relation to
school performance, independent t(2200) = 3.07, p = 0.02, others. For example, “takes in information with diffi-
Cohen’s d = 0.13; however, the boys and girls did not sig- culty.”
nificantly differ on any other demographic variable (see 4. The understanding subscale assesses whether the child
Table 1). experiences any difficulty understanding social informa-
tion such as “takes things literally, e.g. does not under-
stand certain expressions.”
Gender Variance 5. The stereotyped subscale represents stereotyped behav-
ioral reactions, a preoccupation with objects and sensory
The Gender Identity Questionnaire for Children (GIQC) is stimuli, and atypical responses to information from the
a 16-item standardized parent-report questionnaire rated senses. For instance, a sample item includes “constantly
on a 5-point Likert scale and is used to assess gender- feels objects.”
typed behavior in children (Johnson et al. 2004). The 6. The change subscale refers to a fear of new situations
rating scale ranges from 1 (stereotypically opposite to and resistance to change, which are assessed using items
birth-assigned gender) to 5 (stereotypically same birth- such as “remains clammed up in new situations or if
assigned gender). Based on the factor analysis of Johnson change occurs.”
et al. (2004), items 8 and 16 were left out of the analysis as
the one-factor solution containing the remaining 14-items With respect to its psychometric properties, the CSBQ
was found to best fit the data, accounting for 43.7% of the has good test–retest and inter-rater reliability as well as
variance. Typically, a lower GIQC total score is indicative internal and discriminant validity when used on various
of increased GV; however, for the present study, scoring groups of children (i.e. PDD-NOS, high functioning autism
for the GIQC was reversed to allow for easier interpreta- and clinical controls; Luteijn et al. 2000). In terms of the
tion of the results (i.e. higher scores reflect elevated GV). validity of the subscales, Hartman et al. (2006) performed
The GIQC is psychometrically valid. For the present study, several factor analyses to support the discrete subscales; and
the Cronbach’s alphas were found to be 0.77 and 0.64 for items were removed to increase the distinctiveness of the
birth-assigned boys and girls, respectively. Additionally, subscales. Each subscale also had a high conceptual valid-
the GIQC has been found to show negligible age effects ity. Pertaining to the current study, Cronbach’s alpha for the
and is known to effectively encompass most of the core CSBQ total score was found to be 0.95 and ranged between
behavioral characteristics of those with GD (Johnson et al. 0.78 and 0.89 for each of the individual subscales. In addi-
2004). tion, unlike other instruments assessing ASD characteristics,
the CSBQ is not directly related to the DSM-criteria of ASD
(de Bildt et al. 2009). Further, each of the subscales showed
ASD Characteristics large effect size differences when comparing the neurotypi-
cal to the PDD-NOS and to high functioning autism groups,
The refined version of the Children’s Social Behav- respectively. Thus, this instrument and its subscales provide
ior Questionnaire (CSBQ) is a 49-item parent-report a more sensitive screening measure of milder expressions
of ASD and ASD characteristics that are present in the

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 Table 1  Descriptive statistics for demographic variables for the clinical and nonclinical subgroup of children
Variables Clinical Nonclinical
2 a
Girls Boys Total t- or χ -value p-value Girls Boys Total t- or χ2-value p-valuea

Age (in years) 1.2 ns − 0.0 ns

n 165 276 441 1022 982 2004
M (SD) 9.6 (2.0) 9.3 (1.9) 9.4 (1.9) 8.7 (2.0) 8.7 (2.0) 8.7 (2.0)
School ­performanceb − 1.3 ns 2.9 0.004
n 153 253 406 920 876 1796
M (SD) 2.8 (0.8) 2.9 (0.7) 2.9 (0.8) 3.4 (0.6) 3.3 (0.6) 3.3 (0.6)
Annual income, n (%)c 3.1 ns 6.0 ns
< $23,999 29 (17.6) 37 (13.5) 66 (15.0) 100 (9.8) 97 (9.9) 197 (9.9)
$24,000–49,999 41 (24.8) 69 (25.2) 110 (25.1) 233 (22.9) 183 (18.7) 416 (20.9)
$50,000–$79,999 30 (18.2) 62 (22.6) 92 (21.0) 224 (22.0) 225 (23.0) 449 (22.5)
$80,000–$124,999 43 (26.1) 63 (23.0) 106 (24.1) 251 (24.7) 264 (27.0) 515 (25.8)
> $125,000 16 (9.7) 29 (10.6) 45 (10.3) 165 (16.2) 169 (17.3) 334 (16.7)
Unknown 6 (3.6) 14 (5.1) 20 (4.6) 45 (4.4) 39 (4.0) 84 (4.2)
Parent’s marital status, n (%) 3.9 ns 0.5 ns
Journal of Autism and Developmental Disorders (2019) 49:1570–1585

Married/common-law 61 (37.0) 77 (28.0) 138 (31.4) 236 (23.1) 214 (21.8) 450 (22.5)
Other 104 (63.0) 198 (72.0) 302 (68.6) 786 (76.9) 768 (78.2) 1554 (77.5)
Area type, n (%) 1.8 ns 3.0 ns
Urban 56 (34.1) 80 (29.1) 136 (31.1) 321 (31.5) 304 (31.1) 625 (31.3)
Suburban 70 (42.7) 118 (42.9) 188 (42.8) 449 (44.1) 398 (40.7) 847 (42.4)
Rural 38 (23.2) 77 (28.0) 115 (26.2) 249 (24.4) 275 (28.1) 524 (26.3)
Geographic region, n (%) 3.5 ns 3.5 ns
Ontario 98 (59.4) 148 (53.6) 246 (55.8) 589 (57.9) 542 (55.3) 1131 (56.6)
Quebec 8 (4.8) 8 (2.9) 16 (3.6) 27 (2.7) 34 (3.5) 61 (3.1)
Eastern Canada 15 (9.1) 30 (10.9) 45 (10.2) 104 (10.2) 98 (10) 202 (10.1)
Western Canada 43 (26.1) 89 (32.3) 132 (29.9) 296 (29.1) 301 (30.7) 597 (29.9)
Northern Canada 1 (0.6) 1 (0.4) 2 (0.5) 2 (0.2) 5 (0.5) 7 (0.4)
Ethnicity, n (%) 16.1 0.041 7.4 ns
European Canadian 100 (60.6) 137 (50.0) 237 (54.0) 545 (53.7) 546 (56.3) 1091 (55)
South Asian 0 (0) 4 (1.5) 4 (0.9) 25 (2.5) 33 (3.4) 58 (2.9)
East Asian 1 (0.6) 0 (0) 1 (0.2) 18 (1.8) 11 (37.9) 29 (1.5)
African 0 (0) 0 (0) 0 (0) 3 (0.3) 3 (0.3) 6 (0.3)
Latin 1 (0.6) 1 (0.4) 2 (0.5) 15 (1.5) 9 (0.9) 24 (1.2)
Caribbean 3 (1.8) 1 (0.4) 4 (0.9) 11 (1.1) 15 (1.5) 26 (1.3)
Aboriginal 4 (2.4) 17 (6.2) 21 (4.8) 54 (5.3) 43 (4.4) 97 (4.9)

13
1575
 Table 1  (continued)
1576

Variables Clinical Nonclinical

2 a
p-value p-valuea

13
Girls Boys Total t- or χ -value Girls Boys Total t- or χ2-value

Arab 0 (0) 0 (0) 0 (0) 6 (0.6) 4 (0.4) 10 (0.5)

Other 21 (12.7) 57 (20.8) 78 (17.8) 134 (13.2) 128 (13.2) 262 (13.2)
Multiple 35 (21.2) 57 (20.8) 92 (21.0) 203 (20) 178 (18.4) 381 (19.2)
Religion, n (%) 17.6 0.024 7.7 ns
Roman Catholic 23 (13.9) 54 (19.6) 77 (17.5) 191 (18.7) 197 (20.2) 388 (19.4)
Protestant 10 (6.1) 32 (11.6) 42 (9.5) 77 (7.5) 61 (6.2) 138 (6.9)
Christian 48 (29.1) 65 (23.6) 113 (25.7) 252 (24.7) 247 (25.3) 499 (24.9)
Muslim 1 (0.6) 4 (1.5) 5 (1.1) 29 (2.9) 24 (2.5) 53 (2.7)
Jewish 0 (0) 4 (1.5) 4 (0.9) 13 (1.3) 7 (0.7) 20 (1.0)
Buddhist 0 (0) 2 (0.7) 2 (0.5) 5 (0.5) 6 (0.6) 11 (0.6)
Hindu 0 (0) 0 (0) 0 (0) 17 (1.7) 9 (0.9) 26 (1.3)
Sikh 0 (0) 1 (0.4) 1 (0.2) 3 (0.3) 4 (0.4) 7 (0.4)
No religion 58 (35.2) 93 (33.8) 151 (34.3) 380 (37.2) 357 (36.5) 737 (36.9)
Other 25 (15.2) 20 (7.3) 45 (10.2) 55 (5.4) 66 (6.7) 121 (6.1)

M mean, SD standard deviation

a
Birth-assigned boys and girls were compared for each demographic variable using independent samples t-tests or Chi-square tests
b
School performance includes average performance in the following subject areas: math, science, English, social science, and language studies. Response scale = A (coded as 4) to D (coded as
1)
c
The percentage (%) refers to the percent within the child’s birth-assigned gender for the Boys and Girls columns. In the total column, this value refers to the percent of the total clinical or non-
clinical subgroups (i.e. boys and girls included), respectively
Journal of Autism and Developmental Disorders (2019) 49:1570–1585
Journal of Autism and Developmental Disorders (2019) 49:1570–1585 1577

nonclinical population (de Bildt et al. 2009; Hartman et al. variables that were significantly correlated with the GIQC
2006; Luteijn et al. 2000). scores were identified as relevant control variables. All rel-
evant control variables were included in a step-wise multi-
Behavioral and Emotional Challenges ple linear regression examining unique associations between
various diagnostic categories and GIQC scores, relative to
The Child Behavior Checklist (CBCL) is a standardized the reference group of children who did not have a develop-
parent-report questionnaire used to assess a broad range mental/mental health diagnosis. In addition, for any diagno-
of behavioral and emotional challenges (Achenbach and sis that was found to have a significantly unique association
Rescorla 2001). The CBCL consists of 113-items, and par- with elevated GIQC scores, the possible moderating influ-
ents were asked to rate their child’s behavior over the past ence of gender was assessed by including their respective
2 months on a 3-point Likert scale ranging from 0 (not true) interactions with gender as predictor variables in an addi-
to 2 (very or often true) (Achenbach and Rescorla 2001). tional step in the regression model. Again, any participants
T-scores derived from the parents’ answers were calculated for whom data were missing for any of the control variables
as age-standardized measures of children’s total behavioral included in the regression were deleted pairwise from this
and emotional challenges. This measure has extensive nor- analysis.
mative data available, and it is a well-validated and reliable Statistical significance was initially evaluated at a con-
measure (Nakamura et al. 2009). The CBCL was found to ventional critical value of 0.05. For CSBQ subscales and
have a high internal consistency (α = 0.78–.97) and inter- developmental/mental health diagnoses that were unique
rater reliability (ICC = 0.95; Achenbach and Rescorla 2001). predictors of GIQC scores in the regression analyses, we
performed the Benjamini-Hochberg (1995) procedure for
Statistical Analysis multiple comparisons to inform the possibility of false dis-
coveries (i.e. Type I error). For these analyses, we set the
All statistical analyses were conducted using SPSS version false discovery rate (FDR) at 5%.
24. To assess the relationship between GV and ASD char-
acteristics in a nonclinical sample, relations between CSBQ
total and subscale scores and GIQC scores were examined.
Results
For the nonclinical sample, the analysis was limited to chil-
dren whose parents did not report a history of developmen-
Gender Differences in Gender Variance
tal/mental health diagnoses and completed all items pertain-
ing to the CSBQ (n = 1851; 949 girls, 902 boys). Zero-order
Overall, the mean GIQC score of girls (mean, M = 2.11,
correlations were conducted with the CSBQ total score, the
standard deviation, SD = 0.40) was significantly higher
six CSBQ subscales, and demographic variables including
than that of boys (M = 1.97, SD = 0.37; p < 0.05, Cohen’s
the child’s birth-assigned gender, age, annual income and
d = 0.36). In the nonclinical subgroup, the mean GIQC
school performance as well as the CBCL T-score. Doing so
score of girls (M = 2.10, SD = 0.39) was significantly higher
conveyed the correlation structure among these variables
than that of boys (M = 1.96, SD = 0.37; p < 0.05, Cohen’s
and also identified which demographic variables were asso-
d = 0.37). Similar results were found in the clinical subgroup
ciated with the GIQC and should, therefore, be included as
where the mean GIQC score of girls (M = 2.16, SD = 0.43)
control variables. Second, a multiple linear regression pre-
was significantly higher than that of boys (M = 2.01,
dicting GIQC scores was conducted in which all variables
SD = 0.35; p < 0.05, Cohen’s d = 0.38).
identified as needing to be controlled (Step 1) and each of
the CSBQ subscales (Step 2) were included as predictors. In
a third step, the interactions between gender and CSBQ sub- Gender Variance and ASD Characteristics
scales that predicted elevations in GIQC scores were entered in Nonclinical Children
as predictors. Participants for whom data were missing for
any of the control variables included in the regression were For the nonclinical subgroup, the mean total CSBQ score
deleted pairwise from this analysis. and the mean CSBQ subscale scores for boys and girls sepa-
For the analyses regarding GV and developmental/men- rately are presented in Table 2. Table 3 shows the zero-order
tal health diagnoses, the same approach was used. Zero- correlations between GIQC and CSBQ scores, as well as
order correlations were employed across the entire sample possible control variables including birth-assigned gender,
to examine whether GV was associated with each of the income, school performance, and CBCL total T-scores.
particular diagnoses as well as to determine whether birth- Higher CSBQ total scores and higher GIQC scores were both
assigned gender, age, annual income, or school perfor- associated with increased emotional and behavioral chal-
mance were associated with GIQC scores. The demographic lenges based on the CBCL total T-scores, and birth-assigned

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1578 Journal of Autism and Developmental Disorders (2019) 49:1570–1585

Table 2  Means and standard deviations for the CSBQ total and sub- assessed the possible moderating effect of gender in pre-
scales scores of birth-assigned boys and girls in the nonclinical sub- dicting GIQC scores on the orientation and stereotyped sub-
group of children
scales and found no significant moderating effects. Using the
ASD characteristics Boys (n = 902) Girls (n = 949) Benjamini-Hochberg procedure with a 5% FDR, the orien-
M SD M SD tation (p < 0.001) and not tuned (p = 0.003) subscale effects
remained significant relative to their respective correspond-
CSBQ total 18.1 12.3 17.3 12.0 ing adjusted critical alphas of 0.008 and 0.017. In contrast,
Not tuned 7.84 4.70 7.88 4.77 the p-value for the stereotyped subscale effect (p = 0.036)
Social 2.29 3.13 2.01 2.97 was not lower than the corresponding adjusted critical alpha
Orientation 2.81 2.66 2.57 2.63 (p = 0.025). Thus, at a 5% FDR, it may be that this latter
Understanding 2.60 2.70 2.35 2.53 effect is a case of Type I error.
Stereotyped 1.54 2.40 1.41 2.26
Change 1.05 1.34 1.08 1.35
Gender Variance and Clinical Diagnoses
M mean, SD standard deviation, CSBQ Children’s Social Behavior
Questionnaire
Table 5 shows the zero-order correlations between GV and
primary variables of interest in the clinical subgroup of chil-
Table 3  Zero order correlations among study variables for CSBQ total and subscales among the nonclinical subgroup
Variables 1 2 3 4 5 6 7 8 9 10 11 12

1. GIQC –
2. Gender − 0.19** –
3. Age − 0.01 0.00 –
4. Annual Income − 0.01 0.01 0.01 –
5. School performance 0.02 − 0.07** − 0.11** 0.00 –
6. CBCL T-score 0.09** 0.02 − 0.04 − 0.04 0.23** –
7. CSBQ total 0.11** 0.03 0.06* − 0.01 0.27** 0.74** –
8. Not ­tuneda 0.02 − 0.00 0.07** − 0.00 0.19** 0.65** 0.75** –
9. ­Socialb − 0.10** 0.05* − 0.09** − 0.02 0.15** 0.45** 0.72** 0.34** –
10. ­Orientationc 0.16** 0.04* − 0.05 − 0.01 0.32** 0.58** 0.79** 0.44** 0.52** –
11. ­Understandingd − 0.10** 0.05* − 0.06** − 0.01 0.24** 0.56** 0.78** 0.45** 0.46** 0.65** –
12. ­Stereotypede 0.11** − 0.03 − 0.13** − 0.02 0.11** 0.40** 0.64** 0.27** 0.40** 0.45** 0.47** –
13. ­Changef 0.04 − 0.01 − 0.01 − 0.02 0.14** 0.47** 0.58** 0.30** 0.44** 0.37** 0.38** 0.39**

GIQC Gender Identity Questionnaire for Children, CSBQ Children’s Social Behavior Questionnaire, Gender refers to birth-assigned gender
**p-value < 0.01
*p-value < 0.05
a
Not tuned: behavior not optimally changed to the situation
b
Social: reduced social interest and contact
c
Orientation: orientation problems in activity, time, place or in relation to others
d
Understanding: difficulties in understanding social information
e
Stereotyped: stereotyped behavior and a preoccupation with sensory stimuli
f
Change: fear or resistance to change

gender was significantly associated with the GIQC. As such,

dren. The GIQC was found to have a significant negative
these two variables were controlled for subsequently.
correlation with birth-assigned gender. Controlling for gen-
Table 4 shows the multiple linear regression analysis
der (Step 1), a step-wise multiple linear regression analysis
predicting GIQC scores. Predictors included the CBCL
was conducted (Table 6). In Step 2, ASD, SPD, and ODD
total T-score and gender (Step 1), and the CSBQ subscales
were unique significant predictors of elevated GIQC scores.
(Step 2). In Step 2, the not tuned subscale was significantly
Step 3 included the interaction of gender with ASD, SPD,
associated with GIQC scores in the negative direction; the
and ODD, respectively, and found no moderating influ-
orientation and stereotyped subscales were significant posi-
ence of gender on the associations between these diagnoses
tive predictors of GIQC scores. Step 3 of the regression

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Journal of Autism and Developmental Disorders (2019) 49:1570–1585 1579

Table 4  Multiple linear B SE ß t-value p-value

regression analyses comparing
the effect of ASD characteristics Step 1: Control variables
on predicting gender variance
Gender − 0.15 0.02 − 0.19 − 8.0 < 0.001
CBCL T-score 0.00 0.00 0.09 3.9 < 0.001
Step 2: ASD characteristics
Not ­tuneda − 0.01 < 0.001 − 0.09 − 2.9 0.003
Socialb 0.01 0.00 0.04 1.3 ns
Orientationc 0.02 0.00 0.15 4.3 0.001
Understandingd 0.00 0.01 0.00 0.01 ns
Stereotypede 0.01 0.01 0.06 2.1 0.036
Changef − 0.02 0.01 − 0.05 − 1.9 ns
Step 3: ASD characteristics × gender
Orientation × gender 0.01 0.01 0.07 1.6 ns
Stereotyped × gender − 0.00 0.01 0.01 − 0.30 ns

SE standard error, CBCL child behavior checklist, ASD autism spectrum disorder, Gender refers to birth-
assigned gender
a
Not tuned: behavior not optimally changed to the situation
b
Social: reduced social interest and contact
c
Orientation: orientation problems in activity, time, place or in relation to others
d
Understanding: difficulties in understanding social information
e
Stereotyped: stereotyped behavior and a preoccupation with sensory stimuli
f
Change: fear or resistance to change

Table 5  Zero-order correlations among study variables and diagnoses for the full sample
Variables 1 2 3 4 5 6 7 8 9 10 11 12

1. GIQC –
2. Gender − 0.18* –
3. Age − 0.01 0.01 –
4. Annual income − 0.00 0.00 0.03 –
5. School performance − 0.02 − 0.07** − 0.12** 0.02 –
6. ASD 0.07** 0.07** 0.03 − 0.02 − 0.06** –
7. ADHD 0.01 − 0.11** 0.10** 0.02 − 0.22** 0.08** –
8. OCD 0.03 0.04* 0.05** 0.01 − 0.05* 0.04** 0.15** –
9. SPD 0.06** 0.04* − 0.01 − 0.00 − 0.05 0.09** 0.17** 0.18** –
10. Mood/anxiety 0.01 0.01 0.11** − 0.01 − 0.09** 0.02 0.25** 0.19** 0.17** –
11. ODD 0.05* 0.02 0.03 0.01 − 0.08** 0.03 0.32** 0.11** 0.07** 0.11** –
12. Neurodevelopmental 0.02 0.04* 0.05* − 0.01 − 0.06** 0.10** 0.18** 0.20** 0.11** 0.14** 0.09** –
13. Learning disorders − 0.01 − 0.00 0.07** 0.00 − 0.21** 0.02 0.10** − 0.01 0.58 0.08** 0.12 0.05*

GIQC gender identity questionnaire for children, ASD autism spectrum disorder, ADHD attention-deficit/hyperactivity disorder, OCD obsessive–
compulsive disorder, SPD sensory processing disorder, ODD oppositional defiant disorder, Gender refers to birth-assigned gender
*p-value < 0.05
**p-value < 0.01

and GIQC scores. Using the Benjamini–Hochberg proce- diagnostic categories as well as the significant independent
dure with a 5% FDR, the effects for ASD (p < 0.001), SPD effects found in the multiple linear regression.
(p = 0.009), and ODD (p = 0.011) remained significant rela- Further, to discern whether GV was distinct from ASD
tive to their respective corresponding adjusted critical alphas characteristics within the SPD and ODD groups, we evalu-
of 0.007, 0.014, and 0.021. Figure 1 presents the mean GIQC ated the correlations between the CSBQ subscales and GIQC
score for the nonclinical sample and each of the individual within these two groups, respectively. For the SPD group, the

13
1580 Journal of Autism and Developmental Disorders (2019) 49:1570–1585

Table 6  Multiple linear B SE ß t-value p-value

regression comparing the effect
of each diagnostic category on Step 1: Control variables
predicting gender variance
Gender − 0.14 0.02 − 0.18 − 9.2 < 0.001
Step 2: Clinical diagnoses
ASD 0.16 0.04 0.07 3.7 < 0.001
ADHD − 0.03 0.03 − 0.02 − 1.1 ns
OCD 0.08 0.10 0.02 0.80 ns
SPD 0.20 0.08 0.05 2.6 0.009
Mood/anxiety disorders − 0.00 0.04 − 0.00 − 0.01 ns
ODD 0.17 0.07 0.05 2.6 0.010
Neurodevelopmental disorders 0.05 0.09 0.01 0.59 ns
Learning disorders − 0.05 0.09 − 0.01 − 0.59 ns
Step 3: Clinical diagnoses × gender
ASD × gender − 0.18 0.10 − 0.07 − 1.9 ns
SPD × gender − 0.04 0.16 − 0.01 − 0.22 ns
ODD × gender − 0.08 0.13 − 0.02 − 0.62 ns

SE standard error, GIQC gender identity questionnaire for children, ASD autism spectrum disorder, ADHD
attention-deficit/hyperactivity disorder, OCD obsessive–compulsive disorder, SPD sensory processing dis-
order, ODD oppositional defiant disorder, Gender refers to birth-assigned gender

Fig. 1  Mean (standard error) 3

GIQC scores for the nonclinical 2.8
group and each clinical diagno-
2.6
sis category. Asterisks indicate
groups that significantly 2.4 * * *
differed from the reference 2.2
group (i.e. nonclinical group) 2
in a multiple linear regression
1.8
(p-value < 0.05). ODD oppo-
sitional defiant disorder, ASD 1.6
autism spectrum disorder, OCD 1.4
obsessive–compulsive disorder, 1.2
and ADHD attention-deficit/ 1
hyperactivity disorder

change subscale was negatively associated with the GIQC Discussion

(r = − 0.41, p < 0.05), but all other subscales did not show a
significant association (range of r = − 0.25 to 0.08, all ns). For In a large community sample of children ages 6–12 years,
the ODD group, the GIQC was positively associated with the this study examined two important issues relevant to
understanding subscale of the CSBQ (r = 0.36, p < 0.05), but childhood GV and ASD characteristics. First, it exam-
all other subscales were not significantly associated with the ined whether ASD characteristics were associated with
GIQC (range of r = − 0.07 to 0.21, all ns).

13
Journal of Autism and Developmental Disorders (2019) 49:1570–1585 1581

GV beyond clinical populations, and whether particular subscale may be less inclined to attend to social cues, includ-
domains of ASD characteristics contributed to this asso- ing those about gender, which might have an influence on
ciation. Second, it examined whether clinical diagnoses development in domains like gender expression and gender-
other than ASD might also be associated with GV. stereotyped beliefs (Martin and Ruble 2004; Strang et al.
We found a positive association between characteristics of 2014). Alternatively, stigmatization of childhood GV may
ASD and GV in this nonclinical sample, indicating that such lead one to feel unaccepted, thus lowering their inclination
associations exist beyond the clinical domain (i.e. beyond to socialize. That said, the social subscale, which measures
those who have clinical diagnoses of ASD and/or GD). In the child’s tendencies to seek out and engage in social inter-
particular, the orientation and stereotyped subscales of the actions, was not significantly associated with GIQC scores,
CSBQ were found to be unique predictors of increased GV; which raises some doubt about whether a relative lack of
however, when a 5% FDR criterion was applied, the stereo- social engagement underpins the orientation subscale’s
typed subscale effect was shown to potentially be a case of association with GV. Further, certain facets of ASD might
Type I error. With regards to children with a developmental/ have an alternate association with gender expression because
mental health clinical diagnosis, we found that ASD (GIQC: the not tuned subscale (i.e. difficulties adapting one’s emo-
M = 2.18, SD = 0.44), SPD (GIQC: M = 2.24, SD = 0.42), tional and behavioral reactions to a social context) predicted
and ODD (GIQC: M = 2.19, SD = 0.46) all showed signifi- decreases, rather than increases, in GV.
cantly higher levels of parent-reported GV compared to the Among the parent-reported developmental/mental health
reference group of children without reported clinical diag- diagnoses significantly associated with increased GV, ASD
noses (GIQC: M = 2.01, SD = 0.39). For the GIQC scale, showed the largest effect size. This finding replicated the
as used here, a cut-off score of 2.46 shows good sensitivity results of prior studies showing elevated wishes to be of
(86.8%) and specificity (95%) for identifying children clinic- the opposite gender among children with ASD (Janssen
referred for gender-related concerns (Johnson et al. 2004). et al. 2016; May et al. 2017; Strang et al. 2014), but with
The children in our sample with ASD, SPD, and ODD were a more refined measure of GV. However, Turban and van
0.63, 0.52, and 0.59 SD’s from this cut-off, respectively, as Schalkwyk (2018) noted that associations between social
compared to 1.10 SD’s for the nonclinical sample of chil- impairments and GD/GV may be nonspecific to ASD. In line
dren. As such, the GV of the average child diagnosed with with this argument, increased levels of GV were also found
ASD, SPD, or ODD appears to be intermediate between that among children with a diagnosis of SPD or ODD.
of typically developing children and those clinic-referred SPD is not currently in the DSM but has been concep-
for GD. tualized as a complex condition in the child development
literature (Miller and Schaaf 2008). Like the present finding
Possible Interpretations of the Present Findings regarding the stereotyped subscale, the association between
SPD and elevated GV is in line with the hypothesis that
In the nonclinical sample, elevations in GV were associated some individuals experience altered sensitivity towards cer-
with the CSBQ orientation subscale and, more tentatively, tain sensory input (e.g. tactile sensations) that intersects with
the stereotyped subscale. These scales measure difficulty gender expression. Yet, because there is considerable over-
orienting one’s attention to social cues as well as atypical lap in characteristics between SPD and ASD (i.e. specific
responses to sensory input (i.e. over- or under-responsivity), interests, idiosyncratic sensory experiences; Schoen et al.
respectively. It should be noted, however, that these findings 2009), it is not clear whether our finding regarding SPD and
are somewhat not in line with a recent study of children GV is simply an extension of the GV-ASD link.
clinically referred for GD reporting elevations in all six The association between GV and ODD corresponds to
domains of ASD characteristics using the CSBQ (van der previous reports of youth referred for GD who either had an
Miesen et al. 2018a). As such, GV in gender-referred chil- ODD diagnosis prior to their referral (Kaltiala-Heino et al.
dren might be broadly associated with ASD characteristics, 2015) or met the criteria for ODD based on age-appropriate
whereas elevated GV in non-referred children may not be diagnostic interviews (Drummond et al. 2017; Wallien et al.
related to all facets of ASD. 2007). The understanding subscale of the CSBQ, however,
Still, the present findings were consistent with some was positively associated with GV in the ODD group, sug-
hypotheses proposed in the prior literature. The association gesting that elevations in GV might have stemmed from
between GV and the stereotyped subscale of the CSBQ is associations with ASD characteristics within the ODD
consistent with the idea that strong gender-related interests group.
in children might be related to preferences for specific kinds Of the several other clinical diagnoses examined
of sensory stimulation (e.g. silky materials, bright and shiny here, no associations were observed. Finding no asso-
objects; de Vries et al. 2010; Tateno et al. 2008; Williams ciation between GV and ADHD is of particular inter-
et al. 1996). Children who score higher on the orientation est given prior quantitative work showing an increased

13
1582 Journal of Autism and Developmental Disorders (2019) 49:1570–1585

co-occurrence of ADHD and GV in clinic-referred chil- Limitations and Future Directions

dren and adolescents (de Vries et al. 2011; Strang et al.
2014; Wallien et al. 2007). This lack of replication could This study was correlational and cross-sectional in its
be due, in whole or in part, to methodological differences, approach. Thus, the ability to control for confounding vari-
including the way in which the presence of an ADHD ables and draw conclusions regarding causation or time-
diagnosis was determined. Also, Strang et al. used the related developmental pathways is limited. Other limitations
single item measure of GV from the CBCL, focusing relate to the method of data collection. Online recruitment
more narrowly on wishes to be of the opposite gender. may have produced coverage and self-selection biases. The
In contrast, the present study used the GIQC as a more use of parent-report measures may have introduced other
comprehensive measure for distinguishing levels of gen- kinds of biases (e.g. socially desirable responding). Ret-
der expression (Johnson et al. 2004). Furthermore, incon- rospective reports regarding developmental/mental health
sistencies between these two studies may be due to dif- diagnoses are less favorable than using a diagnostic inter-
ferences in sample characteristics. Strang et al. examined view. As such, associations between GV and ASD charac-
a clinical sample of both children and adolescents from teristics as well as between GV and various developmental/
a hospital database, whereas the present study was con- mental health diagnoses should be interpreted cautiously.
ducted online and consisted only of children. Moreover, it will be important for future research to rep-
It is important to note that the observed associations licate the current findings before any firm conclusions can
cannot be taken as reflecting causal relations. Other com- be drawn.
mon underlying mechanisms may contribute to the pur- There are also limits to the generalizability of the find-
ported GD/GV-ASD link. For instance, biological fac- ings. The sample sizes for the clinical diagnosis groups
tors might contribute to the association between GD/GV were variable (e.g. ranging from n = 17 for the OCD group
and ASD. The extreme male brain (EMB; Baron-Cohen to n = 248 for the ADHD group) and the heterogeneous
2002; Baron-Cohen et al. 2014) theory, for example, pos- neurodevelopmental group consisted of a wide-range of
its that the cognitive characteristics of ASD reflect an neurodevelopmental diagnoses that may be better assessed
‘extreme-male’ profile based on normative gender differ- as different clinical groups. Larger and more even sample
ences in the relative discrepancy in empathizing/system- sizes would have provided more representative groups and
izing abilities (i.e. on average, greater systemizing over more optimal comparisons. Also, although the GIQC is a
empathizing abilities in boys and vice versa in girls). At well-validated and highly sensitive measure of GV, it is not
the level of potential underlying mechanisms, the fetal equivalent to a GD diagnosis (Johnson et al. 2004) and the
androgen theory hypothesizes that such differences in present findings were obtained from a nonclinical sample.
cognitive profiles stem from exposure to differing levels Thus, while the present findings might have some implica-
of androgens prenatally (e.g. higher levels of testosterone tions for the relationship between GV and ASD, their rel-
experienced by boys in utero), which may partly contrib- evance to GD in clinical populations is unknown; however,
ute to the higher prevalence of ASD in males than in they might guide future research to better understand this
females (Auyeung et al. 2013; Baron-Cohen et al. 2011). relationship in clinical contexts. These findings also provide
As such, the fetal androgen theory may explain the co- some tentative evidence to suggest clinicians working with
occurrence of ASD and GV characteristics, particularly children with SPD or ODD should be mindful of possible
in birth-assigned females. Some research shows birth- GV, similar to the recommendations for working clinically
assigned females who experience GD have elevated ASD with adolescents diagnosed with ASD (Strang et al. 2016).
characteristics compared to birth-assigned males with GD This study was limited to children ages 6–12 years and
(Jones et al. 2012) and are similar to typically developing parents did not report on their child’s pubertal status; how-
boys with regards to scores on empathizing abilities (Di ever, the relationship between GV and ASD exists in clini-
Ceglie et al. 2014). Additional research examined birth cal populations of adolescents and adults as well (de Vries
weight as a biological factor relevant to the GD/GV-ASD et al. 2010; Dewinter et al. 2017; George and Stokes 2018a,
link (VanderLaan et al. 2015). Relatively higher birth b; Janssen et al. 2016; Jones et al. 2012; May et al. 2017;
weight has been correlated with masculinized somatic Pasterski at al. 2014; Skagerberg et al. 2015; Strang et al.
features in females (Avidime et al. 2011) and lower testos- 2014; van der Miesen et al. 2018b). Feelings of GD/GV or
terone in the pre/perinatal period in males (Carlsen et al. underlying motives for expressing to be of the other gender
2006). In gender-referred children, higher birth weight are thought to be experienced differently before vs. after the
and greater GV were associated with clinical-range ASD onset of puberty (Burke et al. 2014; Steensma et al. 2011;
characteristics (VanderLaan et al. 2015). Strang et al. 2016) and a number of studies suggest that
GD in prepubertal children is less likely to persist beyond
puberty whereas adolescents with GD are more likely to

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Journal of Autism and Developmental Disorders (2019) 49:1570–1585 1583

pursue treatment options to alter primary and/or secondary Adelson, S. L. (2012). Practice parameter on gay, lesbian, or bisexual
sex characteristics (for discussion, see de Vries et al. 2010; sexual orientation, gender variance, and gender discordance in
children and adolescents. Journal of the American Academy of
Wallien and Cohen-Kettenis 2008). Future research should Child & Adolescent Psychiatry, 51(9), 957–974.
evaluate to what extent, if any, the present findings extend American Psychiatric Association. (2013). Diagnostic and statistical
to other age groups. manual of mental disorders (5th ed.). Washington, DC: Author.
Arcelus, J., Bouman, W. P., Van Den Noortgate, W., Claes, L., Wit-
comb, G., & Gernandez-Aranda, F. (2015). Systematic review
and meta-analysis of prevalence studies in transsexualism.
Conclusion European Psychiatry, 30(6), 807–815.
Auyeung, B., Lombardo, M. V., & Baron-Cohen, S. (2013). Prenatal
and postnatal hormone effects on the human brain and cogni-
The present study found that characteristics of ASD were tion. Pflügers Archiv-European Journal of Physiology, 465(5),
associated with elevations in GV in a nonclinical commu- 557–571.
nity child sample. This association was mainly attributable Avidime, O. M., Avidime, S., Olorunshola, K. V., & Dikko, A. A.
to certain facets of ASD, namely deficits in social orient- U. (2011). Anogenital distance and umbilical cord testosterone
level in newborns in Zaria, Northern Nigeria. Nigerian Journal
ing and possibly also stereotyped behaviors. Further, this of Physiological Sciences, 26(1), 23–28.
study documented higher levels of GV among children with Baams, L., Beek, T., Hille, H., Zevenbergen, F. C., & Bos, H. M.
parent-reported diagnoses of ASD, SPD, or ODD. These (2013). Gender nonconformity, perceived stigmatization, and
findings suggest that the GD/GV-ASD link might reflect a psychological well-being in Dutch sexual minority youth and
young adults: A mediation analysis. Archives of Sexual Behav-
more general pattern that exists beyond clinical populations. ior, 42(5), 765–773.
Also, elevated GV may not be unique to ASD populations, Baron-Cohen, S. (2002). The extreme male brain theory of autism.
but might be found among other clinical populations as well. Trends in Cognitive Sciences, 6(6), 248–254.
Baron-Cohen, S., Cassidy, S., Auyeung, B., Allison, C., Achoukhi,
Acknowledgments We thank Dr. Carla Mazefsky and two anonymous M., Robertson, S., et al. (2014). Attenuation of typical sex dif-
reviewers for thoughful comments on earlier drafts of this article. ferences in 800 adults with autism vs. 3,900 controls. PLoS
ANN and AS were supported by the Social Sciences and Humani- ONE, 9(7), e102251.
ties Research Council (SSHRC) of Canada Undergraduate Research Baron-Cohen, S., Lombardo, M. V., Auyeung, B., Ashwin, E.,
Awards. This research was funded by a University of Toronto Mis- Chakrabarti, B., & Knickmeyer, R. (2011). Why are autism
sissauga Research and Scholarly Activity Fund Award and a SSHRC spectrum conditions more prevalent in males?. PLoS Biology,
Insight Development Grant awarded to DPV. 9(6), e1001081.
Baron-Cohen, S., Wheelwright, S., Skinner, R., Martin, J., & Clubley,
E. (2001). The Autism-Spectrum Quotient (AQ): Evidence from
Author Contributions ANN, AS, and DPV conceived and designed the Asperger syndrome/high-functioning autism, males and females,
study. ANN, AS, DEP, and DPV collected the data. ANN, AIRvdM, scientists and mathematicians. Journal of Autism and Develop-
DEP, MCL and DPV conducted the data analyses. All authors partici- mental Disorders, 31(1), 5–17.
pated in writing and revising the manuscript. Benjamini, Y., & Hochberg, Y. (1995). Controlling the false discovery
rate: A practical and powerful approach to multiple testing. Jour-
Compliance with Ethical Standards nal of the Royal Statistical Society. Series B (Methodological),
57(1), 289–300.
Conflict of interest The authors declare that they have no conflict of Berndt, T. J., & Heller, K. A. (1986). Gender stereotypes and social
interest. inferences: A developmental study. Journal of Personality and
Social Psychology, 50(5), 889–898.
Ethical Approval All procedures performed in studies involving human Bockting, W. O., Miner, M. H., Swinburne Romine, R. E., Hamilton,
participants were in accordance with the ethical standards of the insti- A., & Coleman, E. (2013). Stigma, mental health, and resilience
tutional and/or national research committee and with the 1964 Helsinki in an online sample of the US transgender population. American
declaration and its later amendments or comparable ethical standards. Journal of Public Health, 103(5), 943–951.
This article does not contain any studies with animals performed by Burke, S. M., Cohen-Kettenis, P. T., Veltman, D. J., Klink, D. T., &
any of the authors. Bakker, J. (2014). Hypothalamic response to the chemo-signal
androstadienone in gender dysphoric children and adolescents.
Informed Consent Informed consent was obtained from all individual Frontiers in Endocrinology. https ​ : //doi.org/10.3389/fendo​
participants included in the study. .2014.00060​.
Carlsen, S. M., Jacobsen, G., & Romundstad, P. (2006). Maternal
testosterone levels during pregnancy are associated with off-
spring size at birth. European Journal of Endocrinology, 155(2),
365–370.
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