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Intense/obsessional interests in children with gender dysphoria: A cross-validation study using the Teacher's Report Form

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Abstract

Objective This study assessed whether children clinically referred for gender dysphoria (GD) show symptoms that overlap with Autism Spectrum Disorder (ASD). Circumscribed preoccupations/intense interests and repetitive behaviors were considered as overlapping symptoms expressed in both GD and ASD. Methods To assess these constructs, we examined Items 9 and 66 on the Teacher’s Report Form (TRF), which measure obsessions and compulsions, respectively. ResultsFor Item 9, gender-referred children (n = 386) were significantly elevated compared to the referred (n = 965) and non-referred children (n = 965) from the TRF standardization sample. For Item 66, gender-referred children were elevated in comparison to the non-referred children, but not the referred children. Conclusions These findings provided cross-validation of a previous study in which the same patterns were found using the Child Behavior Checklist (Vanderlaan et al. in J Sex Res 52:213–19, 2015). We discuss possible developmental pathways between GD and ASD, including a consideration of the principle of equifinality.
Zucker et al.
Child Adolesc Psychiatry Ment Health (2017) 11:51
DOI 10.1186/s13034-017-0189-9
RESEARCH ARTICLE
Intense/obsessional interests inchildren
withgender dysphoria: a cross-validation study
using the Teacher’s Report Form
Kenneth J. Zucker1*, A. Natisha Nabbijohn2, Alanna Santarossa2, Hayley Wood3, Susan J. Bradley1,
Joanna Matthews2 and Doug P. VanderLaan2,4
Abstract
Objective: This study assessed whether children clinically referred for gender dysphoria (GD) show symptoms that
overlap with Autism Spectrum Disorder (ASD). Circumscribed preoccupations/intense interests and repetitive behav-
iors were considered as overlapping symptoms expressed in both GD and ASD.
Methods: To assess these constructs, we examined Items 9 and 66 on the Teacher’s Report Form (TRF), which meas-
ure obsessions and compulsions, respectively.
Results: For Item 9, gender-referred children (n = 386) were significantly elevated compared to the referred
(n = 965) and non-referred children (n = 965) from the TRF standardization sample. For Item 66, gender-referred chil-
dren were elevated in comparison to the non-referred children, but not the referred children.
Conclusions: These findings provided cross-validation of a previous study in which the same patterns were found
using the Child Behavior Checklist (Vanderlaan et al. in J Sex Res 52:213–19, 2015). We discuss possible developmental
pathways between GD and ASD, including a consideration of the principle of equifinality.
Keywords: Gender dysphoria, Autism Spectrum Disorder, Teacher’s Report Form, Equifinality, DSM-5
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Background
Children with a DSM-5 diagnosis of gender dysphoria
(GD) [Gender Identity Disorder of Childhood in DSM-III
and III-R and Gender Identity Disorder (GID) in DSM-
IV] have a marked incongruence between the gender
they have been assigned to at birth and their experi-
enced/expressed gender [1].1 e DSM-5 indicators for
the diagnosis, as in DSM-III and DSM-IV, include an
array of sex-typed behaviors (e.g., toy and activity inter-
ests, dress-up play, roles in fantasy play, etc.) that often
signal a strong identification with the other gender. Over
three decades ago, Coates [2] reported the clinical
impression that at least some boys with GD appeared to
show an intense, if not obsessional, interest in
1 We will use primarily GD to reflect the current DSM-5 diagnostic label,
but use GID when it is historically accurate to do so (e.g., regarding the clin-
ical diagnosis of the participants in this study).
gender-related themes, as manifested in their surface
behaviors and in fantasy play, and in their responses dur-
ing projective testing such as the Rorschach [3] (for a
recent clinical example, see Saketopoulou [4]. It is
unclear, however, whether these patterns of behavior are
simply an “inverted” instance of the intense gender-
related interests and behaviors seen in typically-develop-
ing children [5, 6] or represent something that is
qualitatively distinct or, at least, at the extreme end of a
quantitative spectrum.
One relatively recent line of research, stimulated by a
series of clinical case reports and one internet-recruited
sample (of children, adolescents, and adults), has pointed
to a possible link between GD and Autism Spectrum
Disorder (ASD) or at least traits of ASD [719]. Using a
structured diagnostic interview schedule, dimensional
Open Access
Child and Adolescent Psychiatry
and Mental Health
*Correspondence: ken.zucker@utoronto.ca
1 Department of Psychiatry, University of Toronto, Toronto, ON M5T 1R8,
Canada
Full list of author information is available at the end of the article
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Page 2 of 8
Zucker et al. Child Adolesc Psychiatry Ment Health (2017) 11:51
measures, or chart review, several studies have reported,
compared to normative samples, an overrepresentation
of either ASD or ASD traits among clinic-referred chil-
dren and/or adolescents [2023] or adults [24, 25] with
a diagnosis of GID/GD (for an internet-recruited sample,
see also Kristensen and Broome [26] (for reviews, see
Glidden etal. [27], Strang etal. [28], van der Miesen etal.
[29], and van Schalkwyk etal. [30] ).
One potential explanation for the putative link between
GD and ASD is the intense focus on, or an obsessional
interest in, specific activities [31, 32]. Such interests
relate to the DSM-5 ASD criterion pertaining to highly
restricted and fixated interests. For example, it is con-
ceivable that children with ASD who form intense and
focused attention to cross-sex objects or activities may
then begin to express other characteristics of GD (e.g.,
see Strang etal. [33]). Conversely, GD may give rise to
such interests and obsessions, leading to a clinical pres-
entation consistent with ASD. In order to appraise these
two proposed pathways, however, the first step would
be to determine empirically if, in fact, children with GD
manifest an elevated pattern of intense interests and
obsessions.
To our knowledge, only two studies have focused on
a possible elevation in obsessional/repetitive interests
and behaviors in GD children using dimensional met-
rics. Skagerberg etal. [23] used the Social Responsive-
ness Scale (SRS) in a mixed sample of 166 children and
adolescents and found an elevation on the “Autistic
Mannerisms” subscale completed by the parents [now
labeled “Restricted Interests and Repetitive Behav-
iors” (RIRB) on the SRS-2] [34] compared to a norma-
tive sample. However, two methodological issues call
for some caution in appraising the results. First, the
participation rate was only 46%, which may represent
a threat to the internal validity of the sample [35]. Sec-
ond, a clinic-referred comparison group, consisting of
children/adolescents referred for other clinical prob-
lems, was not included. us, it is not clear if the eleva-
tion on the Autistic Mannerisms subscale is specific to
children/adolescents referred for gender dysphoria or
characteristic of clinic-referred children/adolescents in
general.
Taking advantage of a large “archival” data set, Vander-
Laan etal. [36] analyzed two items on the Child Behavior
Checklist (CBCL) [37] pertaining to obsessionality and
repetitive behavior: Item 9 (“Can’t get his/her mind off
certain thoughts; obsessions”) and Item 66 (“Repeats cer-
tain acts over and over; compulsions”) in a sample of 534
children referred clinically for gender identity concerns,
419 siblings, and 1201 referred and 1201 non-referred
children from the CBCL standardization sample [37],
with an age range of 3–12years.2 For both items, parental
responses were dichotomized as either present (“Some-
what or sometimes true”/“Very true or often true”) or
absent (“Not true”). In their study, the parental participa-
tion rate was over 90% for the gender-referred sample.
For Item 9, the percentage of mothers of the gender-
referred children who endorsed it (62.4%) was sig-
nificantly greater than that of their siblings (22.2%) and
significantly greater than the ratings of the mothers of
both the referred (48.7%) and non-referred (21.9%) chil-
dren from the CBCL standardization sample (odds ratios,
with a 95% CI ranged from 1.66 to 10.96). e percentage
of mothers of the referred children who endorsed it was
also significantly greater than the ratings for the siblings
and of the non-referred children. For Item 66, the per-
centage of mothers of the gender-referred children who
endorsed it (25.3%) was significantly greater than that of
their siblings (8.2%) and the ratings of the non-referred
children (5.4%) (odds ratios ranged from 3.04 to 6.77), but
not of the referred children (24.9%), who also had higher
endorsement ratings than the siblings of the gender-
referred children and of the non-referred children. us,
in this study, there was evidence for both specificity and
non-specificity for these two behaviors: On the one hand,
both the gender-referred children and the referred chil-
dren were elevated on both items compared to the sib-
lings and non-referred children (non-specificity); on the
other hand, a greater percentage of the gender-referred
children than the referred children were elevated on Item
9, evidence for at least partial specificity.
For the gender-referred children and their siblings, it
was also possible to code qualitatively the reasons that the
mothers endorsed these two items. A two-option coding
scheme classified the reasons as either gender-related
(e.g., “Cinderella” for Item 9) or non-gender-related (e.g.,
“killing”). For Item 9, VanderLaan etal. [36] found that
gender-related themes were significantly more com-
mon for the gender-referred boys than that of the male
siblings, but the difference between the gender-referred
girls and that of the female siblings was not significant
(possibly due to low power because of the smaller sample
size). For Item 66, there was no significant difference in
2 In developmental clinical psychology and psychiatry, the CBCL [37] is
one of the most widely used parent-report measures of behavioral and emo-
tional problems in children and adolescents. It contains a total of 118 items,
each of which is rated on a 0–2 point scale for frequency of occurrence. Fac-
tor analysis has identified both broad-band (Internalizing, Externalizing)
and eight narrow-band dimensions of behavioral and emotional disturbance
(e.g., “Anxious/Depressed,” “Aggressive Behavior.” Items 9 and 66 load on
the “ought Problems” narrow-band scale, which is part of a suite of three
narrow-band dimensions that do not load on either the Internalizing or
Externalizing broad-band dimensions. On average, completion of the CBCL
takes about 15–17min [37, p. 14].
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Zucker et al. Child Adolesc Psychiatry Ment Health (2017) 11:51
gender-related themes for the gender-referred children
and their siblings.
e purpose of the present study was to cross-validate
the VanderLaan etal. [36] findings for these two items
using teacher ratings on the Teacher’s Report Form [38]
to see if teachers would also report elevations in gender-
referred children when compared to both referred and
non-referred children in the TRF standardization sample
[39].3
Methods
Participants
Between 1986 and 2013, TRFs were obtained for 386
children (304 boys; 82 girls) who were referred to, and
then assessed in, a specialty gender identity service for
children, housed within a child psychiatry program at an
academic health science center. e children had a mean
age of 7.77 years (SD = 2.41). All of the children met
DSM-III, DSM-IV or DSM-5 criteria for GID/GD or were
subthreshold for the diagnosis (e.g., Gender Identity Dis-
order NOS). During this time period, TRFs were not
available for an additional 145 gender-referred children.
e main reasons for this were: the parents did not want
the teacher to complete the TRF (because of concerns
about privacy/confidentiality); a TRF was mailed to the
teacher/school, but it was not returned; the child was too
young for the TRF to be administered (e.g., not yet in
school); the child was being home-schooled; or, the fam-
ily chose not to complete the assessment so the TRF was
not sent to the teacher.4
For comparative purposes, we used the TRF referred
(498 boys; 467 girls) and non-referred (498 boys;
467 girls) standardization samples for children ages
6–12 years from Achenbach and Rescorla [39]. As
reported by Achenbach and Rescorla, the referred sam-
ple was obtained from various mental health and special
educational settings, primarily in the U.S., heterogene-
ous with regard to DSM diagnoses. e non-referred
sample was obtained from the 1999 National Survey of
Children, Youths, and Adults conducted between Febru-
ary 1999 and January 2000. Parents who completed the
CBCL were asked for permission to mail a TRF to one of
their child’s teachers, who received $10 in compensation
3 e TRF [38] is similar in design and format to that of the CBCL. ere
are 25 items on the TRF that are more appropriate for the school setting
(e.g., “Dislikes school”) and these items replace 25 items on the CBCL.
Factor analysis has identified the same broad-band and narrow-band
dimensions of behavioral and emotional disturbance as on the CBCL. e
behavioral and emotional problem items on the TRF can be completed, on
average, in about 10min [38, p. 11].
4 Our clinic began administering the TRF in 1986, when it was first pub-
lished [40]. For preschoolers, the Caregiver-Teacher Report Form for Ages
1–1/2–5 was administered once it became available [41]; unfortunately, this
version of the TRF does not contain the two items analyzed in this study.
for participation. Children were included in the non-
referred sample if they had not received professional help
for behavioral, emotional, substance use, or developmen-
tal problems in the preceding 12months [39, pp. 75–76].
e referred and non-referred samples were matched for
gender, age, socioeconomic status, and ethnicity [39, pp.
75–76, p. 109].
Measures
For both Items 9 and 66, teacher responses were dichoto-
mized where 0= 0 and 1 or 2=1. Using the parental
data from our previous study for the gender-referred
sample [36], we calculated mother–teacher and father–
teacher correlations for both items using the continuous
0 to 2 coding system. For the gender-referred children,
we recorded the comments provided by the teacher if the
items were scored either as a 1 (“somewhat or sometimes
true”) or 2 (“very true or often true”) and then used our
previously-developed two-category qualitative coding
scheme by classifying the teacher descriptions as either
gender-related or non-gender-related. Examples of gen-
der-related themes for Item 9 were “Obsessed with female
actions, colors, activities,” “preoccupied with dressing
up at house center,” and “Spiderman.” Examples of non-
gender-related themes were “frequently day dreams,” “…
food,” and “revengeful thoughts.” Corresponding gender-
related theme examples for Item 66 were “Dresses up like
a female” and “Drawing females” and non-gender-related
themes were “paces” and “repeated cracking knees and
elbows.” Two authors (ANN, JM) independently coded
both items as either gender-related or non-gender-
related. For Item 9 (n=129), the kappa was .87 (p<.001);
for Item 66 (n=47), the kappa was .95 (p<.001). Unfor-
tunately, it was not possible to code for qualitative com-
ments in the referred and non-referred standardization
samples because they were not available in the raw data
file provided to us by Achenbach.
e present study constituted a reanalysis of data from
previous research projects for which there was eth-
ics approval from the [Centre for Addiction and Mental
Health] Research Ethics Board. is research was con-
ducted in accordance with the Declaration of Helsinki.
Results
Preliminary analyses
We first compared the gender-referred children for
whom a TRF was completed vs. those for whom it was
not (including the cases in which the TRF version for
preschoolers was used). As expected, children for whom
the TRF was completed were, on average, significantly
older than those children for whom it was not,
t(529)= 7.02, p<.001. ere was no significant differ-
ence for year of assessment. Children for whom a TRF
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Zucker et al. Child Adolesc Psychiatry Ment Health (2017) 11:51
was completed had a significantly lower Full-Scale IQ (M,
101.1 vs. 108.4), came from a somewhat lower social class
background (M, 42.1 vs. 46.8; absolute range 8–66) [42],
and had higher Internalizing (M, 62.1 vs. 56.8) and Exter-
nalizing (M, 61.5 vs. 54.4) T scores on the CBCL (all
p<.001). With age co-varied, these differences remained
statistically significant, with the exception of social class.5
Teacher ratings forItems 9 and66
Table 1 shows the dichotomized teacher ratings for
Items 9 and 66 (in percent) for the gender-referred chil-
dren, the referred children, and the non-referred chil-
dren, stratified by sex. For both the boys and the girls,
the overall chi square test was statistically significant
for both Items 9 and 66: Item 9 for boys, χ2(2)=90.61,
p<.00001; for girls, χ2(2)=42.86, p< .00001; Item 66
for boys, χ2(2)=42.21, p<.00001; for girls, χ2(2)=16.28,
p=.00029. To decompose the overall effect, three paired
contrasts were conducted for both items: gender-referred
vs. referred children from the standardization sample,
gender-referred vs. non-referred children from the stand-
ardization sample, and referred vs. non-referred children
from the standardization sample, by sex (Table1).
For Item 9, for the boys, it can be seen that teachers
were significantly more likely to endorse this item with
a rating of either a 1 or a 2 for both the gender-referred
and referred samples when compared to the non-referred
sample. It can also be seen that teachers were signifi-
cantly more likely to endorse this item for the gender-
referred boys than for the referred boys. For the girls, the
findings were similar.
For Item 66, for the boys, it can be seen that teachers
were significantly more likely to endorse this item with
a rating of either a 1 or a 2 for both the gender-referred
and referred samples when compared to the non-referred
sample, but the comparison between the gender-referred
boys and the referred boys in the standardization sam-
ple was not significant. For the girls, the findings were
similar.
Correlational analyses
In the gender-referred sample (collapsed across sex), we
calculated the correlation between the continuous ratings
for Items 9 and 66 for the TRF and the CBCL [36]. For
Item 9, the mother-teacher correlation was .28 (n= 337,
p < .001) and the father-teacher correlation was .23
(n=248, p<.001). For Item 66, the mother-teacher corre-
lation was .17 (n=345, p=.002) and the father–teacher
correlation was .11 (n=255, p=.091). We also calculated
the correlation between the continuous ratings for Items 9
and 66 and age (collapsed across sex), which were.11
5 ese analyses are available from the corresponding author upon request.
(p=.029) and .00 (ns), respectively. For the referred sam-
ple, the correlations were .05 (ns) and .07 (p=.033),
respectively. For the non-referred sample, the correlations
were .01 and .02, respectively (both ns).6 us, age effects
were either non-existent or extremely small.
Qualitative analysis
For the qualitative analyses, teachers provided writ-
ten comments for 84.3% (n=129/153) of the gender-
referred sample for whom Item 9 was rated as a 1 or a
2 and for 74.6% (n=47/63) of the sample for who Item
66 was rated as a 1 or a 2 (see Table1). For Item 9, 47.2%
of the comments for boy were coded as gender-related
compared to 30.4% for girls, a non-significant difference,
χ2(1)=1.52. For Item 66, the corresponding percentages
were 32.4 and 0%, respectively, which was also not sig-
nificant, χ2(1)<1.
Discussion
An emerging clinical and research literature has sug-
gested a co-occurrence between GD and ASD (or ASD
traits). VanderLaan et al. [36] had hypothesized that
this link might be due, at least in part, to an elevated
presence of intense/obsessional interests that involve
gender-related behaviors. In their study, parents of
gender-referred children endorsed CBCL Item 9 more
frequently than they did for siblings and by parents in
both referred and non-referred children from the CBCL
standardization sample. is finding was, therefore, con-
sistent with the proposition that the basis of the GD-
ASD link is the tendency of gender-referred children to
present clinically in a manner that corresponds to the
ASD criterion pertaining to highly restricted and fixated
interests. In this regard, it is important to note that this
item corresponds very closely to two items on the SRS-2
that load on the RIRB subscale (Items 26: “inks or talks
about the same thing over and over” and Item 31: “Can’t
get his or her mind off something once he or she starts
thinking about it”). e results for Item 66 also suggested
that the ASD diagnostic criterion pertaining to repeti-
tive behaviors and routines might also be relevant to
GD in children. For this item, parental ratings were also
elevated compared to siblings and non-referred children,
but not when compared to referred children, so there was
less support for a specificity effect. In relation to the SRS-
2, this item bears some similarity to RIRB subscale Item
4: “When under stress…shows rigid or inflexible patterns
of behavior…” In a comparative perspective, however, it
could be argued that intense/obsessional interests (Item
6 It was not possible to calculate mother–teacher correlations for Items 9 and
66 in the standardization samples because the raw data for the CBCL and
TRF were in separate SPSS files.
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Zucker et al. Child Adolesc Psychiatry Ment Health (2017) 11:51
9) provide a stronger basis than repetitive behaviors/rou-
tines (Item 66) for the link between GD and ASD.
Using the TRF, the present study provided a cross-
validation of the CBCL findings [36]. For Item 9, the
gender-referred children had significantly higher ratings
than both the referred and non-referred children in the
standardization sample but, for Item 66, the ratings were
significantly higher only when compared to the non-
referred children. Although the percentage of gender-
referred children for which Items 9 and 66 were endorsed
by teachers was lower than the percentage for which the
items were endorsed by parents in VanderLaan etal. [36],
the same was true for the referred and non-referred chil-
dren. Also as in VanderLaan etal., gender-related themes
were identified on both Items 9 and 66 for boys and,
on Item 9, for girls as well. For example, on Item 9 for
boys, 47% of the descriptors pertained to gender-related
themes, which was similar to the percentage of 54%
that mothers provided. us, the pattern across the two
informants (parents, teachers) was very similar.
If there is, indeed, an empirical basis for the role of
gender-related obsessionality that contributes to the GD-
ASD link, the possible developmental pathways need to
be formulated. As noted earlier, one idea is that ASD
sometimes leads to intense interests in cross-sex objects
or activities, giving rise to a clinical presentation of GD.
us, on this basis, one would predict that GD children
would also exhibit additional features of ASD. In the
study by Skagerberg etal. [23], this appeared to be the
case: although Skagerberg et al. did not provide formal
statistical tests, our own analysis of their data showed
that, compared to a normative sample, children and ado-
lescents with GD had significantly higher ratings on all of
the other subscales of the SRS, not just the one pertain-
ing to restricted interests and repetitive behaviors.7
7 We conducted t tests on the data provided in Table2 in Skagerberg etal.
[23]. ese analyses are available from the corresponding author upon
request.
Table 1 Teacher ratings ofTRF Items 9 and66 asa function ofgroup andsex
Referred and non-referred raw data from Achenbach and Rescorla [39] provided by Achenbach in an SPSS le
Ratings ofobsessions (Item 9)
Groups 0 1 or 2 χ2(1) pOR (95% CI)
n % n %
Boys
Gender-referred vs. 172 58.3 123 41.7
Referred 332 66.7 166 33.3 5.23 .022 1.43 (1.06–1.92)
Non-referred 433 86.9 65 13.1 82.45 < .001 4.76 (3.36–6.75)
Referred vs. non-referred 56.36 < .001 3.33 (2.41–4.58)
Girls
Gender-referred vs. 49 62.0 30 38.0
Referred 356 76.2 111 23.8 6.39 .011 1.96 (1.18–3.24)
Non-referred 414 88.7 53 11.3 35.12 < .001 4.78 (2.78–8.18)
Referred vs. non-referred 24.03 < .001 2.43 (1.70–3.47)
Ratings ofcompulsions (Item 66)
Groups 0 1 or 2 χ2(1) pOR (95% CI)
n % n %
Boys
Gender-referred vs. 247 81.2 55 18.2
Referred 415 83.3 83 16.7 < 1 ns 1.11 (.76–1.62)
Non-referred 473 95.0 25 5.0 34.90 < .001 4.21 (2.56–6.92)
Referred vs. non-referred 33.74 < .001 3.78 (2.37–6.03)
Girls
Gender-referred vs. 72 90.0 8 10.0
Referred 421 90.1 46 9.9 < 1 ns 1.01 (.46–2.24)
Non-referred 451 96.6 16 3.4 5.56 .018 4.90 (1.99–12.07)
Referred vs. non-referred 14.52 < .001 3.07 (1.71–5.52)
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Zucker et al. Child Adolesc Psychiatry Ment Health (2017) 11:51
e Skagerberg etal. [23] data would appear to chal-
lenge another developmental pathway proposed by
VanderLaan etal. [36]. If restricted and intense cross-
sex interests are simply a manifestation of GD, the ASD
“flavor” might be only subclinical or even superficial,
because the intensity of the interests is only a marker of
the GD and not an underlying ASD. If such were the case,
then few, if any, additional ASD features should accom-
pany intense cross-sex interests. But this was clearly not
the case in the Skagerberg etal. data set.
From Skagerberg etal. [23] and other systematic stud-
ies of GD samples (noted earlier), it is clear that there
are many children with GD who would not be diagnosed
with an ASD or would even be in the clinical range on
dimensional measures of ASD traits, as, for example,
on the SRS. Recognition of this variability is consistent
with the principle of equifinality [43]. ASD or ASD traits,
including the presence of intense and restricted interests,
may lead to gender dysphoria, but for those GD children
without ASD or ASD traits the presence of intense and
restricted interests may be caused by other underly-
ing processes. is would, of course, be consistent with
multifactorial models of gender dysphoria, in which the
relative contribution of risk factors will vary in their rela-
tive weight from one child to the next [44]. Along similar
lines, it should also be noted that there are now several
studies which document an elevation in ASD traits, as
measured by the SRS, in children referred for a variety of
clinical problems [4549], not just in children referred for
GD, which clearly points to a pattern of non-specificity.
is non-specificity effect is a clear indication that the
hypothesized GD-ASD link requires a more nuanced
examination. One such strategy would be to design formal
tests of equifinality in which GD children are divided into
two subgroups: those with ASD or ASD traits and those
without. One could then examine whether or not the two
subgroups differ in other important ways. In one study,
VanderLaan etal. [50] reported in a sample of children
with GD that those with higher ASD traits and a higher
score on a dimensional measure of gender-variant behav-
ior had a higher birth weight. VanderLaan etal. [50] noted
that high birth weight has been identified as a risk factor
for ASD and that it is also associated with lower prena-
tal levels of testosterone in males and with masculinized
somatic features, such as a greater anogenital distance,
in females. is finding is consistent with one study that
reported an association between the degree of demascu-
linizing endocrine disruptor chemicals in maternal blood
and ASD traits in children [51]. In another study, Shumer
et al. [52] found that mothers (but not fathers) in the
Nurses’ Health Study II and the Growing Up Today Study
1 who had higher self-reported SRS scores rated their
children as higher in gender-variant behaviors, suggesting
some type of underlying biological liability, perhaps along
the maternal line, for both variables. ese two stud-
ies lend some support for further tests of the equifinality
principle with regard to the GD-ASD link.
Limitations
ere are four limitations to the current study that should
be noted. First, we assessed the focal variables of obses-
sional interests and repetitive behaviors using only single
items from the TRF and our primary analysis was based
on a dichotomous (present vs. absent) metric. Although
both our prior CBCL analysis and the current TRF analy-
sis were quite successful in detecting significant between-
groups effects, we recognize that dimensional measures,
such as the SRS, would be psychometrically superior as
this line of research continues. However, given the cur-
rent intense interest in the GD-ASD link in the literature,
it was our view that the use of a large “archival” data set
(i.e., using a sample of children going back several dec-
ades) would add to this contemporary discourse. Sec-
ond, although we were able to obtain TRFs on 73% of
the entire sample of gender-referred children assessed
between 1986 and 2013, we were not able to use the TRF
data that were available for preschoolers because the rel-
evant items are not on this version. us, future research
should use the SRS so that the restricted interests and
repetitive behaviors construct can be evaluated dur-
ing the developmental period in which GD is often first
expressed [1]. ird, it should be considered whether or
not parents and teachers who endorsed Items 9 and 66
and provide gender-related themes were “over-reacting”
because the child’s gendered behavior was atypical or if
the ratings represent bona fide evidence of obsessional-
ity and compulsivity. On this point, one could test this by
looking at children whose parents describe them as being
preoccupied with gender-typical behaviors, as in the
Halim etal. [6] study of the “pink frilly dresses” phenom-
enon in young girls and to see if they too would be more
likely to endorse these items when compared to girls who
are not seen as overly preoccupied with gender norma-
tive behaviors. Lastly, it should be emphasized that our
data speak more to the potential presence of ASD traits
than to the categorical ASD diagnosis.
We recognize that our data only speak to one aspect
of an ASD but not other core elementss, such as marked
impairment in social communication and social interac-
tion. us, we in no way wish to argue that elevations
in obsessional interests/behaviors per se are sufficient
in making any kind of definitive conclusion about ASD.
However, it is important to note that our data are con-
sistent with one study that analyzed CBCL and TRF
items that discriminated children with an ASD diag-
nosis from clinic-referred children classified as having
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
Page 7 of 8
Zucker et al. Child Adolesc Psychiatry Ment Health (2017) 11:51
an internalizing disorder, an externalizing disorder, no
diagnosis, and children from the general population [53].
So etal. [53] found that 10 CBCL/TRF items were sig-
nificantly higher in the ASD group than the other four
groups: Items 9 and 66 were two of these items, with
between-groups odds ratios ranging from 1.25 to 2.08 for
the 10 CBCL items and 1.17–1.55 for the 10 TRF items.
Given these findings, it is our view that Items 9 and 66,
at least in children, may be more suggestive of ASD traits
than traits suggestive of an Obsessive–Compulsive Dis-
order because natural history data suggest that OCD
onsets at a much later age than ASD [1].
To date, the GD-ASD literature in children has been
largely limited to case reports. Other than our own work
[36, 49], only the Skagerberg etal. [23] study used a dimen-
sional assessment measure to assess putative ASD traits
and only one study, which used a selective sub-sample
of children and adolescents referred for gender dyspho-
ria, employed a structured diagnostic interview sched-
ule to ascertain an autism diagnosis [20]. Going forward,
researchers in this specialty area will need to decide if there
would be benefits in using more formal diagnostic meth-
ods, such as the Autism Diagnostic Observation Sched-
ule [54], to ascertain the percentage of children referred
for gender dysphoria who would meet criteria for the
diagnosis.
Conclusion
Our TRF study provides a cross-validation of our previ-
ous CBCL study of an elevation in intense interests/obses-
sional traits among children referred for gender dysphoria
as compared to both referred and non-referred children
in the standardization sample and, to a lesser extent, with
regard to repetitive behaviors. ese findings, therefore,
give some support to the idea that there may be a link
between gender dysphoria and ASD traits. However, the
emerging literature that suggests a non-specific pattern of
elevations in ASD traits among clinic-referred children in
general calls for a more focused examination of why such
a link may be present among at least some children with a
DSM diagnosis of gender dysphoria.
Abbreviations
ASD: Autism Spectrum Disorder; CBCL: Child Behavior Checklist; DSM:
Diagnostic and Statistical Manual of Mental Disorders; GD: gender dysphoria;
GID: Gender Identity Disorder; SRS: Social Responsiveness Scale; TRF: Teacher’s
Report Form.
Authors’ contributions
KJZ, HW, SJB, and DPV were responsible for the conceptual basis of the study
and its design. KJZ, ANN, AS, and DPV were involved in the data analysis and
interpretation. JM contributed to data coding. The manuscript was prepared
by KJZ with assistance from all coauthors. All authors read and approved the
final manuscript.
Author details
1 Department of Psychiatry, University of Toronto, Toronto, ON M5T 1R8,
Canada. 2 Department of Psychology, University of Toronto Mississauga, Mis-
sissauga, ON, Canada. 3 Psychological Services, Toronto District School Board,
Toronto, ON, Canada. 4 Underserved Populations Research Program, Child,
Youth and Family Division, Centre for Addiction and Mental Health, Toronto,
ON, Canada.
Acknowledgements
Not applicable.
Competing interests
The authors declare that they have no competing interests.
Availability of data
The datasets used and/or analyzed during the current study are available
from Dr. Doug P. VanderLaan (doug.vanderlaan@utoronto.ca) on reasonable
request.
Consent for publication
Not applicable.
Ethical approval
The present study constituted a reanalysis of data from previous research
projects for which there was ethics approval from the Centre for Addiction
and Mental Health Research Ethics Board. This research was conducted in
accordance with the Declaration of Helsinki.
Funding
DPV was supported by a Canadian Institutes of Health Research Postdoctoral
Fellowship, the Centre for Addiction and Mental Health, and the University of
Toronto Mississauga. AS and ANN were supported by University of Toronto
Excellence Awards funded by the Social Sciences and Humanities Research
Council of Canada.
Publisher’s Note
Springer Nature remains neutral with regard to jurisdictional claims in pub-
lished maps and institutional affiliations.
Received: 6 May 2017 Accepted: 16 September 2017
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... There were four mentions of this theory and no claims of evidence for or against it. All four papers (Janssen et al., 2016;Nabbijohn et al., 2019;van der Miesen et al., 2018a, Zucker et al., 2017 cited the same study by Vanderlaan et al. (2015). In this study, Vanderlaan et al. (2015) found that high birth weight was associated with high autistic traits and high gender nonconformity. ...
... Still, several authors made their arguments based on other studies. For example, Zucker et al. (2017) argued for the role of prenatal hormones by referring to Vanderlaan et al. (2015), who suggested that risk factors for ASC were associated with lower prenatal testosterone levels in trans AMABs. In contrast with this, George and Stokes (2018) with ASC traits. ...
... Genetic factors were mentioned as an explanation four times without claimed empirical evidence. Two of the studies, Cheung et al. (2018) and Zucker et al. (2017), referred to other studies on a possible genetic link between ASC and TGM. Based on the study by Vanderlaan et al. (2015), Cheung et al. (2018) proposed shared (epi)genetic factors for gender identity and ASC. ...
Article
Full-text available
While research on the prevalence of co-occurring autism spectrum conditions (ASC) and trans gender modality (TGM) is available, less is known about the underlying mechanism of this association. Insight is needed to improve treatment of trans autistic people. This review provides an overview of theories on the ASC-TGM link and the available evidence for/against them published between January 2016 and October 2020. A systematic search was performed in PubMed, PsycINFO, Web of Science, and Scopus. This resulted in 36 studies, in which 15 theories were identified. Results indicate all theories lack substantial empirical support. Unlikely and promising theories were identified. The most promising theories were those on resistance to social norms and weakened sex differences. Future directions are provided.
... In recent years, there has been a strong and growing interest in the co-occurrence of Gender Dysphoria (GD) and Autism Spectrum Disorder (ASD), both in children/adolescents and adults [1][2][3][4][5][6][7][8][9]. This association is of great clinical interest because of its implications in the management of both conditions. ...
Article
Objectives We evaluated differences in Autism Spectrum Quotient (AQ) scores between a sample of hormone-naïve transgender and cisgender people, the possible impact of gender-affirming hormonal treatment (GAHT) on AQ scores across time and the role of alexithymia and social anxiety as possible mediators of changes in AQ scores. Methods A cross-sectional comparison between cisgender and transgender people before GAHT and a prospective study on the effects of GAHT over time were performed. Transgender and cisgender people completed several psychometric tests: the AQ, the Gender Dysphoria Questionnaire for Adolescents and Adults (GIDYQ-AA), the Body Uneasiness Test (BUT), the Toronto Alexithymia Scale (TAS), the Liebowitz Social Anxiety Scale (LSAS) and the Symptom Checklist 90 revised (SCL-90-R). A total sample of 789 persons (n=229 cismen;n=172 ciswomen;n=206 transmen;n=182 transwomen) referring to the Florence and Rome Gender Clinics was enrolled. Of these, 62 participants referring to the Florence Gender Clinic were evaluated in a prospective study at baseline and 12 months after GAHT. Results Groups showed significant differences in terms of autistic traits: ciswomen showed lower scores of AQ while cismen reported higher scores of AQ than all other groups (p<0.001). Transgender individuals showed significant higher levels of Gender Dysphoria (GD; GIDYQ-AA) and body uneasiness (BUT) than the other groups (p<0.001). Considering alexithymia (TAS) and social anxiety (LSAS), transgender individuals scored significantly higher when compared to cisgender ones (p<0.001 and p<0.05 respectively). No significant differences in general psychopathology (SCL-90-R) were found between groups (p=0.61). Across time, transmen and transwomen showed a significant reduction in AQ scores (p<0.002). The decrease in alexithymia and social anxiety after GAHT did not predict the change in AQ scores. Conclusions The autistic traits measured in our sample may represent an epiphenomenon of GD rather than being part of an Autism Spectrum Disorder (ASD) condition, since they significantly decreased after 12 months of GAHT. Conflicts of Interest The authors declare no conflict of interest.
... In recent years, there has been a strong and growing interest in the co-occurrence of Gender Dysphoria (GD) and Autism Spectrum Disorder (ASD), both in children/adolescents and adults [1][2][3][4][5][6][7][8][9]. This association is of great clinical interest because of its implications in the management of both conditions. ...
Article
Objectives We evaluated differences in Autism Spectrum Quotient (AQ) scores between a sample of hormone-naïve transgender and cisgender people, the possible impact of gender-affirming hormonal treatment (GAHT) on AQ scores across time and the role of alexithymia and social anxiety as possible mediators of changes in AQ scores. Methods A cross-sectional comparison between cisgender and transgender people before GAHT and a prospective study on the effects of GAHT over time were performed. Transgender and cisgender people completed several psychometric tests: the AQ, the Gender Dysphoria Questionnaire for Adolescents and Adults (GIDYQ-AA), the Body Uneasiness Test (BUT), the Toronto Alexithymia Scale (TAS), the Liebowitz Social Anxiety Scale (LSAS) and the Symptom Checklist 90 revised (SCL-90-R). A total sample of 789 persons (n=229 cismen;n=172 ciswomen;n=206 transmen;n=182 transwomen) referring to the Florence and Rome Gender Clinics was enrolled. Of these, 62 participants referring to the Florence Gender Clinic were evaluated in a prospective study at baseline and 12 months after GAHT. Results Groups showed significant differences in terms of autistic traits: ciswomen showed lower scores of AQ while cismen reported higher scores of AQ than all other groups (p<0.001). Transgender individuals showed significant higher levels of Gender Dysphoria (GD; GIDYQ-AA) and body uneasiness (BUT) than the other groups (p<0.001). Considering alexithymia (TAS) and social anxiety (LSAS), transgender individuals scored significantly higher when compared to cisgender ones (p<0.001 and p<0.05 respectively). No significant differences in general psychopathology (SCL-90-R) were found between groups (p=0.61). Across time, transmen and transwomen showed a significant reduction in AQ scores (p<0.002). The decrease in alexithymia and social anxiety after GAHT did not predict the change in AQ scores. Conclusions The autistic traits measured in our sample may represent an epiphenomenon of GD rather than being part of an Autism Spectrum Disorder (ASD) condition, since they significantly decreased after 12 months of GAHT. Conflicts of Interest The authors declare no conflict of interest.
... In recent years, there has been a strong and growing interest in the co-occurrence of Gender Dysphoria (GD) and Autism Spectrum Disorder (ASD), both in children/adolescents and adults [1][2][3][4][5][6][7][8][9]. This association is of great clinical interest because of its implications in the management of both conditions. ...
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... Notably, autistic females report higher masculinity and lower femininity than non-autistic females (Cooper et al., 2018), often prefer socializing with males than females (Bargiela et al., 2016), report elevated rates of tomboyism in childhood and adolescence (Bejerot & Eriksson, 2014;Ingudomnukul et al., 2007), and may not automatically identify with the construct of femininity as strongly as non-autistic females do (Kanfiszer et al., 2017). However, it remains unclear whether such factors are causes and/or consequences (or merely correlates) of gender diversity, and indeed autistic features (e.g., intense/obsessional interests; Landén & Rasmussen, 1997;Zucker et al., 2017) could be mistaken for gender dysphoria symptoms and vice versa (Heylens et al., 2018). Additionally, other social processes (e.g., social and communication difficulties associated with autism ;Landén & Rasmussen [1997]) may play important roles in explaining the link between autism and gender dysphoria. ...
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Childhood gender variance (GV) is associated with autism spectrum disorder (ASD) diagnosis/traits; however, this association has mainly been investigated in clinical samples. An ASD screening measure based on 10 items from the commonly used Child Behavior Checklist (CBCL) might enable investigation of this association in a wider variety of (non-clinical) populations where the CBCL and a measure of GV are available. We investigated whether GV in 6- to 12-year-olds (N = 1719; 48.8% assigned male at birth) from a community sample showed an association with the CBCL 10-item ASD screener. The Gender Identity Questionnaire for Children measured GV. The CBCL 10-item ASD screener measured ASD traits. The remaining CBCL items provided a measure of children's general emotional and behavioral challenges. Higher GV was associated with higher CBCL ASD screener scores, including when controlling for the remaining CBCL items. The CBCL 10-item ASD screener can be useful for investigating the link between GV and ASD traits in 6- to 12-year-olds. Given that the CBCL is commonly employed, secondary analyses of existing datasets that also included a measure of GV could enable investigation of how widely the association between GV and ASD applies across a variety of populations.
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Evidence indicates an overrepresentation of youth with co-occurring autism spectrum disorders (ASD) and gender dysphoria (GD). The clinical assessment and treatment of adolescents with this co-occurrence is often complex, related to the developmental aspects of ASD. There are no guidelines for clinical care when ASD and GD co-occur; however, there are clinicians and researchers experienced in this co-occurrence. This study develops initial clinical consensus guidelines for the assessment and care of adolescents with co-occurring ASD and GD, from the best clinical practices of current experts in the field. Expert participants were identified through a comprehensive international search process and invited to participate in a two-stage Delphi procedure to form clinical consensus statements. The Delphi Method is a well-studied research methodology for obtaining consensus among experts to define appropriate clinical care. Of 30 potential experts identified, 22 met criteria as expert in co-occurring ASD and GD youth and participated. Textual data divided into the following data nodes: guidelines for assessment; guidelines for treatment; six primary clinical/psychosocial challenges: social functioning, medical treatments and medical safety, risk of victimization/safety, school, and transition to adulthood issues (i.e., employment and romantic relationships). With a cutoff of 75% consensus for inclusion, identified experts produced a set of initial guidelines for clinical care. Primary themes include the importance of assessment for GD in ASD, and vice versa, as well as an extended diagnostic period, often with overlap/blurring of treatment and assessment.
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The current literature shows growing evidence of a link between gender dysphoria (GD) and autism spectrum disorder (ASD). This study reviews the available clinical and empirical data. A systematic search of the literature was conducted using the following databases: PubMed, Web of Science, PsycINFO and Scopus; utilizing different combinations of the following search terms: autism, autism spectrum disorder (ASD), Asperger's disorder (AD), co-morbidity, gender dysphoria (GD), gender identity disorder (GID), transgenderism and transsexualism. In total, 25 articles and reports were selected and discussed. Information was grouped by found co-occurrence rates, underlying hypotheses and implications for diagnosis and treatment. GD and ASD were found to co-occur frequently - sometimes characterized by atypical presentation of GD, which makes a correct diagnosis and determination of treatment options for GD difficult. Despite these challenges there are several case reports describing gender affirming treatment of co-occurring GD in adolescents and adults with ASD. Various underlying hypotheses for the link between GD and ASD were suggested, but almost all of them lack evidence.
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Purpose: Transgender and gender nonconforming people who fulfill diagnostic criteria for autism spectrum disorders (ASDs) often present to mental health providers with concerns that are distinct from those without ASDs. Gender Dysphoria (GD) and ASDs have been proposed to share etiologic mechanisms and there is evidence that ASDs may be more common in transgender and gender nonconforming people. We explore the impact of ASD characteristics on individual gender identity, expression, and the process of psychotherapy. Method: The authors present two case studies of high-functioning individuals with ASD and GD diagnoses. Results: The limited ability to articulate an inner experience, deficits in Theory of Mind (ToM), along with the intolerance of ambiguity as a manifestation of the cognitive rigidity characteristic of ASDs, may present special difficulties to gender identity formation and consolidation and create challenges in psychotherapy. Conclusions: The authors suggest that ASDs do not preclude gender transition and that individuals with high-functioning ASDs are capable of making informed decisions regarding their medical care and life choices. The authors also consider possible challenges and suggest techniques for assisting such clients in exploring their gender identities.
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Purpose: There is evolving evidence that children and adolescents with gender dysphoria have higher-than-expected rates of autism spectrum disorder (ASD), yet clinical data on ASD among youth with gender dysphoria remain limited, particularly in North America. This report aims to fill this gap. Methods: We conducted a retrospective review of patient chart data from 39 consecutive youth ages 8 to 20 years (mean age 15.8 years, natal male: n = 22, natal female: n = 17) presenting for evaluation at a multidisciplinary gender clinic in a large U.S. pediatric hospital from 2007 to 2011 to evaluate the prevalence of ASD in this patient population. Results: Overall, 23.1% of patients (9/39) presenting with gender dysphoria had possible, likely, or very likely Asperger syndrome as measured by the Asperger Syndrome Diagnostic Scale (ASDS). Conclusion: These findings are consistent with growing evidence supporting increased prevalence of ASD in gender dysphoric children. To guide provision of optimal clinical care and therapeutic intervention, routine assessment of ASD is recommended in youth presenting for gender dysphoria.
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The Rorschachs of 14 feminine boys were intensively studied. Relative to the records of normal subjects, feminine boys displayed an inordinate investment in quasi-human at the expense of fully human responses. When percepts had individuals in interaction, the preponderance of these interactions were malevolent. Boundary disturbances, as indicated by lapses of thought organization, were commonplace. Females were frequently represented in idealized, stereotypical, or caricatured ways. Lastly, overt gender confusion was seen in single percepts of humans. It is proposed that these results provide evidence of severely disturbed internalized object representations. As such, boyhood femininity is inextricably linked to psychopathology and not a secondary consequence of peer rejection, as has been argued by others.
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Recent studies have reported a high prevalence of autistic spectrum conditions (ASCs) in adults with gender dysphoria (GD) versus the general population (Jones et al., 201215. Fombonne, E. (2009). Epidemiology of pervasive developmental disorders. Pediatric Research, 65, 591–598. doi: 10.1203/PDR.0b013e31819e7203View all references; Pasterski, Gilligan, & Curtis, 2014). This study utilized snowball sampling to collect data on gender identities, self-reported diagnoses of ASCs, and AQ-10 scores of individuals with GD. Of 446 respondents, 14% reported an ASC diagnosis. Higher AQ-10 scores were observed in those defining as male (M = 5.66, 95% CI [5.10, 6.22]) versus those defining as female (M = 4.25, 95% CI [3.55, 4.35]) and in those assigned female-at-birth (M = 5.67, 99.9% CI [4.91, 6.43]) versus those assigned as male-at-birth (M = 4.11, 99.9% CI [3.53, 4.69]). The genderqueer group (M = 5.73, 95% CI = [5.20, 6.26]) had the highest observed mean AQ-10 score. This study has implications for the management of those with GD and for researchers owing to both the large number of nonbinary individuals identified and to the complexities identified surrounding language when researching this group.
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Introduction: There is a growing clinical recognition that a significant proportion of patients with gender dysphoria have concurrent autism spectrum disorder (ASD). Aim: The purpose of this review is to systematically appraise the current literature regarding the co-occurrence of gender dysphoria and ASD. Methods: A systematic literature search using Medline and PubMed, PsycINFO, and Embase was conducted from 1966 to July 2015. Main outcome measures: Fifty-eight articles were generated from the search. Nineteen of these publications met the inclusion criteria. Results: The literature investigating ASD in children and adolescents with gender dysphoria showed a higher prevalence rate of ASD compared with the general population. There is a limited amount of research in adults. Only one study showed that adults attending services for gender dysphoria had increased ASD scores. Another study showed a larger proportion of adults with atypical gender identity and ASD. Conclusion: Although the research is limited, especially for adults, there is an increasing amount of evidence that suggests a co-occurrence between gender dysphoria and ASD. Further research is vital for educational and clinical purposes.