Characteristics of Restricted Interests in Girls With ASD Compared To Boys: A Systematic Review of The Literature

European Child & Adolescent Psychiatry
https://doi.org/10.1007/s00787-022-01998-5
REVIEW
Characteristics of restricted interests in girls with ASD compared

to boys: a systematic review of the literature
Lise Bourson1 · Camille Prevost2
Received: 12 July 2021 / Accepted: 23 April 2022

© The Author(s), under exclusive licence to Springer-Verlag GmbH Germany 2022
Abstract
The existence of a female phenotype profile in autistic spectrum disorder is one of the current hypotheses to explain the
diagnostic discrepancy between men and women. In this context, an international literature review was carried out to evi-
dence and describe the characteristics of restricted interests found in girls with autistic spectrum disorder. A documentary
search was conducted on PubMed and a systematic literature review was carried out based on the PRISMA methodology. We
selected studies with a population of boys and girls diagnosed as autistic according to the DSM-IV or the DSM-5, in which
quantitative and descriptive comparisons of restricted interests, according to gender were carried out. Nineteen studies were
found to be relevant. Fifteen enabled a refining of the characteristics of restricted interests among females: fewer restricted
interests were identified in comparison with boys, and the autistic girls’ interests seem to be closer to those of neurotypical
girls than to those of autistic boys, which thus led to more complex screening. Age and Intelligence quotient seem to be two
factors that trigger variations in restricted interests differently according to gender. Representations among professionals also
have an impact on diagnoses among girls. For future research, one of the perspectives could be a comparison between girls
with autism and neurotypical girls to limit gender bias. The present results contribute to potentially extending knowledge of
a female phenotypical profile in autism and show the need to improve the general population’s awareness, to improve health
professionals’ training and possibly to revise the diagnostic tools.
Keywords Autistic spectrum disorder · Diagnosis · Girl · Women · Restricted interests · Circumscribed interests
Introduction disorder can be very heterogeneous, hence the term “spec-

trum”. It was previously defined according to the DSM-
Autistic spectrum disorders (ASD) are pathologies belong- IV [3] as a triad of main symptoms involving qualitative
ing to neurodevelopmental disorders. About four men are impairment in social interaction, qualitative impairments
diagnosed against one woman (diagnosis of pervasive devel- in communication and restricted repetitive and stereotyped
opmental disorders with Kanner’s criteria, Lotter’s and Rut- patterns of behaviours, interests, and activities. Since 2013,
ter’s definitions before 1982, and DSM-based definitions or the DSM-5 [4] has grouped this triad under two criteria:
ICD-10 since 1990; studies published between 1966 and (A) persistent deficits in communication and social interac-
1999) [1] and up to six against one in the absence of intel- tions, and (B) restricted and repetitive behaviours, interests
lectual deficiency (same author as the previous review: addi- and activities. Among the items for criterion B, two items
tion of studies published between 1998 and 2008; diagno- out of four need to be detected for the criterion to be met.
sis of Asperger syndrome with DSM-III or -IV or ICD-10) Criterion B3 is as follows: highly restricted, fixated interests
[2]. ASD diagnosis is clinical, and clinical profiles of this that are abnormal in intensity or focus (e.g. strong attach-
ment to or preoccupation with unusual objects, excessively
circumscribed or perseverative interests).
* Lise Bourson Restricted interests are not specific to ASD, they can be
[email protected] observed in children with intellectual deficiency without
1 ASD, and even in children with normal development and
The Université de Paris, Paris, France
intelligence [5].
2
Université Paris Saclay ‑ CESP INSERM U1178, CCU-AH
at the Centre Hospitalier Fondation Vallée, Gentilly, France
13
Vol.:(0123456789)
In the last few years, the male/female ratio has been the The inclusion criteria were: populations comprising boys
subject of debate, following the hypothesis that women with and girls; patients with a diagnosis of autism spectrum dis-
ASD tend to be underdiagnosed, and diagnosed later. Kim order according to the DSM-IV (-TR) [3] and/or DSM-5
et al. [6] and Mattila et al. [7] observed male/female ratios criteria [4]; existence of a quantitative or descriptive com-
of 2.5:1 and 1.8:1, on the basis of the DSM-IV (diagnosis of parisons on the restricted interests of the patients according
autistic disorder; cohort of 7-12 years old in a South Korean to gender/sex; articles published in English or French; and
community) and DSM-IV-TR (diagnosis of ASD; population retrospective and prospective observational studies. For a
of 8 years old in Finland), respectively. study to be included, it must positively meet all the inclusion
Kopp et Gillberg [8] have already raised the hypothesis of criteria listed.
a particular female profile based on six clinical observations It is worth noting that we did not make any difference
of girls with ASD. This could enable a reduction in the male/ between the terms “gender” and “sex” in this article, as the
female diagnostic discrepancy. preferential use of one or the other varied greatly depending
Kirkowski et al. [9] recognized the existence of a behav- on the authors.
ioural phenotype of ASD that differs between girls and boys, Comparisons were made either with the tools usually used
while the diagnostic criteria were established mainly on the for ASD diagnoses (ADI-R, Autism Diagnostic Interview-
basis of symptoms observed among boys—for instance, Revised, for instance), or with tools that were more specific
Kanner’s observations in 1943. to RIs (RBS-R, the Repetitive Behaviour Scale-Revised, for
The items of the criterion "persistent deficits in commu- example). Patients were directly examined by a practitioner
nication and social interactions" have been the subject of and given a diagnosis, or a questionnaire was filled in by the
many studies in recent years to compare their differences child’s parents, by their schoolteachers or by health profes-
according to gender [12]. This is not the case for the cri- sionals usually in charge of their care.
terion “restricted and repetitive behaviours, interests and The exclusion criteria were: articles published in a lan-
activities”. Van Wijngaarden-Cremers’ meta-analysis (2014) guage other than English or French; literature reviews and
[13] and Allely’s systematic review (2019) [14] evidenced meta-analyses; articles where the main topic was not the
the fact that women with ASD presented lower scores on clinical aspects of autism (neurobiology, epidemiology,
the “restricted, repetitive behaviours or interests” criteria and drugs, for example) and articles dealing with clinical
compared to men with ASD. Leekam et al. [15] in 2011 had description in the field of another pathology, for example
already concluded that each sub-type of this criterion should genetic studies; studies that did not include a mixed male/
be differentiated, as the difference could vary for each one of female population; studies that did not carry out compari-
them. Details of the female phenotypic profile still remain sons on restricted interests according to gender.
unclear today. Given the limited availability of the literature on this sub-
In this context, it seemed interesting to study “restricted ject, no restrictions on publication dates or the age range
interests” (RIs) more specifically (item B.3. in the DSM-5), of the populations studied were included, but most studies
for which Kreiser et White [16] in 2014 and Lai et al. [17] were carried out on paediatric populations (only 2 studies
in 2015 laid the foundations. A literature review was thus included adults in their population [19, 20]).
carried out to evidence and describe, in type of interests A growing interest in the subject has been observed over
and quantitative terms, the differences in RIs found in the about 10 years now (results of our search: 3 articles pub-
literature according to gender, in the setting of ASD. lished in 2010 against 38 in 2020).
To ensure the inclusion of all relevant articles, the biblio-
graphic references in the articles identified were examined
Patients and method after the selection phase.
A flowchart (Fig. 1) summarizes the different stages, from
The PRISMA method was used to guide our research meth- identification to inclusion.
odology (Appendix) [18].
The documentary search was conducted on PubMed,
using the following research equation (latest research update Results
27/04/2021): ((autism[Title/Abstract]) AND ((gender[Title/
Abstract]) OR (sex[Title/Abstract]) OR (females[Title/ In all, 19 articles were identified as relevant for this review.
Abstract])) AND ((restricted repetitive behaviour) OR Table 1 summarizes the main data extracted from each
(restricted interests))). study (characteristics of the study, the population, the com-
As the search was less conclusive using the MeSH term parison, etc.). It is divided into four parts: the first three parts
“Autism Spectrum Disorder”, it was decided not to use it in concern the studies that have at least a quantitative com-
the search equation. parison of restricted interests by gender (studies showing a
13
Fig. 1 Flowchart
significant difference, or a predictive association between Wang et al. [32] concluded that the ADI-R was less sen-
gender and RIs, studies showing the absence of significant sitive for diagnosis among girls, particularly for domain C
difference or the absence of predictive association, and “RRB” (repetitive behaviour and restricted interests).
studies showing a significant difference but only for part of Jamison et al. [33] were interested in clinicians’ percep-
their results) and the last part concerns the two studies that tions of a difference, or absence thereof, in clinical autism
have only a descriptive comparison (the type of interests by between girls and boys. It is important to be aware that 86%
gender). of the respondents were female clinicians. A majority of
Table 2 summarizes the main results extracted from each the respondents stated they had not adjusted their diagnos-
study: quantitative comparison of RIs by gender (details, if tic evaluation methods to gender, but about a third of them
available, on the presence or absence of RIs, their number, relied on their clinical impression. Jamison et al. deline-
intensity, evolution by age or IQ), descriptive comparison of ated four age ranges for the questionnaire: infancy, primary
RIs by gender, and particularities of each study. school age, adolescence and adulthood. Most responses for
Out of the 19 articles, 15 [19, 21–34] found a significant these four age ranges showed similar severity between girls
difference in their results for RIs between girls and boys, and boys concerning RIs, but for each age range, 33%, 38%,
using a quantitative measure, and/or on the nature and con- 31% and 22% of the respondents, respectively, considered
tent of the RIs. that girls presented lower levels of RIs than boys. The pri-
Thirteen articles [19, 23–34] found a significant quantita- mary school period was the one where this difference was
tive difference in RIs between girls and boys (the differences more marked. Conversely, the period in which the severity of
in some cases only concerned part of the RI criterion). This RIs seemed greater among girls than among boys was ado-
difference was in favour of less marked RIs among girls. lescence—despite the fact that the percentage of respondents
RIs were either less pronounced in terms of quantity or the was very low (6%).
RI criterion was not met, or only partially met among girls, Four articles [20, 35–37] did not describe any significant
unlike boys. For details of the results of each study, see difference in RIs according to gender, or any predictive asso-
Table 2. ciation. For details of the results of each study see Table 2.
The following are comments that we found relevant to The following is a comment that we found relevant to one
some of the studies reviewed. of the studies we reviewed.
13
Table 1 The main data extracted from each study (quantitative comparison & descriptive comparison of restricted interests according to gender)
Author(s) and Study country ASD popula- Diagnostic classi- Group of compari- Mean age (M) ± SD IQ of the patients Type of meas- Person filling the
[year] tion (n = ; % girls fication ± subcat- son (ASD boys? [interval] (years) ure of restricted questionnaire /per-
13
(= number of egory of ASD TD girls? TD interests (RI) with son interviewed
girls)) girls + boys?) comparisons girls/
boys
Studies with significant results (quantitative comparison)

Szatmari et al. North America & 2028; 20% DSM-IV g ASD vs b ASD M = 8.5 ± 5.6 SD 66%: IQ ≥ 70 "BEH" scale Parent interview
[2011] Europe (= 406) subgroup on IQ 34%: IQ < 70 (= repetitive
behaviour)
from the ADI-R
Hiller et al. Australia 138; 50% DSM-IV-TR or g ASD vs b ASD g: M = 8.06 ± 4.03 (n = 97) DSM-IV-TR and/ Parent- and teacher-
[2014] (= 69) DSM-5 SD g: M = 97.76 ± 15.31 or DSM-5 (with report
High-Functioning b: M = 8.76 ± 3.91 SD an RI list) questionnaire
ASD SD b: M = 100.31 ± 13.27
SD
Frazier et al. USA 2418; 13% DSM-IV-TR g ASD vs b ASD g: M = 9.32 ± 3.67 g: Total IQ: "RBS-R" scale: Parent-report ques-
[2014] (= 304) / SD M = 74.7 ± 27.59 "restricted inter- tionnaire
b: M = 9.01 ± 3.56 SD ests"
SD b: Total IQ:
[4 to 18] M = 82.56 ± 27.59
SD
Fulceri et al. Italy 79; 19% DSM-IV-TR g ASD vs b ASD M = 4.3 NV IQ: M = 90.9 ± "RBS-R" scale: Parent-report ques-
[2015] (= 15) / & [1.9 to 5.9] 20.6 SD "restricted inter- tionnaire
g + b ASD vs g + b ests"
TD
Hiller et al. Australia 152; 40% (DSM-5 severity g ASD vs b ASD M = 10.94 ± 3.11 SD population without 40-item online Parent-report ques-
[2015] (= 60) scale) [6 to 17] ID survey: includ- tionnaire
/ ing "From 3
to 5 years of
age was your
child unusu-
ally interested
in mechanical
objects such
as the stove or
vacuum?"
+ item with free-
report on child's
main obsession
(type of RI)
Table 1 (continued)
boys
Frazier and USA 2643; 13% DSM-IV-TR g ASD vs b ASD g: M = 9.23 ± 3.77 V IQ: g: item 40 RBS-R: Parent-report ques-
Hardan [2016] (= 352) / SD M = 74.1 ± 32.2 SD "Preoccupation tionnaire
b: M = 9.01 ± 3.55 b: M = 78.7 ± 30.9 with Subject" & parent interview
SD SD + item 68 ADI-R:
[4 to 17] NV IQ: g: "Circumscribed
M = 77.9 ± 26.1 SD Interests"

b: M = 85.7 ± 26.0 + item 39 SRS:
SD "Narrow Inter-
ests"
Beggiato et al. France & Switzer- 1) 594; 22% DSM-IV-TR g ASD vs b ASD 1) M = 10.7 ± 6.5 SD 1) M = 56.3 ± 29.2 items ADI-R Parent interview
[2016] land (= 129) / 2) g: SD (domain C)
2) 1716; 20% (= 338) M = 9.2 years ± 4.4 2) g: M = 92.1 ± 22.8 "restricted inter-
SD SD ests" (RI)
b: M = 9.4 years ± 4.4 b: M = 93.7 ± 22.7 + "unusual preoc-
SD SD cupations" (UP)
Antezana USA 615; 18% DSM-IV-TR g ASD vs b ASD M = 10.26 ± 4.2 SD (n = 495 (80.8%)) "RBS-R" scale: Parent-report ques-
et al. [2018] (= 108) / [3 to 18] M = 88.14 ± 25.0 SD "restricted inter- tionnaire
ests"
McFayden USA 75; 27% DSM-5 g ASD vs b ASD M = 12.43 [2 to 57] g: M = 103.13 ± 15.55 "RBS-R" scale: Parent-report ques-
et al. [2018] (= 20) / (78% = < 17 SD "restricted inter- tionnaire
years) b: M = 91.80 ± 18.01 ests"
SD + descriptive
coding: 10 cat-
egories of RI
Studies with an absence of significant results or difference / prediction according to gender (quantitative comparison)
Mayes and USA 777; 16% DSM-IV g ASD vs b ASD M = 6.5 ± 3.2 n = 461: IQ ≥ 80 Item from the Parent and teacher
Calhoun [2010] (= 122) subgroup on IQ [1 to 17] n = 316: IQ < 80 CASD: "Obses- interview
sions
(restricted interests
and fixations)"
Solomon et al. USA 40; 50% DSM-IV-TR g ASD vs b ASD g: M = 12.0 ± 3.42 g: Total IQ: "RBS-R" scale: Parent-report ques-
[2011] (= 20) / & g ASD vs g TD SD M = 104.2 ± 15.29 "restricted inter- tionnaire
b: M = 12.45 ± 3.72 SD ests"
SD b: Total IQ:
[8 to 18] M = 103.95 ± 16.87
SD
13
Table 1 (continued)
13
boys
Anthony et al. USA 109; 15% DSM-IV-TR g ASD vs b ASD M = 12.70 ± 3.79 SD Total IQ: "Interests Scale" Parent-report ques-
[2013] (= 16) High-Functioning & g + b ASD vs [7.13 to 22.92] M = 111.56 ± 17.48 (Bodfish) tionnaire
ASD g + b TD SD
[73 to 159]
Uljarevic et al. Western Australian 3 647; 17.7% DSM-IV-TR g ASD vs b ASD M = 6.56 ± 4.7 SD (n = 1554) DSM-IV-TR Scale completed
[2020] (= 640) / Total IQ: "restricted inter- by clinicians (the
M = 82.7 ± 22.2 SD ests" presence and
(including 451 severity)
(28.8%): IQ < 70)
Studies including one part with a significant results and one part with non-significant results (quantitative comparison)
Nicholas et al. USA 295; 49% DSM-IV-TR g ASD vs b ASD 8 60.4%: QI ≤ 70; DSM-IV-TR Scale completed by
[2008] (= 145) subgroup on IQ 39.6%: IQ > 70 "restricted inter- clinicians
ratio b:g for IQ ≥ ests"
70 = 4.9:1
and for
IQ < 70 = 2.4:1
Wang et al. China 1064; 21% DSM-IV-TR g ASD vs b ASD g: (V): M= Population without ADI-R (domain Parent interview
[2017] (= 228) / 4.3 ± 1.5 ID C): items
(NV): M= - "restricted inter-
2.96 ± 0.9 ests" (RI)
b: (V): M= - "unusual preoc-
4.6 ± 1.4 cupations" (UP)
(NV): M= - "repetitive use
3.03 ± 0.9 of objects/inter-
[2 to 6.9] est in parts of
objects" (RUO/
IPO)
Jamison et al. USA & United 106 clinicians; DSM-5 Reflection on the / / Survey on clini- Clinicians-report
[2017] Kingdom & 86% women (= 91) / comparison: cians: difference questionnaire
Canada g ASD vs b ASD according to gen-
der for the sever-
ity of restricted
interests per
developmal age
group
Table 1 (continued)
boys
Knutsen et al. USA & Canada 1024; 50% DSM-IV or g ASD vs b ASD [2 to 12] Fr each gender: ADOS-2 RRB Clinicians assess-
[2019] (= 512) DSM-5 for each gender: - n = 243 (47.5%): IQ "Unusually ment
subgroup on IQ 68.6% = < 6 years ≥ 70 repetitive inter- (the presence and
- n = 269 (52.5%): ests or severity)
IQ < 70 stereotyped behav-
iors"
Studies with only a descriptive comparison (content of restricted interests)
Wallace Nowell USA 54; 50% DSM-5 g ASD vs b ASD g: M = 8.44 ± 1.46 (mental age from "Interests Scale" Parent-report ques-
et al. [2019] (= 27) / & SD DAS-2) (Bodfish) tionnaire
g + b b: M = 9.46 ± 0.84
ASD vs g + b TD SD
[6 to 10]
Sutherland et al. Australia 334; 51% (question about g ASD vs b ASD [5 to 18] (question about A survey devel- Parent-report ques-
[2017] (= 171) confirming (% for each age schooling oped from an tionnaire
the diagnosis of categories) / type of class) earlier pilot
ASD) study of 282 par-
ents of girls only
(open questions):
multiple-choice
questions (option
of adding
comments) and
open-ended
questions
ASD Autism Spectrum Disorder, TD Typically Developing (children), DSM Diagnostic and Statistical Manual of mental disorders, vs versus, g girls, b boys, IQ Intellectual Quotient, ID Intel-
lectual Deficiency, V verbal; NV non verbal, RBS-R Repetitive Behavior Scale-Revised, ADI-R Autism Diagnostic Interview-Revised, ADOS-2 RRB Autism Diagnostic Observation Schedule
(second edition), sub-score: Restricted and Repetitive Behavior, SRS Social Responsiveness Scale, CASD Checklist for Autism Spectrum Disorder, RI Restricted Interests, UP Unusual Preoccu-
pations, RUO/IPO Repetitive Use of Objects/Interest in Parts of Objects
13
Table 2 The main results of studies with quantitative and descriptive comparison of restricted interests by gender
Results of the girls/boys comparison for restricted interests
- Presence or absence of RI?
Author(s) Type of measure of restricted interests (RI)
- Number of RI? Descriptive comparison
and [year] with comparisons girls/boys Quantitative comparison Particularities of the study
- Intensity/severity of RI? (impact on quality of life) (content/type of RI, gender difference)
(depending on the tool used in the study)
- Evolution with age? with IQ?
STUDIES WITH SIGNIFICANT RESULTS (QUANTITATIVE COMPARISON)
BEH score (g) < BEH score (b) Rating of:
Szatmari "BEH" scale (= repetitive behaviour)
UP: p < 0.001; RI: p = 0.002 ; - the presence or absence of RI / /
and al. [2011] from the ADI-R
RUO/IPO: p = 0.03; "UP + RI": p < 0.001 - the intensity
Interest type that predicts girls: - Only High-Functioning ASD
- seemingly random (e.g. animals, rocks, shells, books, - Teachers' point of view: expressed
Rating of: stickers, pens): OR = 1.68 substantially fewer concerns for girls than for
DSM (-IV-TR / -5) "restricted, fixated interests": predicts gender
- the presence or absence of RI: criterion not met / only - toys (e.g. teddy-bear): OR = 1.33 boys
Hiller and in favour of a girl if the criterion is not met (OR = 8.32 / 4.64)
DSM-IV-TR and/or DSM-5 (with an RI list) partielly met / criterion met - 2 age categories (<7 and >7 years):
al. [2014] or only partially met (OR = 2.30 / 3.42)
- the intensity: not problematic (0) / somewhat Interest type that predicts boys: --- girls: <7 and >7 "seemingly random" = 60%
(2 categories of age)
problematic (1) / problematic (2) - specific TV program/character: OR = 1.29 (the highest % of RI type whatever the age)
- wheeled vehicles (e.g. cars): OR = 4.00 --- boys: <7 "wheeled toys" is the highest %
- screens (e.g. videogames) constant: OR = 5.00 & >7 "screens constant" is the highest %
Item is rated on a four-point Likert scale ranging

p = 0.001 (Cohen's d = - 0.13) - Equivalence measures
Frazier and - from (0) ‘‘behavior does not occur’’
"RBS-R" scale: "restricted interests" (of note: equivalence measures, therefore in favour of a / - Lower RI in females with ASD was
al. [2014] - to (3) ‘‘behavior occurs and is a severe problem"
non-equivalence of RIs among girls compared to boys) independant of reductions in IQ
(rating of the presence or absence of RI and the intensity)
Item is rated on a four-point Likert scale ranging
Fulceri and - from (0) ‘‘behavior does not occur’’ Comparison of girls and boys with ASD to TD
"RBS-R" scale: "restricted interests" p = 0.033 (Cohen's d: score = 0.58, endorsement = 0.44) /
al. [2015] - to (3) ‘‘behavior occurs and is a severe problem" girls and boys
(rating of the presence or absence of RI and the intensity)
Interest type that predicts girls:
- repetitive behaviour with a toy (e.g. teddy bears,
40-item online survey: including "From 3-5
figurines, Barbie dolls): OR = 2.87
years of age was your child unusually
"no interest": predictive odds = 3.33 (p < 0.001), therefore predicts - fascination for apparently random objects (e.g. collecting - Population of pre-school children
Hiller and interested in mechanical objects such as the
female gender if the response to the question is "no interest" / seashells /stickers/feathers): OR = 1.82 - Survey with combination of multiple-choice
al. [2015] stove or vacuum?"
(controlling for age) and free-response options
+ item with free-report on child's main
Interest type that predicts boys:
obsession (type of RI)
- fascination with wheeled-toys (cars, trucks) or parts of
those toys: OR = 13.54
RBS-R 40: "weak: χ2 = 18.28; DF = 3; p < 0.001"
item 40 RBS-R: "Preoccupation with "strong: χ2 = 8.84; DF = 7; p = 0.264"
Rating of:
Frazier and Subject" ADI-R 68: Δχ2 (p) = 23.57 (< 0.0001)
- the presence or absence of RI / - Equivalence measures
Hardan [2016] + item 68 ADI-R: "Circumscribed Interests" SRS 39: Δχ2 (p) = 10.85 (0.013)
- the intensity Differentiation in the population of verbal
+ item 39 SRS: "Narrow Interests" (of note : equivalence measures, therefore in favour of a nd non-verbal children (verbal IQ
non-equivalence of RIs among girls compared to boys) nd non-verbal IQ): "cognitively able females
2 cohorts: discriminant analysis on the first
items ADI-R (domain C) "restricted interests" Rating of:
Beggiato 1) RI: SCFC = 0.355; UP: SCFC = 0.403 * one, and then a similar analysis on an
(RI) - the presence or absence of RI /
and al. [2016] 2) RI: SCFC = 0.472; UP: SCFC = 0.244 * independent sample (second one) to validate
+ "unusual preoccupations" (UP) - the intensity
the results
Bonferroni-corrected univariate ANOVA:
- Fascination with movement of object: p < 0.01
- Preoccupation with parts of an object: p < 0.05 Item is rated on a four-point Likert scale ranging
Antezana - (Strongly attached to one object: Ps < 0.10) - from (0) ‘‘behavior does not occur’’
"RBS-R" scale: "restricted interests" / /
and al. [2018] DFA : - Strongly attached to one object: SCFC = 0.52 - to (3) ‘‘behavior occurs and is a severe problem"
- Fascination with movement of object: SCFC = - (rating of the presence or absence of RI and the intensity)
0.50 $
- Fascination wit one subject/activity: SCFC = -
Descriptive profile of RIs according to gender:
Both sexes expressed the same average number of
- girls: narrower range of RIs, mainly interested in living
interests
beings (animals, celebrities, science), able to share them
Item is rated on a four-point Likert scale ranging 4 age ranges in the population (years):
in a more sociable way than boys
McFayden "RBS-R" scale: "restricted interests" - from (0) ‘‘behavior does not occur’’ 0-6, 7-12, 13-17, 18-57
p = 0.03 - boys: rather interested in objects (vehicles, stories),
and al. [2018] + descriptive coding: 10 categories of RI - to (3) ‘‘behavior occurs and is a severe problem"
more difficult to talk about them in a suitable way in
(rating of the presence or absence of RI and the intensity) (78% = <17 years)
society
+ RRB severity composite score (RRB subscales of ADI-R
- both genders: similar interest in “screens/electronics”
and ADOS-2, RBS-R, SRS-2)
and “reading”
Table 2 (part 1) : the main results of studies with quantitative and descriptive comparison of restricted interests by gender
Results of the girls/boys comparison for restricted interests

- Presence or absence of RI?
Author(s) Type of measure of restricted interests (RI)
- Number of RI? Descriptive comparison
and [year] with comparisons girls/boys Quantitative comparison Particularities of the study
- Intensity/severity of RI? (impact on quality of life) (content/type of RI, gender difference)
(depending on the tool used in the study)
- Evolution with age? with IQ?
STUDIES WITH AN ABSENCE OF SIGNIFICANT RESULTS OR DIFFERENCE / PREDICTION ACCORDING TO GENDER (QUANTITATIVE COMPARISON)
Mayes and no significant association with gender (even after having divided the
Item from the CASD : "Obsessions 2 categories of IQ (≥ 80 and < 80): "no
Calhoun population into 2 on the basis of IQ) and gender is not significantly Rating of the presence or absence of obsessions /
(restricted interests and fixations)" significant association between IQ and RIs"
[2010] predictive of this item
Item is rated on a four-point Likert scale ranging - ASD groups were matched on IQ and age
Solomon and restricted interests: χ2 = 43.34 (p = 0.015); followed by follow-up - from (0) ‘‘behavior does not occur’’ - Comparison of girls with ASD to typically
"RBS-R" scale: "restricted interests" /
al. [2011] comparisons ( adjusted alpha): no significant difference girls/boys - to (3) ‘‘behavior occurs and is a severe problem" developing girls (no comparison specifically on
(rating of the presence or absence of RI and the intensity) IR)
Primary Interest by group and gender (%):

- g ASD: numbers/math (18.8)> music (12.5) > other (6.3) - Only High-Functioning ASD, matched group
Anthony and gender does not predict the number of interests (p = 0.54) Number of RIs - g TD: reading (18.8)> people (12.5) > other (6.3) on age and IQ
"Interests Scale" (Bodfish)
al. [2013] nor their intensity (p = 0.67) Intensity of RI - b ASD: video games (21.5)> legos (7.5)> playing games - Comparison of girls and boys with ASD to
alone (4.3) typically developing girls and boys
- b TD: sports (26.7)> reading (8.3)> video games (6.7)
- b & g: a significant linear regression line for the RI score

(a decrease of 0.012 units a year) from the age of 8
DSM-IV-TR "restricted interests" - 4 ages ranges in the population (years):
- no significant association between male gender and restricted - b & g: a score of 2 out of 3 for intensity of RIs reached the
+ 4-point Lik ert severity scale: from 0-3, 4-6, 7-12 and >13
Uljarevic et interests (OR = 0.93, 95% CI [0.79-1.09]) highest percentage across all age ranges, whether or not
0 ‘Criteria definitely NOT met’ / - Distinction between the presence or absence
al. [2020] - g & b: older age at the time of diagnosis was significantly there was ID
to 3 ‘Clearly meets criterion of ID
associated with a lower score for RI - the higher the IQ and the older the boy, the fewer the RIs
(to an extreme degree)' - 11% of the population = ADHD comorbidity
(intensity), especially after the age of 13 (not statistically
significant for girls)
STUDIES INCLUDING ONE PART WITH A SIGNIFICANT RESULTS AND ONE PART WITH NON-SIGNIFICANT RESULTS (QUANTITATIVE COMPARISON)
Interest in IQ: "in the IQ >70 range, the
Nicholas and RI: p = 0.136 male:female prevalence ratio was 4.9:1,
DSM-IV-TR "restricted interests" / /
al. [2008] + preoccupation with parts of objects: p = 0.009 whereas in the IQ <70 range, the male:female
prevalence ratio was 2.4:1"
ADI-R (domain C) : items
RI: verbal children (VC): p = 0.011
- "restricted interests" (RI) Rating of:
Wang and non-verbal children (NVC): p = 0.141 Distinction between verbal and non-verbal
- "unusual preoccupations" (UP) - the presence or absence of RI /
al. [2017] UP: (VC): p = 0.005; (NVC): p = 0.032 children
- "repetitive use of objects/interest in parts of - the intensity
RUO/IPO: (VC): p < 0.001; (NVC): p < 0.001
objects" (RUO/IPO)
Decrease of the RI intensity according to gender (clinicians' Open-ended responses on RIs in ASD girls compared to
- Interest in clinician's perceptions of a
- early childhood : 33% "fewer" and 65% "similar" point of view): ASD boys: “RIs more socially accepted”, “more typical for
survey on clinicians: difference according to difference (or not) in clinical of ASD between
Jamison and - school age : 38% "fewer" and 59% "similar" - girls had less severe RIs in the primary school period than the age range in infancy”, “less obvious”, “more intense”,
gender for the severity of restricted interests girls and boys
al. [2017] - adolescence : 31% "fewer" and 63% "similar" boys "fewer in number or inexistent”, “not always unusual in
per developmal age group - 4 age ranges in the population (years):
- adulthood : 22% "fewer" and 76% "similar" - this difference was reversed during adolescence, where nature so that they merge with their peers, or neglected
0-5, 6-11, 12-17, ≥18
the clinicians perceived more severe RIs among girls interests”
2-6 years-old girls and IQ ⩾ 70: p = 0.041 (OR = 0.45)

ADOS-2 RRB "Unusually repetitive interests Rating of:
Knutsen and 2-6 years-old girls and IQ < 70: p = 0.537
or - the presence or absence of RI / - 2 categories of IQ:
al. [2019] 6-12 years-old girls and IQ ⩾ 70: p = 0.317
stereotyped behaviors" - the intensity
6-12 years-old girls and IQ < 70: p = 0.033 (OR = 0.27)
STUDIES WITH ONLY A DESCRIPTIVE COMPARISON (CONTENT OF RESTRICTED INTERESTS)
Primary Interest by gender (%):
- g: TV (36) > psychology (e.g. playing with Barbie dolls,
Wallace
figurines, relationships) (22) > games (15) = manual Comparison of girls and boys with ASD to TD
Nowell "Interests Scale" (Bodfish) / /
activities (15) > physics (11) = language/reading (11) girls and boys
and al. [2019]
- b: games (33) > physics (e.g. trains, Lego) (30) > sport
(7) = mathematics (7)
A survey developed from an earlier pilot Interests (%): Boys vs Girls & Interest type that predicts girls:
Sutherland study of 282 parents of girls only - art: OR = 0.257
/ / - books: OR = 0.400
and al. [2017] (open questions) : MCQ (option of adding
comments) and open-ended questions - singing/music: OR = 0.192
Interest type that predicts boys:

- technology: OR = 4.193
- dinosaurs: OR = 4.353
Table 2 (part 2) : the main results of studies with quantitative and descriptive comparison of restricted interests by gender - transport: OR = 46.962
The table 2 legend:

- ASD = Autism Spectrum Disorder
- TD = Typically Developing (children)
- DSM = Diagnostic and Statistical Manual of mental disorders
- ADHD = Attention Deficit Hyperactivity Disorder
- g = girls
- b = boys
- IQ = Intellectual Quotient
- ID = Intellectual Deficiency
- RBS-R = Repetitive Behavior Scale-Revised
- ADI-R = Autism Diagnostic Interview-Revised
- ADOS-2 RRB = Autism Diagnostic Observation Schedule (second edition), sub-score: Restricted and Repetitive Behavior
- SRS = Social Responsiveness Scale
- CASD = Checklist for Autism Spectrum Disorder
- SCFC = Standardized Canonical Function Coefficient
- MCQ = Multiple-Choice Questions
- RI = Restricted Interests
- UP = Unusual Preoccupations
- RUO/IPO = Repetitive Use of Objects/Interest in Parts of Objects
- * (SCFC) = positive coefficient mean that boys had higher item scores than girls
- $ (SCFC) = negative coefficient mean that boys had higher item scores than girls
- OR = Odds Ratio
- TV = television
Characteristics of restricted interests in girls with ASD compared to boys: a systematic review of the literature; European Child and Adolescent Psychiatry; L. Bourson, intern in child psychiatry,
Université de Paris, France (corresponding author: [email protected]), and C. Prevost, CCU-AH at the Centre Hospitalier Fondation Vallée, 7 rue Benserade 94250 Gentilly, France Université Paris Saclay CESP INSERM U1178
13
Table 2 (continued)
ASD Autism Spectrum Disorder, TD Typically Developing (children), DSM Diagnostic and Statistical Manual of mental disorders, ADHD
Attention Deficit Hyperactivity Disorder, g girls, b boys, IQ Intellectual Quotient, ID Intellectual Deficiency, RBS-R Repetitive Behavior
Scale-Revised, ADI-R Autism Diagnostic Interview-Revised, ADOS-2 RRB Autism Diagnostic Observation Schedule (second edition), sub-
score: Restricted and Repetitive Behavior, SRS Social Responsiveness Scale, CASD Checklist for Autism Spectrum Disorder, SCFC Standard-
ized Canonical Function Coefficient, MCQ Multiple-Choice Questions, RI Restricted Interests, UP Unusual Preoccupations, RUO/IPO Repeti-
tive Use of Objects/Interest in Parts of Objects, *(SCFC) positive coefficient mean that boys had higher item scores than girls, $(SCFC) negative
coefficient mean that boys had higher item scores than girls, OR Odds Ratio, TV television
Regarding the results of Uljarevic et al. [37], for both fascination with one subject and therefore ask several ques-
genders, older age at the time of diagnosis was signifi- tions (e.g.: ADI-R, RBS-R).
cantly associated with a lower score for restricted interests According to the study, some of the questionnaires are
(OR = 0.96; 95% CI [0.94–0.97]). Uljarevic et al. described a intended for parents, teachers or health professionals caring
significant linear regression line for the RI score (a decrease for this patient profile. The questions are either closed or
of 0.012 units a year) from the age of 8. A score of 2 out of 3 open ended with the possibility of giving examples.
for intensity of RIs reached the highest percentage across all All these elements are a source of bias in the interpreta-
age ranges in girls as in boys, whether or not there was intel- tion of the results. These different variables are also to be
lectual deficiency. A measure of total IQ was available for taken into account in the development of future studies, to
1554 out of 3647 patients, of whom 28.8% presented a total specify which aspect of the restricted interest is compared
IQ < 70. Uljarevic et al. found that an increase in one meas- and thus facilitate the conclusions.
urement unit for the intensity of RIs in men was significantly Despite the large number of variables to be interpreted
associated with a lower score for total IQ (non-normalised in this review, we have tried to bring out some key ideas.
coefficient—3.74; 95% CI [− 5.11, − 2.38]) and increased
probability (OR = 1.52, 95% CI [1.31–1.77]) of a lower IQ.
Effect sizes were smaller and not statistically significant for Restricted interests evolve with age
women, but the trend was the same.
Seven articles [19–22, 24, 27, 33] included a qualitative Several studies [33–35, 37] separated their populations
description of the nature and the content of RIs, with a dif- by age range to compare RIs according to gender, and
ference found according to gender. For details of the results Solomon et al. [36] and Hiller et al. [24, 27] performed a
of each study, see Table 2. matching on age before making the comparisons. This idea
of separating populations into age groups seems relevant
to us to follow the evolution of RIs with the child’s devel-
Discussion opment (the evolution of interests with age in neurotypical
children) and with the pathology. More precise compari-
The variability of measurement instruments sons can thus be made to evidence any differences. Four
for the comparison of RI age ranges appear relevant for the study: infancy (0- to
5-year-olds), primary school age (6- to 11-year-olds), ado-
Comparisons were made using very different types of meas- lescence (12- to 17-year-olds) and adulthood (18 years and
ures in these 19 studies. Table 2 summarizes the type of over). In studies showing a significant quantitative differ-
measure of RI used in each study and what the measurement ence in RIs according to gender, the “primary school age”
the tool explores: quantitative and/or descriptive aspects. group proves to be the most widely pinpointed. This con-
The DSM criteria, for instance, only explore the quanti- curs with Uljarevic’s descriptions [37] mentioned above
tative aspect: the presence or absence of RI, as well as its in the results section of this article, and in particular a
intensity. The Interest Scale adds a descriptive dimension to significant linear-regression line from the age of 8 years.
its comparison: the presence or absence of 41 categories of Jamison et al. [33] highlighted this decrease according to
RI must be rated, and for the three main interests, the inten- gender: according to the opinions of the clinicians who
sity and impact on daily life of the interest must be rated. stated they had observed a difference, girls had less severe
The RBS-R scores the presence of RI in the past month, RIs in the primary school period than boys, but this dif-
while the Interest Scale scores its presence in the past or ference was reversed during adolescence, where the cli-
present. nicians perceived more severe RIs among girls. Seltzer
Some scales, such as the CASD, have a single global item et al. [38] drew the same conclusions on the evolution of
on RI (less precise), while others differentiate preoccupation symptoms with age, including the evolution of repetitive
with parts of an object, strong attachment to one object and behaviour and RIs in particular. One of the hypotheses
13
to explain this inversion could be the ability to “camou- However, the autism spectrum is broad and covers clinical
flage” (a learned adaptation, by imitation of behaviours profiles with differences in intellectual levels and adaptation
expected in relational terms or in society), which is more strategies.
frequently deployed and more efficient in girls with a Our recommendations for future studies would be to take
good intellectual level in the primary school period, but IQ into consideration in the comparison of RIs, to prevent it
which is overtaken by expectations from society and by from biasing the results (matching or IQ ranges).
relational issues during adolescence [10, 11, 39]. This
hypothesis could also explain Knutsen’s results [34], Should we make comparisons according to gender
which only showed a significant difference for girls under or compare children with ASD to neurotypical
6 years with an IQ ≥ 70 and 6- to 12-year-old girls with children to avoid a gender bias?
an IQ < 70 (the ability to camouflage for 6- to 12-year-old
girls with IQ > 70 becomes insufficient, less efficient and We carried out a review on the comparison between girls and
IR increases in adolescence). boys with ASD. This comparison generated a gender bias,
Our recommendations for future studies would be to because there are already differences according to gender
take age into consideration in the comparison of RIs, among neurotypical children in their centers of interest.
to prevent it from biasing the results (matching or age To limit this bias, we need to consider further research com-
ranges—infancy, primary school age, adolescence and paring girls with ASD to neurotypical girls and ASD girls to
adulthood). ASD boys. Our results appear to corroborate this idea that RIs
in ASD girls are closer to those of neurotypical girls than to
Is IQ a variable to be taken into consideration? those of ASD boys. Some of the authors whose study includes
a descriptive comparison detail this finding. Wallace Nowell
Intuitively, and because we found RIs among children with et al. [21] hypothesize a continuum for the interests of females
intellectual deficit but not ASD, we could hypothesize that with ASD between those of males with ASD and TD females.
the lower the IQ, the more restricted are the interests. Sutherland et al. [22], Hiller et al. [24] and Jamison's study
Is it the case? [33] have a concurring point of view. They find a difference
In all the studies with a quantitative comparison (except in the subjects of interest for girls with ASD and boys with
for Jamison et al. [33]), the authors were looking for a direct ASD: girls with ASD would be more engaged in the types of
estimation of their patients’ IQ, even though this data was interests generally expected of their gender. The younger the
not always available. The definition of intellectual deficit girls, the greater would be this difference.
is an IQ < 70, and it was used as an exclusion criterion in Given the current gender ratio in ASD, we understand
two studies [27, 32]. In the 14 studies with direct screening the difficulties for these past studies in including a sufficient
for IQ without the exclusion of intellectual deficiencies, 12 number of girls with ASD to carry out this type of analysis
presented a population with an average IQ of 70 or over; on a large scale. The definition of a female phenotypical
however, the intervals were sometimes wide, with IQs as profile could enable underdiagnoses to be reduced among
low as 50 in Frazier’s study [25, 28], or conversely, reach- girls with ASD and would therefore facilitate inclusion in
ing around 120 in Solomon’s study [36]. Uljarevic et al. studies. This female phenotypical profile contains, among
[37] integrated IQs into his comparison of RIs according other things, the adaptive abilities of girls with ASD.
to gender in his supplementary calculations, and found the Our recommendations for future studies would be to com-
following results for boys: the higher the IQ and the older pare girls with ASD to neurotypical girls and girls with ASD
the boy, the fewer were the RIs, especially after the age of to boys with ASD.
13 years. This difference was non-significant in girls, despite
a tendency towards it. Mayes et Calhoun [35] did not find Could the clinicians’ point of view influence
any significant association between IQ and RIs. diagnosis?
Following analysis, and in spite of our hypothesis, IQ
does not emerge as a variable enabling a significant result Jamison et al. [33] focused on the perceptions of clini-
to be detected for RIs in the studies selected. cians who had been recruited on a voluntary basis. 85%
A total IQ for a child with ASD is often difficult to obtain of respondents were women. Female clinicians seem to
because of heterogeneous inter-indexes, which could be a show greater interest in the search for potential differences
bias in our analysis. between girls and boys with ASD, and therefore more read-
Furthermore, certain studies have included populations ily evidence a female autistic profile if there is one. We can
with a wide IQ range and have not systematically included include ourselves in this remark, as both of us are female
a matching on this variable in their methodology, or IQ cat- clinicians. Moreover, among the first authors of the 19 arti-
egories for comparison. cles analysed, 13 were women.
13
This interest can also be observed among female patients varied according to the age of the population and its intel-
who have created social movements to promote their differ- ligence quotient.
ence and therefore reduce delays in their diagnosis, a delay Gould [41] commented the paper by Duvekot et al. [42]
that often leads to a negative impact on quality of life. We on the measurement bias related to the usual tools in his arti-
can quote for instance the “Aspergirl” or “Aspie” movement cle, which leads to an underestimation of the numbers girls
for Asperger’s syndrome. This neologism was invented by who in fact meet the diagnostic criteria: “A very important
Liane Holliday Willey, doctor in education and researcher, finding was that girls were less likely to receive an ASD
and diagnosed with the illness in 1999. Patients can also take diagnosis based on the standardised diagnostic instruments.
part in research by sharing their experience in qualitative Diagnostic instruments and/or manuals need to be adapted
research [11] (website: http://autisminpink.net), and write to provide examples that are more characteristic of girls.
books and blogs to share their story with others. This is particularly the case for special interests and rou-
Therefore, author/reviewer gender may lead to a greater tines.”. Fulceri and al. [26] set an example for the RBS-R
sensitivity in identifying a diagnosis of autism in girls. scale: “A possible explanation that concurs to the higher
prevalence of RRB in ASD males than ASD females might
A quantitative difference in RIs according to gender: be that the items of RBS-R are, almost in part, male-ori-
how relevant are the usual diagnostic tools? ented (for example, the restricted interests behaviors sub-
scale frequently refer to cars, trains, dinosaurs), and thus
Most studies have shown a difference in RIs between girls they may under-detect RRB in females with ASD. This
and boys: girls have fewer and less intense RIs and they do possible gender bias should be adequately considered in
not meet the diagnostic criteria for the RI item on the usual the interpretation of results and would benefit from includ-
diagnostic tools (ADOS, ADI-R, DSM criterion), contrast- ing female-specific items in the questionnaires addressed
ing with boys [19, 23–34]. for ASD participants”. These comments potentially help to
RI measure tools differed across the studies analysed understand why a majority of the published studies use the
(refer to Table 1 for details). Some authors used the usual RBS-R scale. This would perhaps make it easier to find a
diagnostic tools (ADI-R, ADOS, DSM criterion) [23, 24, 29, difference by gender due to measurement bias. However,
31, 32, 34, 37], but these are less sensitive to the female pro- study results showing a difference are more easily published
file because they are mainly based on clinical observations (i.e. publication bias). The instruments usually used in the
of boys with ASD. Other authors used more specific tools diagnostic process, particularly in France the ADOS and
for RIs [19, 20, 25, 26, 30, 36], such as the RBS-R scale with the ADI-R, are less represented in the results of our review.
the “restricted interests” item and Bodfish’s “interest scale” This may be due to results that show no difference and lead
[40], but they have been seldom used in France; dedicated to less publication. We mentioned above that these tools are
questionnaires could be created for a study [22, 27, 33]. less sensitive to the female phenotypic profile, so this also
The RBS-R scale is the most represented with 6 studies contributes to their underrepresentation. To overcome these
out of 19 that use it. Among the six studies, five studies problems, two authors make suggestions:
found a significant result and one study (Solomon and al. Beggiato et al. [29] suggested integrating correction fac-
[36], 40 patients) did not fiund a significant result or predic- tors according to gender for ASD diagnosis in the use of
tion according to gender (see Table 2). the ADI-R tool, to limit gender bias. They specified: “Spe-
Among the four studies with an absence of significant cifically, the scoring weights of items related to restricted
difference and an absence of predictive association accord- behaviors may be different according to patient’s gender
ing to gender, we can observe that some have large samples (n°67 “unusual preoccupations”, n°68 “restricted inter-
such as Uljarevic and al. [37] (n = 3647) and Mayes and ests” and n°69 “repetitive use of objects or interest in parts
Calhoun [35] (n = 777) and, respectively, used the "RI" item of objects”). […] With our current analysis, we were not
of the DSM-IV-TR and the CASD item: "Obsessions (RI and able to determine precisely which and how items should
attachments)". be re-scored depending on gender. Further studies includ-
Among the four studies including one part with signifi- ing subjects around the threshold of ASD assessed with
cant results and another part with non-significant results, standardized methods will provide a gender-neutral ADI-R
we can note that some have large samples such as Wang and algorithm.”
al. [32] (n = 1064) and Knutsen and al. [34] (n = 1024), and Frazier et al. [27] suggested it would be wise to include
respectively used some ADI-R’s items and ADOS-2 RRB female-specific RI exemplars in new or revised versions of
“unusually repetitive interests or stereotyped behaviors”. diagnostic instruments and autism symptom questionnaires,
In Wang's study, results were not significant for non-verbal and particularly in the ADI-R tool to facilitate scoring for
patients. In Knutsen's study, the significance of the results girls.
13
In the light of our review, it seems essential that an adjust- interests in girls with ASD are not adequately captured by
ment of the diagnostic tools commonly used is needed for most current diagnostic instruments and are less likely to
the female phenotype, to limit underdiagnosis. Attwood, be recognized by clinicians as being characteristic of ASD.
Garnett and Rynkiewicz in 2011 created a screening ques- It is therefore important to consider the pre-existing gen-
tionnaire adapted to the female phenotypical profile: the der bias, societal expectations and the camouflage effect as
Girls’ Questionnaire for Autism Spectrum Conditions (GQ- elements limiting the identification and access to the diag-
ASC) [43]. nosis of girls with ASD.
Does society play a role in the underdetection of RIs

A difference in RI content and nature according among girls with ASD and in the camouflage effect?
to gender: a gender bias? A camouflage effect?
A societal bias? Camouflage and discrimination—people with ASD justify
the implementation of camouflage as a response to relational
Depending on studies, interest in technologies/television/ and societal expectations in social situations. Camouflage
video games/screens is either the main interest in percent- is motivated by a desire to be integrated among others, to
ages for both girls and boys [21], or is identified as an inter- be an active member of society and establish links, but it
est predictive of male gender (5 times more predictive of is also implemented to reduce feelings of stress and avoid
the male gender after the age of 7 in Hiller’s study [24] and dangerous situations (discrimination, harassment, aggres-
4 times more during primary school for Sutherland et al. sive attacks). It can be grouped under two strategies: the
[22]). Children’s screen time may be a bias and increase compensation using the most appropriate expressions; plan-
this interest. ning each conversation, and the masking avoiding being “too
The other types of interest seem to differentiate according autistic”—playing the most appropriate role to the situation.
to gender, with interests potentially closer to those of neuro- However, this strategy of adaptation to our society leads to
typical girls of the same age, such as dolls, animals, manual fatigue and anxiety, or even depression; it affects patients'
activities, objects to be collected, music or reading [21, 22, self-image, with a loss of identity. It may limit the support
24]. It would seem that this differentiation occurs at least they receive as a result of lack of perception by others of
during infancy and school age, with girls then restricting their difficulties. It would seem that women are more able
themselves to these interests and with a discrepancy occur- to use camouflage, even though the desire to do so can con-
ring later compared to neurotypical girls—as mentioned in cern either gender. People with ASD have suggested that
the responses to Jamison’s questionnaire [33]. improving the general public’s awareness and acceptance
These RIs could be less clearly perceived by parents, of their symptoms would improve their social experiences,
teachers or professionals, insufficiently informed on the their integration and relationships, without having to resort
female profile, particularly because these RIs are more to camouflage to reach these objectives [44].
socially acceptable. Hence, RIs could be less intense in girls, Indeed, the desire to have a social relationship can materi-
who are more able to share their interests with others in alize in people with ASD, but it is limited by their difficulties
society. McFayden et al. [19] concluded that girls were more in social interaction and communication. This is aggravated
interested in living beings than boys who showed more inter- by negative preconceived ideas or negative first impressions
est in objects. Dinosaurs and transport appear as two other that neurotypical people may have about them (speech and
types of RI predictive of male gender, 4 and 46 times more pronunciation, facial expressions, gaze, body postures, cloth-
in boys than in girls, respectively, according to Sutherland ing, etc.) [45].
et al. [22]! It is therefore important to remain aware of this Camouflage appears as one of the hypotheses to explain
difference in RI profiles according to gender so as to detect diagnostic discrepancies in ASD according to gender [16,
them better. Assessment is carried out on their intensity, 17, 39].
their repetitive nature (there is not necessarily a symbolic Because RIs are by definition abnormal, and therefore
play), and their pervasion in daily life. This assessment are likely to be negatively perceived, people with ASD are
would enable girls with ASD to fill the RI criterion at the more likely to try and hide them, to avoid standing out in
time the diagnosis is mentioned. society and avoid negative repercussions. However, RIs
Gould and Ashton-Smith [39] agree with these comments can be used as an asset in patient care. Indeed, RIs can be
after studying the factors limiting or distorting the diagnosis a means for people with ASD to revitalize themselves by
of girls with ASD: girls with ASD have interests closer to withdrawing into themselves and into an activity. This with-
those of neurotypical girls. What differentiates their interests drawal enables the decrease of the external information flow
is the quality and intensity of these interests in addition to which is difficult to process simultaneously. It is one of their
the time they dedicate to them. They point out that repetitive self-anxiolytic techniques. RIs are also source of pleasure
13
and shape an important part of their lives. RIs can therefore observing and reporting behaviors, may have an influence on
provide a basis for initiating therapy and helping a patient symptom documentation and interpretation. Hirt explains: the
with ASD to enter into relationships, with this interest as a retrieval of original information from the subject's memory
starting point, alternating time for the relationship time and is impacted by the expectations he will have when he tries to
time alone to refuel. remember it. “Over the two reported experiments, we have
SCHOOL: Camouflage strategy also has an effect at obtained strong evidence in support of the proposed expec-
school in the identification of girls with ASD by teachers. tancy-guided retrieval model. In all conditions, the outcome
In fact, teachers reported substantially fewer concerns with information and subjects' expectancies guided the retrieval
girls’ behaviour than with boys. Girls with ASD show more process, resulting in expectancy-consistent recall of the past.
commonly appropriate classroom behaviour, and their learn- Moreover, the results of Experiment 2 indicated that subjects
ings are less impacted by their restricted interests in class generate and use their own expectancies under conditions in
[24]. Girls with ASD tend to be with their peers during which they are not explicitly provided with an expectancy.”
school break time, sharing similar interests and activities, We noted above that the gender of the person who identi-
but they alternate between being jointly engaged and with- fies or performs the diagnosis has an impact on her expecta-
drawn from the relationship close to the group; boys with tions of the diagnostic result. We mentioned that women
ASD, on the other hand, tend to play very solitary games are more proficient in looking for clinical differences for the
which are less socially accepted. This difference leads to a female phenotypic profile (detection and diagnosis). There-
weaker detection of girls by the school staff, if they have not fore this person's gender has an impact on what they are
been trained about these subtleties [10]. looking for (evocation of the diagnosis, making the diagno-
PARENTS: What is frequently found in the literature is sis), but also on their memories to answer the questionnaires
a presentation with more externalized symptoms in boys during the diagnostic process.
(hyperactivity, impulsiveness, etc.), compared to girls, who Today, there is a lack of qualitative studies concerning
tend to have more internalized symptoms, whatever the type gender-related social representations of the different pro-
of pathology or the culture [46, 47]. tagonists involved in symptom detection and diagnosis.
Duvekot et al. [42] showed that it was necessary for girls Therefore, the adaptation strategies of children with ASD,
to present considerable emotional and behavioural disorders and the identification capacities of the different protagonists
to increase the probability of parental concern and therefore (parents, school staff and health professionals) are key ele-
the chance of access to a diagnosis. ments for girls with ASD to have access to diagnosis.
There can be a discrepancy between what parents report
and what is clinically visible during assessment, according
to the child’s gender with ASD. Holtmann et al. [48] explain Limitations
this as an “interpretation bias” among ASD children’s par-
ents, who may expect more sociably acceptable behaviours We conducted our literature review on a single database, so
from their daughters than their sons. They may therefore this is a limitation to our results.
overlook the detection of internalized symptoms in their The term “restricted interests” is not a MeSH term; there-
daughters, as these symptoms are not disruptive in society. fore it was difficult in the implementation of a research equa-
PROFESSIONALS: Clinicians are more likely to rank tion to obtain a sufficiently sensitive equation. This difficulty
boys in the ASD category than girls, even when both gen- was increased by the fact that, depending on countries and
ders present symptoms associated with disorders detected the period of time, the term has evolved to become “cir-
by the school or by professionals (intellectual deficiency for cumscribed interests”, probably in an effort to de-stigmatize,
instance). There are therefore preconceived ideas among as the term “restricted” bears a negative connotation. This
professionals that have an impact on ASD diagnoses in girls explains how a certain number of articles were included on
[49–51]. Girls receiving an ASD diagnosis are on average the basis of the relevance of their references.
likely to be older than diagnosed males, to have extra needs, The definition of restricted interests has evolved over the
and they are more likely to have enhanced intellectual dis- years and is still a subject of debate as to what it should
ability [52], and emotional and behavioural disorders [42]. or should not contain; the delineation is hazy for certain
Hirt [53] proposed an expectancy-guided retrieval model in aspects, such as the stereotypies category. Similarly, the evo-
which preconceived ideas necessarily influenced perception lution of the definition of autism, with a broadening of the
and judgement. Factors such as gender of the clinician and spectrum over the course of the different DSM classifica-
gender of the child, or even gender of the parent or teacher tions, has led to a variation in the analysed populations. The
13
number of patients included was also very variable, from a perspectives towards better dissemination, better-tailored
minimum of 40–3647, and with a percentage of girls in each training on this female profile for the general public and
population ranging from 13 to 50% (out of 18 studies, 11 health professionals, a possible revision of the diagnostic
had fewer than 30% girls in their population). The different tools for the RI criterion, or the implementation of correc-
studies covered the paediatric period, and only certain stud- tion factors according to gender.
ies carried out age group comparisons or kept to a particular The objective in refining knowledge of this profile is to
age range. Furthermore, comparisons were made using very limit underdiagnosis and diagnostic delay for girls with
different types of measures, from the usual diagnostic tools ASD. Early diagnoses enable a reduction of the conse-
to the scale dedicated to RIs or questionnaires specifically quences on their quality of life: depression and anxiety dur-
devised for the study. The results of the questionnaires are ing adolescence, suffering because of their difference, dif-
impacted by the type of the person filling in the question- ficulties of integration at school and in subsequent social and
naire (professionals, parents, teachers, patient himself). working life, impact on affective relationships, etc., which
All these aspects had an impact on the comparison of stud- would then enable more adapted and earlier care.
ies and could have led to an interpretation bias in the results.
The tools used for the comparison between sex and every
study population vary greatly, which makes it more difficult Appendix: checklist with all the items
to draw a conclusion. Therefore, we have focused the discus- of the PRISMA‑P statement (2015)
sion paragraphs on relevant variables that should be consid-
ered in future studies to facilitate future review conclusions. Moher et al.: Preferred Reporting Items
Another limitation to consider in this review is the pub- For Systematic Review and Meta‑Analysis Protocols
lication bias: it is possible that we had a majority of studies (PRISMA‑P) 2015 statement. Systematic Reviews
finding a significant result because these studies are more 2015 4:1. (https://doi.org/10.1186/2046-4053-4-1)
easily published than those not finding a significant result.
This important limitation leads us to qualify the results of
our review and to give recommendations for future study
methodologies. Indeed, we cannot draw a direct conclusion Section/topic Item Checklist item
due to the reviewed studies variability. Administrative information
Title 1 “Characteristics of restricted interests in girls
with ASD compared to boys: a systematic
Conclusions review of the literature”
Registration 2 Methodology inspired by Prospero criteria
This review of the literature on 19 articles explored the dif- but not submitted to the site (staff ask
students not to submit their academic work)
ferences in restricted interests in girls compared to boys in
Authors 3 Lise BOURSON, intern in child psychiatry,
the setting of ASD. The main results were the following: a Contact Université de Paris, France (corresponding
quantitative difference with fewer RIs in girls and an unfilled Contributions author: [email protected])
or partially filled diagnostic criterion, and a difference of Garanthor of the review
kind of interests where girls’ RIs seem to be closer to neu- Camille Prevost, CCU-AH at the Centre Hos-
pitalier Fondation Vallée, 7 rue Benserade
rotypical girls’ interests than to those of boys with ASD. RIs 94,250 Gentilly, France Université Paris
were therefore less detected in girls. Saclay—CESP INSERM U1178
To confirm this result, it would be interesting in future Supervised the review process
studies to conduct comparisons with adjustment on the Amendments 4 Not concerned
“age” and “IQ” variables (matching or age and IQ ranges) Support 5 No funding was received to assist with the
to take the evolution of RIs with age and intellectual level Source preparation of this review. No sponsor
Sponsor
into consideration. It would seem relevant to compare RIs
Introduction
between girls with ASD and neurotypical girls, to gain in
sensitivity, or even to carry out a dual comparison between
girls and boys with ASD, and between girls with ASD and
neurotypical girls.
Our results contribute to potentially extending knowledge
of a female phenotypical profile in ASD and open up new
13
Section/topic Item Checklist item Section/topic Item Checklist item

Rationale 6 About four men are diagnosed against one Objectives 7 International literature review to high-
woman and up to six against one in the light and describe the characteristics of
absence of intellectual deficiency. In the restricted interests found in girls with
last few years, the male/female ratio has autistic spectrum disorder (quantitative and
been the subject of debate, following the descriptive aspects of the type of restricted
hypothesis that women with ASD tend to interests)
be underdiagnosed and diagnosed later. Methods
Kim et al. and Mattila et al. observed male/
Eligibility 8 PICO:
female ratios of 2.5:1 and 1.8:1 on the
criteria P = girls and boys with ASD, diagnosis of
basis of the DSM-IV and DSM-IV-TR,
ASD according to the DSM-IV (-TR) and/
respectively. Kopp et Gillberg have already
or DSM-5 criteria
raised the hypothesis of a particular female
I = comparison of the restricted interests of
profile based on six clinical observations
girls with ASD versus boys with ASD
of girls with ASD. This could enable a
C = comparison with the tools usually used
reduction in the male/female diagnostic
for ASD diagnoses (ADI-R “Autism Diag-
discrepancy. Van Wijngaarden–Cremers’
nostic Interview-Revised”, for instance), or
meta-analysis (2014) and Allely’s system-
with tools that were more specific to RIs
atic review (2019) evidenced the fact that
(RBS-R, “Repetitive Behaviour Scale-
women with ASD presented lower scores
Revised”, for example)
on the “restricted, repetitive behaviours or
O = to describe the particularities of the
interests” criteria compared to men with
restricted interests of girls with ASD
ASD. Details of the female phenotypic
(quantitative and descriptive aspects)
profile still remain unclear today
Inclusion criteria:
In this context, it seemed interesting to study
Populations comprising boys and girls
“restricted interests” more specifically
Patients with a diagnosis of autism spectrum
(item B.3. in the DSM-5)
disorder according to the dsm-iv (-tr) and/
Kim YS, Leventhal BL, Koh Y-J, et al. (2011)
or dsm-5 criteria
Prevalence of Autism Spectrum Disor-
Existence of a quantitative or descriptive
ders in a Total Population Sample. AJP
comparisons on the restricted interests of
168:904–912. https://doi.org/10.1176/appi.
the patients with asd according to gender/
ajp.2011.10101532
sex
Mattila M-L, Kielinen M, Linna S-L, et
Articles published in english or french
al. (2011) Autism Spectrum Disorders
Retrospective and prospective observational
According to DSM-IV-TR and Comparison
studies
With DSM-5 Draft Criteria: An Epide-
Exclusion criteria:
miological Study. Journal of the American
Articles published in a language other than
Academy of Child & Adolescent Psychiatry
English or French
50:583–592.e11. https://doi.org/10.1016/j.
Literature reviews and meta-analyses
jaac.2011.04.001
Articles where the main topic was not the
Kopp S, Gillberg C (1992) Girls with social
clinical aspects of autism (neurobiology,
deficits and learning problems: Autism,
epidemiology, and drugs, for example)
atypical Asperger syndrome or a variant
Articles dealing with clinical description in
of these conditions. European Child &
the field of another pathology, for example
Adolescent Psychiatry 1:89–99. https://doi.
genetic studies
org/10.1007/BF02091791
Studies that did not include a mixed male/
Van Wijngaarden-Cremers PJM, van Eeten
female population
E, Groen WB, et al. (2014) Gender and Age
Studies that did not carry out comparisons on
Differences in the Core Triad of Impair-
restricted interests according to gender
ments in Autism Spectrum Disorders: A
No restrictions on publication dates or the
Systematic Review and Meta-analysis. J
age range of the populations studied
Autism Dev Disord 44:627–635. https://
doi.org/10.1007/s10803-013-1913-9 Information 9 PubMed database
Allely C (2019) Exploring the female autism sources Latest research update on 27 April 2021
phenotype of repetitive behaviours and
restricted interests (RBRIs): a systematic
PRISMA review. Advances in Autism
5:171–186. https://doi.org/10.1108/AIA-
09-2018-0030
13
Section/topic Item Checklist item Section/topic Item Checklist item

Search strategy 10 Testing several research equations to evaluate Data items 12 For each study, the following data are
which one gives the most specific and sen- searched:
sitive results without losing relevant studies Authors
Final research equation: ((autism[Title/ Year of publication
Abstract]) AND ((gender[Title/ Country of the study
Abstract]) OR (sex[Title/Abstract]) OR Characteristics of the autistic population
(females[Title/Abstract])) AND ((restricted (total number, number, and percentage of
repetitive behaviour) OR (restricted inter- girls)
ests))) ASD diagnostic classification
As the search was less conclusive using the Comparison group
MeSH term “Autism Spectrum Disorder”, Average age of the population
it was decided not to use it in the research Intelligence quotient of the patients
equation Type of measure of restricted interests with
We did not make any difference between the comparisons girls/boys
terms “gender” and “sex” in this review Person filling the questionnaire/person
The term “restricted interests” is not a interviewed
MeSH term; therefor,e it is difficult in the Main results of the gender comparison
implementation of a research equation to A table is created to gather all this data
obtain a sufficiently sensitive equation. To Outcomes and 13 Primary purpose: to describe the differences
ensure the inclusion of all relevant articles, prioritization between the restricted interests of girls and
the bibliographic references in the articles boys with ASD from a quantitative and
identified were examined after the selection descriptive perspective (characteristics of
phase restricted interests in girls with ASD)
No restrictions on publication dates or the Main outcomes:
age range of the populations studied Quantitative comparison
Study records 11 Creation of a flowchart: Descriptive comparison
Identification: result of the research equation Supplementary outcomes for quantita-
Selection: read all article titles and abstracts tive comparison: absence or presence of
to assess whether the study meets the inclu- restricted interests, their intensity, their
sion criteria (including target population) number, their evolution with age and IQ
or contains exclusion criteria Risk of bias 14 Selection bias: including recruitment bias
excluded article if the study contains an in individual Measurement bias: including interviewer
exclusion criterion or if the study does not studies subjectivity, family, or patient memory bias
meet the inclusion criteria Confusion bias
in case of doubt about the relevance of the Publication bias
article: article read in full in the eligibility
Data synthesis 15 A table is created to gather all the data (see
phase
the "data items" section). The table makes
To ensure the inclusion of all relevant
it possible to improve the synthesis of the
articles, the bibliographic references in the
results: the results are more readable and
articles identified were examined after the
therefore easier to compare. A further
selection phase
description of the results is given in the
Eligibility: full-text article evaluated for
body of the article in the results section in
eligibility
a narrative form
One reviewer performs the inclusion of stud-
ies. If there is any doubt about the inclu- Meta-bias(es) 16 Not concerned
sion of a study, the opinion of the second Confidence in 17 Level of evidence difficult to quantify
reviewer is sought (read the entire article) cumulative
Inclusion: study included if relevant to the evidence
criteria of the literature review
Funding No funding was received to assist with the preparation of

this manuscript.
Availability of data and material Not applicable.
Code availability Not applicable.
Declarations
Conflict of interest The authors declare they have no conflict of inter-

est.
13
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13

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European Child & Adolescent Psychiatry

Characteristics of restricted interests in girls with ASD compared

Received: 12 July 2021 / Accepted: 23 April 2022

Introduction disorder can be very heterogeneous, hence the term “spec-

Studies with significant results (quantitative comparison)

M = 77.9 ± 26.1 SD Interests"

Item is rated on a four-point Likert scale ranging

Results of the girls/boys comparison for restricted interests

Primary Interest by group and gender (%):

- b & g: a significant linear regression line for the RI score

2-6 years-old girls and IQ ⩾ 70: p = 0.041 (OR = 0.45)

Interest type that predicts boys:

The table 2 legend:

Does society play a role in the underdetection of RIs

Section/topic Item Checklist item Section/topic Item Checklist item

Section/topic Item Checklist item Section/topic Item Checklist item

Funding No funding was received to assist with the preparation of

Availability of data and material Not applicable.

Code availability Not applicable.

Conflict of interest The authors declare they have no conflict of inter-

References protocols (PRISMA-P) 2015 statement. Syst Rev 4:1. https://​

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References protocols (PRISMA-P) 2015 statement. Syst Rev 4:1. https://