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Characteristics of restricted interests in girls with ASD compared to boys: a systematic review of the literature

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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 evidence 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.
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European Child & Adolescent Psychiatry (2024) 33:987–1004
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 / Published online: 30 May 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
Autistic spectrum disorders (ASD) are pathologies belong-
ing to neurodevelopmental disorders. About four men are
diagnosed against one woman (diagnosis of pervasive devel-
opmental disorders with Kanner’s criteria, Lotter’s and Rut-
ter’s definitions before 1982, and DSM-based definitions or
ICD-10 since 1990; studies published between 1966 and
1999) [1] and up to six against one in the absence of intel-
lectual deficiency (same author as the previous review: addi-
tion of studies published between 1998 and 2008; diagno-
sis of Asperger syndrome with DSM-III or -IV or ICD-10)
[2]. ASD diagnosis is clinical, and clinical profiles of this
disorder can be very heterogeneous, hence the term “spec-
trum”. It was previously defined according to the DSM-
IV [3] as a triad of main symptoms involving qualitative
impairment in social interaction, qualitative impairments
in communication and restricted repetitive and stereotyped
patterns of behaviours, interests, and activities. Since 2013,
the DSM-5 [4] has grouped this triad under two criteria:
(A) persistent deficits in communication and social interac-
tions, and (B) restricted and repetitive behaviours, interests
and activities. Among the items for criterion B, two items
out of four need to be detected for the criterion to be met.
Criterion B3 is as follows: highly restricted, fixated interests
that are abnormal in intensity or focus (e.g. strong attach-
ment to or preoccupation with unusual objects, excessively
circumscribed or perseverative interests).
Restricted interests are not specific to ASD, they can be
observed in children with intellectual deficiency without
ASD, and even in children with normal development and
intelligence [5].
* Lise Bourson
Lise.bourson@gmail.com
1 The Université de Paris, Paris, France
2 Université Paris Saclay - CESP INSERM U1178, CCU-AH
attheCentre Hospitalier Fondation Vallée, Gentilly, France
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
... The authors claim that the higher proportion of externalizing behaviors in girls in this sample may imply a referral bias towards girls with higher behavioral difficulties and reflect under-referrals of girls with different clinical presentations. Meanwhile, Bourson and colleagues performed a literature review of 19 studies to describe the characteristics of restricted interests in girls with ASD [2]. Despite the higher source of bias depending on the instruments and the different ways to administer them, their results show fewer restricted interests in girls than in boys and indicate that girls' interest differed also qualitatively: they are closer to neurotypical girls and found a differential association by sex. ...
... The authors suggest that these associations may be linked to sex differences in the hypothalamic-pituitary-adrenal axis and brain maturation during adolescence, but also point at differences between both sexes in stress response strategies. Results from these studies [1][2][3][4][5] call for a more thorough investigation of sex differences in child psychiatry, particularly in disorders whose prevalence varies by sex, such as neurodevelopmental disorders. ...
... Many studies report a tendency for males to score higher than females on quantitative measures of RRBs, including the RBS-R [15]. More recently, there has been a suggestion that behaviors most common in females may not be represented on the instruments used to evaluate RRBs, thereby contributing to the misconception that females have fewer RRBs [16,17]. This certainly could contribute to differences identified on the RBS-R. ...
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Assessment of restricted, repetitive behaviors (RRB) in autism evaluations often assumes that these behaviors negatively impact the individual. Qualitative studies of first-person accounts indicate the negative impact of the stigma associated with RRBs but also provide insights into the positive aspects. The current study explores how framing response options as negative (i.e., level of problem associated with occurrence) or positive (i.e., level of benefit associated with occurrence) affects RRB self-reports in autistic adults. Sixty-six autistic adults aged 18–59 filled out the Repetitive Behavior Scale-Revised (RBS-R) and a modified RBS-R+, assessing problems and benefits of reported behaviors, respectively. There was a moderate to strong correlation between the forms, each assessing problems and benefits in terms of the number of behaviors endorsed (r = 0.746) and the levels of benefits and problems (r = 0.637). Autistic adults reported a higher number of RRBs in the form that assessed problems, but the number of behaviors was comparable between the forms when counting in the response option of the occurrence of behavior without having a benefit. Despite some variability in the level of problems and the benefits across the subdomains of RRB, autistic adults largely rated comparable levels of associated benefits and problems, highlighting the complexity of RRBs as having both positive and negative impacts. Future screening and diagnostic tools for adults should aim to assess both positive and negative aspects of autistic features to afford a more nuanced understanding of individual experiences while still yielding diagnostically relevant information. Qualitative studies are needed to better understand the complex experiences associated with these behaviors; however, it may be important to ensure that options for endorsement of behaviors without a specific benefit are also needed to ensure some behaviors (e.g., self-injurious behaviors) are not missed.
... As well, longitudinal studies which track predictor of repetitive behavior. This is an important omission to note given the associations between sex and gender differences and different manifestations of repetitive behaviors often seen between autistic men and women (Bourson & Prevost, 2022). Further, only two studies included participants with an intellectual impairment or below average intelligence, despite individuals with intellectual differences representing as much as 38% of the autistic community (Maenner et al., 2023). ...
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... In addition, the clinical presentation of the core symptoms of ASD can vary in males and females with ASD. Females with ASD may have fewer or less pronounced restricted interests and repetitive behaviors (reviewed in ref. [3]) and may demonstrate stronger social competence compared to males with ASD (reviewed in ref. [4]). The need to study both males and females with ASD has been recognized. ...
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Sex-based differences in the prevalence of autism spectrum disorder (ASD) are well-documented, with a male-to-female ratio of approximately 4:1. The clinical presentation of the core symptoms of ASD can also vary between sexes. Previously, positron emission tomography (PET) studies have identified alterations in the in vivo levels of translocator protein (TSPO)—a mitochondrial protein—in primarily or only male adults with ASD, with our group reporting lower TSPO relative to whole brain mean in males with ASD. However, whether in vivo TSPO levels are altered in females with ASD, specifically, is unknown. This is the first pilot study to measure in vivo TSPO in the brain in adult females with ASD using [ ¹¹ C]PBR28 PET-magnetic resonance imaging (MRI). Twelve adult females with ASD and 10 age- and TSPO genotype-matched controls (CON) completed one or two [ ¹¹ C]PBR28 PET–MRI scans. Females with ASD exhibited elevated [ ¹¹ C]PBR28 standardized uptake value ratio (SUVR) in the midcingulate cortex and splenium of the corpus callosum compared to CON. No brain area showed lower [ ¹¹ C]PBR28 SUVR in females with ASD compared to CON. Test-retest over several months showed stable [ ¹¹ C]PBR28 SUVR across time in both groups. Elevated regional [ ¹¹ C]PBR28 SUVR in females with ASD stand in stark contrast to our previous findings of lower regional [ ¹¹ C]PBR28 SUVR in males with ASD. Preliminary evidence of regionally elevated mitochondrial protein TSPO relative to whole brain mean in ASD females may reflect neuroimmuno-metabolic alterations specific to females with ASD.
... For example, autistic girls are less capable of identifying relational conflict than neurotypical girls [35]. Compared to autistic boys, autistic girls may be better at initiating friendships, but may have more difficulties maintaining these friendships [37]. The core symptom of 'repetitive and restricted patterns of behaviour' is significantly less commonly present, less pronounced and different in presentation in autistic women compared to autistic men [38,39]. ...
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... In addition, the study findings not only show the learning capabilities of autistic young people, but also their narrow topics of interest. These topics of interest may be a good avenue for communication, but social relationships may be quite constrained if they do not find other people who share the same interests, resulting in a loss of interest in any other topic of conversation [32,33]. Moving on to feelings and affective-sexual relationships, these difficulties with communication and social interaction represent both intra-and interpersonal barriers [7] while decreasing their opportunities to obtain sex-related information from their classmates and peers [34]. ...
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The purpose of this work is to accomplish earlier identification of females and those with more nuanced autistic traits. This chapter lays the groundwork for early identification with comprehensive evaluation for young children, with more details in subsequent chapters for school-age children, adolescents, and adults. Barriers to identification, performance of screeners and measures in female children, first-person accounts, and a discussion of comprehensive evaluations are included. Implementation information (e.g., screeners and measures) are given as a guide to building a comprehensive evaluation battery for the assessment of females and other nuanced cases. Finally, directions for support needs are included.KeywordsAutismFemaleNuancedAssessmentComprehensive evaluationScreeningSurveillanceIdentificationAdaptive
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Objective Restricted and repetitive pattern of behaviours and interests (RRB) are a cardinal feature of autism spectrum disorder (ASD), but there remains uncertainty about how these diverse behaviours vary according to individual characteristics. This study provided the largest exploration to date of the relationship between Repetitive Motor Behaviours, Rigidity/Insistence on Sameness and Circumscribed Interests with other individual characteristics in newly diagnosed individuals with ASD. Method Participants ( N = 3,647; 17.7% females; Mage = 6.6 years [ SD = 4.7]) were part of the Western Australian (WA) Register for ASD, an independent, prospective collection of demographic and diagnostic data of newly diagnosed cases of ASD in WA. Diagnosticians rated each of the DSM‐IV‐TR criteria on a 4‐point Likert severity scale, and here we focused on the Repetitive Motor Behaviours, Insistence on Sameness and Circumscribed Interests symptoms. Results The associations between RRB domains, indexed by Kendall's Tau, were weak, ranging from non‐significant for both Circumscribed Interests and Repetitive Motor Behaviours to significant (.20) for Insistence on Sameness and Repetitive Motor Behaviours. Older age at diagnosis was significantly associated with lower Circumscribed Interests and significantly associated with higher Insistence on Sameness and Repetitive Motor Behaviours. Male sex was significantly associated with higher Repetitive Motor Behaviours but not Insistence on Sameness or Circumscribed Interests. Conclusions The pattern of associations identified in this study provides suggestive evidence for the distinctiveness of Repetitive Motor Behaviours, Insistence on Sameness and Circumscribed Interests, highlighting the potential utility of RRB domains for stratifying the larger ASD population into smaller, more phenotypically homogeneous subgroups that can help to facilitate efforts to understand diverse ASD aetiology and inform design of future interventions.
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Purpose The purpose of this paper is to address the need for increased understanding, awareness and recognition of the autism female phenotype in terms of repetitive behaviours and restricted interests (RBRIs). Design/methodology/approach A systematic PRISMA review was conducted. The main aim of the present systematic review is to identify studies which have investigated RBRIs in females with autism spectrum disorder (ASD) or the differences in RBRIs between males and females with ASD. Findings In sum, 19 relevant articles were identified: 5 studies found no significant evidence to support the notion of sex differences in RRBIs in ASD; 1 study did not report any differences in RRBIs between males and females with ASD; 12 studies found evidence that males with ASD had significantly more RRBIs compared to females with ASD; and, lastly, 1 study found that girls with ASD have features of RRBIs which are exhibited more compared to boys with ASD. Research limitations/implications There is a real lack of in-depth knowledge and understanding of the female phenotype of ASD, and such lack of knowledge has a detrimental impact on the identification of autistic females and a lack of identification can have negative consequence. This is important to address in future research as it is well established that the earlier the diagnosis, the better the outcomes, due to the timely access to appropriate interventions. Practical implications The RBRIs exhibited in autistic females are not sufficiently captured by most currently diagnostic instruments. Clinicians are less likely to identify the RBRIs in females as they tend not to be the typical repetitive behaviours commonly associated with ASD. It has been recommended that clinicians consider “females as a whole” in terms of their clinical presentation and look for any indication of RBRIs, even repetitive interests which appear clinically innocuous. Originality/value There is relatively little research investigating RBRIs in autistic women and girls. There is a real need to highlight the importance of understanding and recognising how RBRIs can differ between males and females with ASD.
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Purpose Sex differences in circumscribed interests (CI) may delay diagnosis for females with autism spectrum disorder (ASD); therefore, it is important to characterize sex differences in CI to determine if differential approaches to diagnostic assessment are warranted for females with ASD. The purpose of this paper is to examine sex differences in parent-reported quantity, content and functional impairment of children’s interests. Design/methodology/approach Parent responses to the Interests Scale were analyzed using descriptive statistics and ANOVAs to determine diagnostic (ASD vs typical development (TD)) and sex differences between four groups of children ages six to ten years: ASD males, ASD females, TD males and TD females. Findings Groups were comparable on the quantity of interests reported on the Interests Scale. Children with ASD demonstrated significantly more nonsocial interests and had greater functional impairment associated with their interests than TD children. A significant diagnosis×sex effect was found for the number of interests in folk psychology. Descriptively, males with ASD were more likely to have a primary interest in the traditionally male category of physics than females with ASD whose primary interest mainly fell into the categories of TV or the more traditionally female category of psychology. Originality/value These findings strengthen the results of Turner-Brown et al . (2011) by replicating their findings that children with ASD have more nonsocial interests and greater functional impairments related to their interests compared to TD children in a sample that is balanced on biological sex. However, there are distinctions between males and females with ASD in their primary interests that have implications for diagnostic assessment.
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Previous work has found gender differences in restricted and repetitive behaviors and interests (RRBI) for autism spectrum disorder (ASD). Compared to girls, affected boys have increased stereotyped and restricted behaviors; however much less is known about gender differences in other areas of RRBI. This study aims to identify whether specific RRBI (i.e., stereotyped, self‐injurious, compulsive, insistence on sameness, ritualistic, and restricted), as measured by item‐level data on the Repetitive Behavior Scale‐Revised (RBS‐R), can distinguish girls from boys with ASD. Participants included 615 individuals with ASD (507 boys; 82.4%), ages 3–18 years of age (M = 10.26, SD = 4.20), who agreed to share data with the National Database for Autism Research (NDAR). Multivariate analysis of variance and discriminant function analysis (DFA) were used to determine whether item‐level RBS‐R data could correctly classify cases by gender. DFA results suggest that RBS‐R items significantly differentiate gender. Strongly differentiating RBS‐R items had greater success in correctly classifying affected boys (67.90%) than girls (61.00%). Items that best‐discriminated gender were heightened stereotyped behaviors and restricted interests items in boys and compulsive, sameness, restricted, and self‐injurious behavior items in girls. This study is the first to find that girls with ASD may have increased compulsive, sameness, and restricted RRBI compared to boys. Additionally, findings support heightened self‐injurious behaviors in affected girls. Future research should disentangle whether elevated rates of RRBI in girls are central to the presentation of ASD in girls or an epiphenomenon of the high rates of co‐occurring disorders (e.g., anxiety) noted in girls. Autism Res 2018. © 2018 International Society for Autism Research, Wiley Periodicals, Inc. Lay Summary This study is the first to examine a comprehensive measure of repetitive behavior in children with autism, with findings of increased compulsive, insistence on sameness, and self‐injurious behavior characterizing girls and increased stereotyped and restricted behavior characterizing boys. Future research should determine whether these elevated behaviors in girls are directly part of the autism presentation in girls or symptoms of co‐occurring psychopathology. It is important for autism diagnostic measures to best capture the types of repetitive behavior girls may demonstrate.
Article
Previous research found repetitive and restricted behaviors (RRBs) were less predictive of Autism Spectrum Disorder (ASD) in females, indicating the diagnostic construct may not adequately describe RRB presentations in females. This mixed-methods study investigated the female presentation of RRBs, namely restricted interests, in a clinic sample of 125 participants (n = 40 female; ages 2–83 years; 75 ASD). RRB severity did not differ between sexes, t = 1.69, p = 0.094, though male participants scored higher on the Restricted Behavior subscale. Qualitatively, females demonstrated a narrower range of restricted interests and expressed them in a socially oriented manner compared to males. The results suggest unique quantitative and qualitative sex differences in RRB profiles that could shed light on the female ASD phenotype.
Article
Background: Autism is a neurodevelopmental disorder with various clinical presentations. It has been historically considered a male disorder. An increasing number of authors stress the existence of sex/gender bias in prevalence and the need to define sex/gender differences in the clinical presentation. Review: Recently, an increasing number of authors have studied the impact of sex/gender on autism's clinical presentation. The sex ratio of four boys to one girl commonly reported in literature is questioned. Sociocultural and familial influences can impact female clinical presentation as well as the way the difficulties of girls with autism are perceived. Issues of autism diagnostic instruments such as sex/gender bias are also studied since they have an impact on the access to diagnosis for girls. Clinical variability is a part of autism spectrum disorder, but some traits appear to be more specific of the female phenotype: existence of a "camouflage" phenomenon and less unusual play or restricted interests. Discussion: Better understanding and diagnosis of females with autism is required to ensure the access to the support and treatment they need. Professionals must apprehend the sex/gender clinical differences to prevent the frequent misdiagnosis or missed diagnosis of females with autism. Conclusion: Pursuing research on sex/gender differences seems necessary to ensure appropriate support and diagnosis of undiagnosed females.
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Compared to the social communication domain, considerably less is known about the cause, development, and impact of restricted, repetitive behaviors interests and activities in children with autism spectrum disorder, including possible sex differences. This study examined sex differences in clinically identified (Autism Diagnostic Observation Schedule) restricted and repetitive behavior symptoms using the largest known sample (N = 1024) of age-matched and intelligence quotient–matched female and male children with autism spectrum disorder. More similarities than differences were observed; however, younger higher functioning and older lower functioning females presented reduced rates on the Autism Diagnostic Observation Schedule restricted and repetitive behavior subcategory unusually repetitive/excessive, stereotyped behaviors compared to similar males. These findings identify key restricted and repetitive behavior similarities and differences among young females and males with autism spectrum disorder and emphasize the need for a deeper understanding of the female autism phenotype.
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Research studies using existing samples of individuals with autism spectrum disorders have identified differences in symptoms between males and females. Differences are typically reported in school age and adolescence, with similarities in symptom presentation at earlier ages. However, existing studies on sex differences are significantly limited, making it challenging to discern if, how, and at what point in development females with autism spectrum disorder actually exhibit a different behavioral presentation than males. The purpose of this study was to gather impressions from a large group of clinicians to isolate specific areas for future study of sex differences. Clinicians were surveyed about their opinions and perceptions of symptom severity in females, as compared to males, at different points during development. They were also asked to provide open-ended responses about female symptom presentation. Consistent with previous literature, clinicians noted more sex-related differences in restricted and repetitive behaviors and fewer differences for social communication features. Differences were most commonly observed in school age and adolescence, suggesting this time period as a critical and particularly vulnerable window for females with autism spectrum disorder. The results are discussed in the context of other male/female differences across development so that more targeted investigations of autism spectrum disorder sex differences across development.