Are restricted and repetitive behaviours in two- and six-year-olds associated with emotional and behavioural difficulties?

Background: Restricted and repetitive patterns of behaviour (RRBs) serve an adaptive role in development. Elevated levels of RRBs beyond the early years, however, are associated with poorer outcome in language, cognition, and wellbeing, and are seen across a range of neurodevelopmental conditions. This study aimed to characterize the association of distinct RRB subtypes at two and six years of age, with internalising and externalising difficulties in a community sample of children. Methods: 485 parents reported on their child's insistence on sameness (IS) and repetitive sensory and motor (RSM) RRBs at two and six years of age using the Repetitive Behaviour Questionnaire (RBQ-2). Emotional and behavioural difficulties were measured using the Strengths and Difficulties Questionnaire (SDQ) at age six. Results: Consistent with previous research, RRBs later in development better predicted emotional and behavioural difficulties at age six than RRBs earlier in development. Moreover, IS RRBs were selectively associated with internalising behaviours and RSM RRBs with externalising behaviours. Importantly, these selective associations depended on when RRBs were measured. Only IS RRBs at age six were significantly associated with internalising behaviour. By contrast, while more RSM RRBs at age six were associated with higher rates of externalising behaviours, higher rates of RSM RRBs at age two were associated with fewer externalising behaviours, adding further support to the previously reported adaptive role of RRBs in early behaviour regulation. Conclusion: Although there is a need for further research to provide a detailed profile of the adaptive periods for IS and RSM RRBs, the present findings support the potential utility of elevated RRBs as a signal for emotional and behavioural difficulties at age six.

The Suicide Assessment Kit-Modified Interview: Development and preliminary validation of a modified clinical interview for the assessment of suicidal thoughts and behavior in autistic adults.

LAY ABSTRACT: People with a diagnosis of autism are at increased risk of death by suicide. There is a need for clinical instruments that are adapted to the needs of autistic people. In this study, we modified and evaluated a clinical suicide interview (Suicide Assessment Kit-Modified Interview) for use with autistic people who do not have an intellectual disability. Autistic people helped us to modify the original version of the instrument by improving the questions, providing explanations for difficult terms or concepts, and recommending that we use different rating scales. Our results support the use of Suicide Assessment Kit-Modified Interview for assessing autistic adults without intellectual disability for suicidal thoughts and behavior. In the future, we will test how well Suicide Assessment Kit-Modified Interview works in clinical settings and with different clinical populations.

Characterizing associations between emotion dysregulation, anxiety, and repetitive behaviors in autistic youth with intellectual disability.

There is a paucity of research that explores associations between emotion dysregulation and the expression and severity of core and co-occurring characteristics of autism spectrum disorder (ASD), especially in autistic youth with co-occurring intellectual disability (ID). This study explored the interplay between developmental characteristics and emotion dysregulation, anxiety, and specific subtypes of RRBs that are elevated in autistic youth with co-occurring ID. Generalized additive models demonstrated that age, sex, language level, dysregulation, and anxiety showed unique patterns of association with subtypes of RRBs in a sample of 150 autistic youth with co-occurring ID who are non-speaking or minimally verbal. More specifically, higher anxiety levels were significantly associated with elevations in repetitive sensory motor behaviors (RSMB), self-injurious behaviors (SIB), insistence on sameness (IS), and unusual interests (UI). While emotion dysregulation was a significant predictor of UI, it demonstrated positive, albeit not significant, associations with the intensity of SIB and RSMB. Language level was a significant predictor of RSMB, such that the intensity of RSMB was higher for individuals who were non-speaking relative to those who spoke in single words. These findings provide preliminary insights into patterns of associations between emotion dysregulation, anxiety, and specific subdomains of RRBs in autistic youth with ID.

Predictors of Change in Wellbeing and Mental Health of Parents of Autistic Pre-Schoolers.

Parenting is a rewarding experience but is not without its challenges. Parents of Autistic children face additional challenges, and as a result can experience lower levels of wellbeing and more mental health problems (i.e., depression, anxiety, stress). Previous studies have identified concurrent correlates of wellbeing and mental health. However, few have investigated predictors of subsequent wellbeing and mental health, or of change over time, among parents of pre-school aged autistic children. We examined child-, parent-, and family/sociodemographic factors associated with change in parents' mental health and wellbeing across three timepoints (spanning approximately one year) among 53 parents of Autistic pre-schoolers (M = 35.48, SD = 6.36 months. At each timepoint, parents reported lower wellbeing and greater mental health difficulties compared to normative data. There was no significant group-level change over time in parent outcomes. However, individual variability in short-term (~ 5 months) wellbeing and mental health change was predicted by a combination of child- and parent-related factors, while variability in medium-term (~ 10 months) change was predicted by parent factors alone. Parents' description of their child and their relationship predicted change in both wellbeing and mental health. Furthermore, participating in a parent-mediated intervention (available to a subgroup) was a significant predictor of change in wellbeing. Our findings highlight potentially modifiable factors (e.g., learning healthier coping strategies) that may positively impact both short- and medium-term change in parental outcomes.

Examination of the Potential Moderating Role of Psychological Wellbeing in the Relationship Between Depression and Thoughts of Self-Harm in Autistic Adolescents and Adults: A Two-Year Longitudinal Study.

PURPOSE: Autistic people have a significantly increased risk of death by suicide relative to the general population. In non-autistic samples, psychological wellbeing has been shown to moderate the relationship between depression and suicidal thoughts and behavior. Thoughts of self-harm may provide a useful indicator of suicidal risk. In this longitudinal study we examined (a) the potential role for psychological wellbeing to moderate the relationship between depressive symptoms and thoughts of self-harm and (b) the contribution of autistic traits to thoughts of self-harm. METHODS: Participants were 209 autistic adolescents and adults aged 15 to 80 years (Mage = 34.20, SD = 15.38 years). RESULTS: At both baseline and 2-year follow-up, 35% of participants reported recent thoughts of self-harm. Wellbeing was associated with autistic traits (r = - .350 to - 0.404) and depression (r = - .480 to - 0.759). Thoughts of self-harm were positively associated with autistic traits and depression (r = .242 to 0.659), and negatively associated with wellbeing (r = - .287 to - 0.609). Controlling for baseline thoughts of self-harm, depression (ß = 0.254, p = .001) and autistic traits (ß = 0.162, p = .007) significantly predicted thoughts of self-harm at 2-year follow-up. CONCLUSION: Despite a lack of support for the hypothesis that wellbeing would moderate the relationship between depression and thoughts of self-harm, correlational data demonstrated significant associations between wellbeing and both depression and thoughts of self-harm. Future research considering psychological wellbeing as a potential protective factor for self-harm in autistic people is warranted.

Measuring self and informant perspectives of Restricted and Repetitive Behaviours (RRBs): psychometric evaluation of the Repetitive Behaviours Questionnaire-3 (RBQ-3) in adult clinical practice and research settgs.

BACKGROUND: Brief questionnaires that comprehensively capture key restricted and repetitive behaviours (RRBs) across different informants have potential to support autism diagnostic services. We tested the psychometric properties of the 20-item Repetitive Behaviours Questionnaire-3 (RBQ-3), a questionnaire that includes self-report and informant-report versions enabling use across the lifespan. METHOD: In Study 1, adults referred to a specialised adult autism diagnostic service (N = 110) completed the RBQ-3 self-report version, and a relative or long-term friend completed the RBQ-3 informant-report version. Clinicians completed the abbreviated version of the Diagnostic Interview for Social and Communication Disorders (DISCO-Abbreviated) with the same adults as part of the diagnostic process. For half of the assessments, clinicians were blind to the RBQ-3 ratings. We tested internal consistency, cross-informant reliability and convergent validity of the RBQ-3. In Study 2, a follow-up online study with autistic (N = 151) and non-autistic (N = 151) adults, we further tested internal consistency of the RBQ-3 self-report version. We also tested group differences and response patterns in this sample. RESULTS: Study 1 showed good to excellent internal consistency for both self- and informant-report versions of the RBQ-3 (total score, α = 0.90, ω = 0.90, subscales, α = 0.76-0.89, ω = 0.77-0.88). Study 1 also showed cross-informant reliability as the RBQ-3 self-report scores significantly correlated with RBQ-3 informant-report scores for the total score (rs = 0.71) and subscales (rs= 0.69-0.72). Convergent validity was found for both self and informant versions of the RBQ-3, which significantly correlated with DISCO-Abbreviated RRB domain scores (rs = 0.45-0.54). Moreover, the RBQ-3 scores showed significantly weaker association with DISCO -Abbreviated scores for the Social Communication domain, demonstrating divergent validity. Importantly, these patterns of validity were found even when clinicians were blind to RBQ-3 items. In Study 2, for both autistic and non-autistic groups, internal consistency was found for the total score (α = 0.82-0.89, ω = 0.81-0.81) and for subscales (α = 0.68-0.85, ω = 0.69-0.85). A group difference was found between groups. LIMITATIONS: Due to the characteristics and scope of the specialist autism diagnostic service, further testing is needed to include representative samples of age (including children) and intellectual ability, and those with a non-autistic diagnostic outcome. CONCLUSIONS: The RBQ-3 is a questionnaire of RRBs that can be used across the lifespan. The current study tested its psychometric properties with autistic adults without intellectual disability and supported its utility for both clinical diagnostic and research settings.

Characterising Insistence on Sameness and Circumscribed Interests: A Qualitative Study of Parent Perspectives.

Manifestations of insistence on sameness (IS) and circumscribed interests (CI) are complex, with individuals varying considerably, not only in the types of behaviours they express, but also in terms of a behaviour's frequency, intensity, trajectory, adaptive benefits, and impacts. However, current quantitative RRB instruments capture only certain aspects of these behaviours (e.g., mostly frequency or general "severity"). Thus, the current study utilised a semi-structured caregiver interview to provide an in-depth, qualitative characterization of different aspects of IS and CI presentation. Caregivers of 27 autistic children and adolescents displaying IS and/or CI behaviours (3-16 years; 18 males; 9 females) participated in a semi-structured interview. Responses were analysed using thematic framework analysis. Framework analysis identified nine different aspects of IS and CI presentation: (1) intensity, (2) frequency, (3) emergence of behaviour, (4) changes over time, (5) day-to-day fluctuations, (6) purpose/adaptive benefit, (7) experiences of distress, (8) challenges for the individual, their family, and their socialisation, and (9) management strategies and their effectiveness. Autistic children and adolescents were reported to vary greatly on each of these dimensions. Findings demonstrate the complexity of IS and CI presentations and highlight the need for more comprehensive quantitative assessments that independently assess the frequency, intensity, and impact of behaviours. Further, findings reported here emphasize the need for ecologically valid measures that assess the contexts in which these behaviours occur and how their presentations can change within and across days.

A repeated cross-sectional study of daily activities of autistic adults.

It is crucial to arrive at a comprehensive understanding of the types of daily activities autistic adults typically engage in. However, previous research has almost exclusively focused on vocational or education activities. Further, it remains unclear how and whether specific daily activities participation rates change proportionally over time, vary by gender, or compare to nationally representative data. Utilizing eight annual data waves from the Netherlands Autism Register (NAR) this study aims to bridge this gap. Participants were 2449 autistic adults who indicated their participation in 18 daily activities. Results suggest that autistic adults engaged most frequently in vocational activities (e.g., paid employment, study) and participation rates were stable over time. Participation rates in non-vocational activities (e.g., hobbies, homemaking) fluctuated proportionally over time, with reports of no structured daytime activities reducing over time. Labor force participation amongst NAR participants was significantly lower than Dutch population data for the same time periods. Unemployment rates fluctuated, and were significantly higher than population data, but not for all time points. Females compared to males were overrepresented in unpaid daily activities (e.g., study, volunteer, housemaker) and work incapacitation, and underrepresented in paid employment. Employment differences in gender corresponded to national data. These findings characterize more clearly the daily activities of autistic adults, and highlights areas where support may have greater impact (e.g., females in employment).

The Neurodiversity Attitudes Questionnaire: Development and Initial Validation.

LAY ABSTRACT: Neurodiversity refers to the idea that brain differences (including disabilities) are valuable and should be accepted. Attitudes toward neurodiversity can have real-life impacts on the lives of neurodivergent people (those whose brains do not fit society's "standard"). These impacts can include effects on daily interactions, as well as how professionals such as teachers and doctors deliver services to neurodivergent people. In order to identify negative attitudes toward neurodiversity and potentially improve them, we first need to measure these attitudes. This article describes the development of the Neurodiversity Attitudes Questionnaire (NDAQ). NDAQ development included revision of questionnaire items based on feedback from experts and neurodivergent people, systematically evaluating the way participants responded to questionnaire items, and analysis of how the NDAQ items are grouped into different factors. A preliminary analysis with 351 individuals mostly living in the United States who were currently working or planning to work in a helping profession (e.g. doctors, teachers, therapists, and so on) indicates that the NDAQ measures attitudes toward neurodiversity, is well understood by participants, and fits a five-factor structure. While the NDAQ represents the first instrument designed to specifically assess attitudes toward the broad idea of neurodiversity, further work is still needed.

Assessing pragmatic language difficulties using the Revised Children's Communication Checklist-2. Exploratory structural equation modeling and associations with restricted and repetitive behaviors.

In this paper, we investigated the psychometric properties of the Child Communication Checklist-Revised (CCC-R) for the first time with an English-speaking sample. We used a confirmatory application of exploratory structural equation modeling (ESEM) to re-evaluate the CCC-R's psychometric properties. We found strong support for its use as an assessment for pragmatic and structural language. Our second main aim was to explore associations between pragmatic and structural language and restricted and repetitive behaviors (RRBs), two hallmark characteristics of autism. We used the CCC-R and Repetitive Behavior Questionnaire (RBQ-2) to investigate these associations in a diverse non-clinical sample of children, taking a transdiagnostic approach. We intentionally excluded autism and other neurodevelopmental diagnoses to test, (1) the CCC-R in a broad sample and (2) the association between pragmatic language and RRB in children not already selected for that association. The sample comprised two groups of children, one was community sampled (n = 123) and the other (n = 143) included children with non-specific behavioral, emotional and/or cognitive difficulties referred to an assessment unit by schools. We found clear associations between pragmatic language difficulties and RRBs in both groups. Regression analysis showed that pragmatic language was the only significant contributor to RRBs even after Grammatical-Semantic score, age, sex, and socioeconomic status were controlled. The pattern was the same for both recruitment groups. However, the effects were stronger for the school-referred group which also had more pragmatic difficulties, grammatical-semantic difficulties and RRBs. A robust link between pragmatic language and RRBs, established in autism, has continuity across the broader non-clinical population.

Employment profiles of autistic people: An 8-year longitudinal study.

LAY ABSTRACT: Autistic adults experience difficulties finding and keeping employment. However, research investigating reasons that might explain this difficulty produce mixed results. We gave a survey to 2449 autistic adults and used a statistic method to group them based on their employment status over 8 years. We identified four employment groups that best captured the experiences of autistic adults; this included a group that experienced stable unemployment, a group that experienced stable employment, a group that had high employment that reduced over time, and a group whose employment increased over the 8 years. Further analysis showed that those with fewer autistic traits, younger age, male gender, higher education, later diagnosis age and no co-occurring conditions were more likely to have stable employment. People whose employment changed over time were more likely to have a higher level of education than the stable unemployment group, and those in the increasing employment group were younger age and had no co-occurring conditions. These findings help us better understand that not all autistic adults' experiences of employment are the same, which helps focus where employment programmes and support may be most needed, for example, people who identify as women or have a co-occurring condition.

Precision microbial intervention improves social behavior but not autism severity: A pilot double-blind randomized placebo-controlled trial.

Autism spectrum disorder (ASD) is characterized by the presence of restricted/repetitive behaviors and social communication deficits. Because effective treatments for ASD remain elusive, novel therapeutic strategies are necessary. Preclinical studies show that L. reuteri selectively reversed social deficits in several models for ASD. Here, in a double-blind, randomized, placebo-controlled trial, we tested the effect of L. reuteri (a product containing a combination of strains ATCC-PTA-6475 and DSM-17938) in children with ASD. The treatment does not alter overall autism severity, restricted/repetitive behaviors, the microbiome composition, or the immune profile. However, L. reuteri combination yields significant improvements in social functioning that generalized across different measures. Interestingly, ATCC-PTA-6475, but not the parental strain of DSM-17938, reverses the social deficits in a preclinical mouse model for ASD. Collectively, our findings show that L. reuteri enhances social behavior in children with ASD, thereby warranting larger trials in which strain-specific effects should also be investigated.

Characterizing subdomains of insistence on sameness in autistic youth.

Insistence on sameness (IS) encompasses a range of behavioral patterns, including resistance to change, routines, and ritualized behaviors, that can be present across social and non-social contexts. Given the breadth of behaviors encompassed by IS, it is important to determine whether this domain is best conceptualized and measured as uni- or a multi-dimensional construct. Therefore, the current study aimed to characterize the structure of IS and explore potentially distinct of patterns of associations between identified IS factors and relevant correlates, including age, sex, IQ, anxiety, social abilities, emotional and behavioral dysregulation, and sensory hypersensitivity. Exploratory graph analysis was conducted using the dimensional assessment of restricted and repetitive behaviors to examine the structure of IS in a sample 1892 autistic youth (Mage = 10.82, SDage = 4.14; range: 3-18 years; 420 females) recruited from the Simons Foundation Powering Autism Research for Knowledge cohort. Three distinct IS subdomains labeled as IS-Ritualistic/sameness, IS-Routines, and IS-Others (referring to IS behaviors during interactions with others) were identified. Generalized additive models demonstrated that each of the IS subdomains showed a unique pattern of association with key variables. More specifically, while sensory hypersensitivity was significantly associated with IS-Ritualistic/sameness and IS-Routines, it was not associated with IS-Others. Further, while emotional dysregulation was a unique predictor of IS-Ritualistic/sameness (but not IS-Routines or IS-Others), social interaction abilities were a unique predictor of IS-Routines (but not IS-Ritualistic/sameness or IS-Others). Current findings provide preliminary evidence that the IS may encompass several distinct subdomains.

Categorical versus dimensional structure of autism spectrum disorder: A multi-method investigation.

Background: A key question for any psychopathological diagnosis is whether the condition is continuous or discontinuous with typical variation. The primary objective of this study was to use a multi-method approach to examine the broad latent categorical versus dimensional structure of autism spectrum disorder (ASD). Method: Data were aggregated across seven independent samples of participants with ASD, other neurodevelopmental disorders (NDD), and non-ASD/NDD controls (aggregate Ns = 512-16,755; ages 1.5-22). Scores from four distinct phenotype measures formed composite "indicators" of the latent ASD construct. The primary indicator set included eye gaze metrics from seven distinct social stimulus paradigms. Logistic regressions were used to combine gaze metrics within/across paradigms, and derived predicted probabilities served as indicator values. Secondary indicator sets were constructed from clinical observation and parent-report measures of ASD symptoms. Indicator sets were submitted to taxometric- and latent class analyses. Results: Across all indicator sets and analytic methods, there was strong support for categorical structure corresponding closely to ASD diagnosis. Consistent with notions of substantial phenotypic heterogeneity, the ASD category had a wide range of symptom severity. Despite the examination of a large sample with a wide range of IQs in both genders, males and children with lower IQ were over-represented in the ASD category, similar to observations in diagnosed cases. Conclusions: Our findings provide strong support for categorical structure corresponding closely to ASD diagnosis. The present results bolster the use of well-diagnosed and representative ASD groups within etiologic and clinical research, motivating the ongoing search for major drivers of the ASD phenotype. Despite the categorical structure of ASD, quantitative symptom measurements appear more useful for examining relationships with other factors.

Development of webcam-collected and artificial-intelligence-derived social and cognitive performance measures for neurodevelopmental genetic syndromes.

This study focused on the development and initial psychometric evaluation of a set of online, webcam-collected, and artificial intelligence-derived patient performance measures for neurodevelopmental genetic syndromes (NDGS). Initial testing and qualitative input was used to develop four stimulus paradigms capturing social and cognitive processes, including social attention, receptive vocabulary, processing speed, and single-word reading. The paradigms were administered to a sample of 375 participants, including 163 with NDGS, 56 with idiopathic neurodevelopmental disability (NDD), and 156 neurotypical controls. Twelve measures were created from the four stimulus paradigms. Valid completion rates varied from 87 to 100% across measures, with lower but adequate completion rates in participants with intellectual disability. Adequate to excellent internal consistency reliability (α = 0.67 to 0.95) was observed across measures. Test-retest reproducibility at 1-month follow-up and stability at 4-month follow-up was fair to good (r = 0.40-0.73) for 8 of the 12 measures. All gaze-based measures showed evidence of convergent and discriminant validity with parent-report measures of other cognitive and behavioral constructs. Comparisons across NDGS groups revealed distinct patterns of social and cognitive functioning, including people with PTEN mutations showing a less impaired overall pattern and people with SYNGAP1 mutations showing more attentional, processing speed, and social processing difficulties relative to people with NFIX mutations. Webcam-collected performance measures appear to be a reliable and potentially useful method for objective characterization and monitoring of social and cognitive processes in NDGS and idiopathic NDD. Additional validation work, including more detailed convergent and discriminant validity analyses and examination of sensitivity to change, is needed to replicate and extend these observations.

Arriving at the empirically based conceptualization of restricted and repetitive behaviors: A systematic review and meta-analytic examination of factor analyses.

An empirically based understanding of the factor structure of the restricted and repetitive behaviors (RRB) domain is a prerequisite for interpreting studies attempting to understand the correlates and mechanisms underpinning RRB and for measurement development. Therefore, this study aimed to conduct a systematic review and meta-analysis of RRB factor analytic studies. Sets of meta-analyses were performed to examine (a) the factor structure of individual RRB instruments, (b) associations between RRB subdomains across instruments, and (c) the association between RRB factors and other variables. Searches for peer-reviewed articles evaluating the factor structure of the RRB domain were performed in PsycINFO (Ovid), Medline (Ovid), and Embase (Ovid). No age, measurement, or informant-type limits were imposed. Quality and risk of bias for individual studies were assessed using relevant COSMIN sections. Among the 53 studies retained for review, 41 examined RRB factor structures among individuals with autism spectrum disorder (ASD) and 12 among non-ASD samples. Meta-analysis of factor correlations provided evidence that the RRB domain encompasses the following eight specific factors: repetitive motor behaviors, insistence on sameness, restricted interests, unusual interests, sensory sensitivity, and repetitive, stereotyped language. Although interrelated, RRB factors were distinct, showing a unique pattern of associations with demographic, cognitive, and clinical correlates. Meta-analyses of the associations between RRB factors and specific correlates, specifically adaptive functioning and communication impairments, should be considered preliminary due to the limited number of studies. Despite limitations, this review provides important insights into the factor structure of the RRB domain and highlights critical conceptual, measurement, and methodological limitations of the current research that will need to be addressed in order to improve our understanding of RRB.

Reliability of the Commonly Used and Newly-Developed Autism Measures.

PURPOSE: The aim of the present study was to compare scale and conditional reliability derived from item response theory analyses among the most commonly used, as well as several newly developed, observation, interview, and parent-report autism instruments. METHODS: When available, data sets were combined to facilitate large sample evaluation. Scale reliability (internal consistency, average corrected item-total correlations, and model reliability) and conditional reliability estimates were computed for total scores and for measure subscales. RESULTS: Generally good to excellent scale reliability was observed for total scores for all measures, scale reliability was weaker for RRB subscales of the ADOS and ADI-R, reflecting the relatively small number of items for these measures. For diagnostic measures, conditional reliability tended to be very good (> 0.80) in the regions of the latent trait where ASD and non-ASD developmental disability cases would be differentiated. For parent-report scales, conditional reliability of total scores tended to be excellent (> 0.90) across very wide ranges of autism symptom levels, with a few notable exceptions. CONCLUSIONS: These findings support the use of all of the clinical observation, interview, and parent-report autism symptom measures examined, but also suggest specific limitations that warrant consideration when choosing measures for specific clinical or research applications.