Sleep Problems and Trajectories of Restricted and Repetitive Behaviors in Children with Neurodevelopmental Disabilities.

Sleep problems are prevalent in children with neurodevelopmental disabilities and are associated with the expression of restricted and repetitive behaviors (RRBs). Children (n = 57) with autism spectrum disorder (ASD, n = 38) or developmental delay (DD, n = 19) participated in multiple assessments of intellectual ability, ASD symptoms, and RRBs (3 timepoints for ASD, 2 for DD). Sleep problems assessed at age 4 via parent report were associated with trajectories of higher-order RRBs (sameness/ritualistic/compulsive behaviors) from age 2-6 in the ASD group, and from age 2-4 in the DD group, even after controlling for intellectual ability, social-affective symptoms, and anxiety. Trajectories of stereotyped/restricted behaviors were unrelated to sleep problems. Sleep problems were associated with trajectories of higher-order (but not lower-order) RRBs in a transdiagnostic sample.

Restricted and Repetitive Behaviors in Males and Females with Fragile X Syndrome: Developmental Trajectories in Toddlers Through Young Adults.

There is limited research on the trajectory of restricted and repetitive behaviors (RRBs) in fragile X syndrome (FXS), with previous studies only examining males and/or examining RRBs as a unitary construct rather than delineating subtypes of RRBs. Thus, we described the trajectory of five subtypes of RRBs in 153 males and females with FXS (aged 1-18 years) with repeated measurement over time (445 total assessments). Multilevel modeling was used to test age-related differences in RRB subtypes between males and females with FXS, controlling for nonverbal IQ. Results showed that lower-order Sensory-Motor behaviors decreased over time for both males and females, while there was no significant change in the higher-order RRBs. The trajectory between males and females differed for Self-Injury.

The SOFIA Study: Negative Multi-center Study of Low Dose Fluoxetine on Repetitive Behaviors in Children and Adolescents with Autistic Disorder.

Fluoxetine is a selective serotonin reuptake inhibitor (SSRI) that reduces obsessive-compulsive symptoms. There is limited evidence supporting its efficacy for repetitive behaviors (RRBs) in autistic spectrum disorder (ASD). We conducted a randomized controlled trial (RCT) of fluoxetine in 158 individuals with ASD (5-17 years). Following 14 treatment weeks (mean dose 11.8 mg/day), no significant differences were noted on the Children's Yale-Brown Obsessive Compulsive Scale; the proportion of responders was similar (fluoxetine: 36%; placebo: 41%). There were similar rates of AEs (e.g., insomnia, diarrhea, vomiting); high rates of activation were reported in both groups (fluoxetine: 42%; placebo: 45%). Overly cautious dosing/duration may have prevented attainment of a therapeutic level. Results are consistent with other SSRI RCTs treating RRBs in ASD.Trial Registration: clinicaltrials.gov Identifier: NCT00515320.

A comparison of differential reinforcement procedures for treating automatically reinforced behavior.

Children diagnosed with autism spectrum disorder (ASD) often exhibit stereotypy, which can be socially stigmatizing, interfere with daily living skills, and affect skill acquisition. We compared differential reinforcement of alternative behavior (DRA) and differential reinforcement of other behavior (DRO) when neither procedure included response blocking or interruption for (a) reducing stereotypy, (b) increasing task engagement, and (c) increasing task completion. DRA contingencies yielded superior outcomes across each measure when evaluated with 3 individuals with autism spectrum disorder.

Understanding the Influence of Social and Motor Context on the Co-occurring Frequency of Restricted and Repetitive Behaviors in Autism.

The social and motor context in which restricted and repetitive behaviors (RRBs) occur in autism and their relationship to social traits are not well-understood. Participants with and without autism completed tasks that varied in social and motor engagement and RRB frequency was measured. Motor and verbal RRBs were most common, RRBs varied based on motor and social context for participants with autism, and social engagement was associated with lower motor and verbal RRBs. Significant correlations between RRBs and autism severity, social synchrony, and nonverbal mental age were also found. This research confirms the importance of context for understanding RRBs during on-going tasks and raises questions about whether the factors that elicit vocal and motor RRBs are unique for individual children.

Prevalence and Risk-Markers of Self-Harm in Autistic Children and Adults.

Self-harm is purportedly common in autistic individuals, but under-researched, particularly in younger samples and those without intellectual disability. This study aimed to describe prevalence, profile and correlates of self-harm in autistic individuals without impairments in adaptive functioning. Parents of autistic participants (n = 83) completed questionnaires regarding the presence/topography of self-harm, demographic characteristics, autism severity, age of diagnosis, affect, activity levels and repetitive behaviour. 24.10% of participants engaged in self-harm. Self-harm was associated with significantly higher levels of impulsivity, over-activity, negative affect, compulsive behaviour and insistence on sameness. Low mood and overactivity/impulsivity predicted the presence of self-harm, with the model correctly classifying 82.9% of cases. Findings highlight a role for impaired behavioural inhibition and low mood in the aetiological mechanisms underpinning self-harm in autism.

Interrelationship Between Cognitive Control, Anxiety, and Restricted and Repetitive Behaviors in Children with 22q11.2 Deletion Syndrome.

Restricted and repetitive behaviors (RRB) are common in individuals with 22q11.2 microdeletion syndrome (22q11.2DS), yet the underlying mechanisms of these behaviors remain poorly characterized. In the present pilot investigation, we aimed to further our understanding of RRB in 22q11.2DS by exploring their relationship with cognitive control and anxiety as well as with sex, chronological age, and full-scale IQ. Parents of 38 children with 22q11.2DS (17 females; Mage = 11.15 years, SD = 2.46) completed the Social Communication Questionnaire as a measure of RRB and social and communication (SC) problems and the Behavioral Assessment System for Children-2 as a measure of anxiety and cognitive control. Higher RRB scores were significantly associated with higher anxiety levels (r = 0.44, P = 0.006), more impairments in cognitive control (r = 0.56, P < 0.001), and higher SC scores (r = 0.43, P = 0.011). In the first step of the hierarchical regression model, anxiety accounted for 24.5% of variance (F = 10.05, P = 0.003); cognitive control accounted for an additional 18.1% of variance (Fchange = 11.15, P < 0.001) in the second step; SC score accounted for only 0.8% of additional variance in the third step (Fchange = 0.40, P = 0.53). The final model explained 43.4% of variance (F = 7.42, P = 0.001), with cognitive control as a unique independent predictor of RRB score (t = 2.52, P = 0.01). The current study provides the first exploration of the cognitive control-anxiety-RRB link in individuals with 22q11.2DS and points to cognitive control as a potentially viable target for treatments aimed at reducing RRB. Autism Res 2019, 12: 1737-1744. © 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: People with 22q11.2 deletion syndrome show high levels of repetitive behaviors, however, the previous research has not explored why people with this syndrome exhibit high rates of repetitive behaviors. Understanding the reasons for the high levels of repetitive behaviors is important given that these behaviors can be highly impairing. Our study found that repetitive behaviors were associated with impaired ability to self-regulate and high levels of anxiety. These findings need to be further replicated; however, they are important as they suggest potentially promising ways of reducing these behaviors.

Clinical Profile of Autism Spectrum Disorder in a Pediatric Population from Northern Mexico.

Autism spectrum disorder (ASD) is a neurodevelopmental condition classified based on needs of support, in order to address impairments in the areas of social communication and restricted and repetitive behavior. The aim of this work is to describe the main clinical features of the ASD severity levels in a group of Mexican pediatric patients. The results show firstly that this condition was more frequent in males than females. Secondly, an inverse relationship was found between the intellectual coefficient and the level of severity of the disorder. Thirdly, deficits in social reciprocity and communication were more evident in Level 3, than in Levels 1 and 2, while the difference was less evident in restricted and repetitive patterns of behavior.

Psychometric analysis of the repetitive behavior scale-revised using confirmatory factor analysis in children with autism.

Research examining restricted and repetitive patterns of behavior or interests (RRB) in autism spectrum disorder (ASD) has increased our understanding of its contribution to diagnosis and its role in development. Advances in our knowledge of RRB are hindered by the inconsistencies in how RRB is measured. The present study examined the factor structure of the Repetitive Behavior Scale-Revised (RBS-R) in a sample of 350 children with ASD ages 2-9. Confirmatory factor analysis designed for items with categorical response types was implemented to examine six proposed structural models. The five-factor model demonstrated the most parsimonious fit based on common overall fit indices that was further supported by examination of local model fit indicators, though, the four- and six-factor models evidenced adequate-to-good fit as well. Examination of RRB factor score approaches indicated only minor differences between summed item subscale scores and extracted factor scores with regard to associations with diagnostic measures. All RRB subtypes demonstrated significant associations with cognitive functioning and adaptive behavior. Implications for future research validating the RBS-R as a more extensive clinical measure of RRB in ASD are discussed. Autism Res 2019, 12: 1399-1410. © 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: Repetitive behaviors are one of the two main symptoms of autism spectrum disorder (ASD). To better understand the role of repetitive behaviors, we must establish effective ways of measuring them. This study assessed the measurement qualities of the Repetitive Behavior Scale-Revised (RBS-R) in a sample of 350 children with ASD ages 2-9. We found that the RBS-R measures multiple types of repetitive behaviors and that these behaviors are related to thinking ability and independence.

Short report: Relationships between sensory processing, repetitive behaviors, anxiety, and intolerance of uncertainty in autism spectrum disorder and Williams syndrome.

Autism spectrum disorder (ASD) and Williams syndrome (WS) share psychopathology relating to sensory processing and repetitive behaviors. The relationships between the sensory features and repetitive behaviors in both disorders, and the mechanisms underlying these relationships are not well understood. The aim of this study was to examine the relationships between sensory processing, repetitive behaviors, anxiety, and intolerance of uncertainty in children with ASD and those with WS to better understand the complexity of psychopathology in these disorders. Parents of 19 children with ASD and 16 children with WS, aged between 4 and 9 years, were asked to complete questionnaires assessing their children's sensory experiences, anxiety symptoms, severity and frequency of repetitive behaviors, and level of intolerance of uncertainty. Serial mediation analysis was performed. Direct significant relationships between sensory features and repetitive behaviors were found only for the ASD group. The relationship between sensory processing difficulties and repetitive behaviors was mediated via intolerance of uncertainty in WS. The findings support the value of considering the complexity of the mechanisms underlying the relationship between sensory processing and repetitive behaviors across neurodevelopmental disorders and the mechanisms underlying these aspects of psychopathology in these groups. Understanding these relationships will shed light on some of the most challenging and intractable characteristics of both conditions and inform suitable interventions to improve quality of life for individuals with either ASD or WS. Autism Research 2019, 12: 759-765. © 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: In autism spectrum disorder (ASD) and Williams syndrome (WS) difficulties processing the sensory aspects of the environment, repetitive behaviors and high levels of anxiety co-occur, but the relationships between these features are not well understood. This study found that sensory difficulties were directly associated with repetitive behaviors in children with ASD, but not WS, and in WS this relationship was mediated by intolerance of uncertainty. The findings support the value of considering the complexity of the mechanisms underlying the relationship between sensory processing and repetitive behaviors across neurodevelopmental disorders.

'People should be allowed to do what they like': Autistic adults' views and experiences of stimming.

'Stereotyped or repetitive motor movements' are characterised as core features in the diagnosis of autism, yet many autistic adults (and the neurodiversity movement) have reclaimed them as 'stimming'. Supported by a growing body of scientific research, autistic adults argue that these behaviours may serve as useful coping mechanisms, yet little research has examined stimming from the perspective of autistic adults. Through interviews and focus groups, we asked 32 autistic adults to share their perceptions and experiences of stimming, including the reasons they stim, any value doing so may hold for them and their perceptions of others' reactions to stimming. Using thematic analysis, we identified two themes: stimming as (1) a self-regulatory mechanism and (2) lacking in social acceptance, but can become accepted through understanding. Autistic adults highlighted the importance of stimming as an adaptive mechanism that helps them to soothe or communicate intense emotions or thoughts and thus objected to treatment that aims to eliminate the behaviour.

An analysis of stereotypical motor movements and cardiovascular coupling in individuals on the autism spectrum.

One of the core diagnostic features of autism spectrum disorder (ASD) is engagement in stereotypical motor movements, although the etiology of this repetitive behavior is unknown. Since the 1960s, it has been hypothesized that stereotypical motor movements serve a homeostatic regulation function, and thereby a putative coupling mechanism to cardiovascular arousal. However, to date, surprisingly few reports explicitly assess cardio-somatic coupling and stereotypical motor movements. The present exploratory study investigates coupling of stereotypical body rocking and hand flapping to heart rate and heart rate variability (HRV) in a convenience sample (n = 10) of children and young adults with moderate to profound ASD. Motor movements were recorded via video and three-axis accelerometry, and simultaneous electrocardiographic signals were obtained to determine cardiovascular indices at or around the onset of naturalistically occurring stereotypy. Analysis of the heart rate revealed both repetitive body rocking and hand flapping in particular were found to associate with a strikingly similar cardiovascular pattern of acceleration and deceleration unrelated to physical demands associated with the movements themselves. Furthermore, neither type of stereotypical movement provoked directional change in heart rate variability. The implications of these results and opportunities for future research are discussed.

Maternal characteristics and behavioural/emotional problems in preschoolers: how they relate to sleep rhythmic movements at sleep onset.

Sleep rhythmic movements have been speculated to be a form of self-soothing. While this sleep-related movement has been associated with lower socioeconomic status, psychopathologies and maternal characteristics, prospective studies with sizeable sample and objective measurements are lacking. The objectives were: (a) to identify maternal characteristics predicting sleep rhythmic movements in children; and (b) to document behavioural/emotional problems in preschoolers with sleep rhythmic movements. Participants were mother-child dyads (N = 529) from the Adversity: Maternal Adversity, Vulnerability and Neurodevelopment cohort. Questionnaires evaluating socioeconomic status (prenatal), maternal depressive symptoms (prenatal, 48 months), sleep rhythmic movements (12, 18, 24, 36, 48 months), maternal anxiety trait (24 months) and children's behavioural/emotional problems (48 months) were used. Maternal sensitivity (accuracy and appropriateness of mother's responses to her baby's needs) was assessed objectively with a filmed mother-infant interaction (6 months). Generalized estimating equation was used to investigate associations between sleep rhythmic movements and maternal characteristics (depression, anxiety and sensitivity). Linear regressions were used to assess associations between sleep rhythmic movements and behavioural/emotional problems in children. Lower maternal sensitivity, higher maternal depressive symptoms and lower socioeconomic status predicted sleep rhythmic movements in children (p < 0.05). To our knowledge, this is the first study showing that sleep rhythmic movements are associated with lower maternal sensitivity, measured objectively. This study also builds on previous reports, by documenting an association between sleep rhythmic movements and behavioural/emotional problems even in preschoolers. The presence of psychosocial factors in sleep rhythmic movements aetiology should be considered in treatment.

Brief Report: Sex Differences in ASD Diagnosis-A Brief Report on Restricted Interests and Repetitive Behaviors.

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.

Large Forehead Mass Resulting From Chronic Head Banging.

Head banging is a commonly observed movement disorder which is typically self-limited. Complications secondary to this behavior are rare. In this report, the authors present the case of a 15-year-old patient who was treated for a forehead mass which developed secondary to chronic head banging. Surgical excision was performed for treatment of the lesion. Results from surgical pathology were notable for fibrosis consistent with history of chronic head banging. Preoperative magnetic resonance imaging and physical examination were also consistent with this diagnosis. This is a rare clinical entity that should be considered in patients presenting with a forehead mass and a history of head banging.

Automated Detection of Repetitive Motor Behaviors as an Outcome Measurement in Intellectual and Developmental Disabilities.

Repetitive sensory motor behaviors are a direct target for clinical treatment and a potential treatment endpoint for individuals with intellectual or developmental disabilities. By removing the burden associated with video annotation or direct observation, automated detection of stereotypy would allow for longer term monitoring in ecologic settings. We report automated detection of common stereotypical motor movements using commercially available accelerometers affixed to the body and a generalizable detection algorithm. The method achieved a sensitivity of 80% for body rocking and 93% for hand flapping without individualized algorithm training or foreknowledge of subject's specific movements. This approach is well-suited for implementation in a continuous monitoring system outside of a clinical setting.

Brief Report: Repetitive Behaviour Profiles in Williams syndrome: Cross Syndrome Comparisons with Prader-Willi and Down syndromes.

This study describes the profile of repetitive behaviour in individuals with Williams syndrome, utilising cross-syndrome comparisons with people with Prader-Willi and Down syndromes. The Repetitive Behaviour Questionnaire was administered to caregivers of adults with Williams (n = 96), Prader-Willi (n = 103) and Down (n = 78) syndromes. There were few group differences, although participants with Williams syndrome were more likely to show body stereotypies. Individuals with Williams syndrome also showed more hoarding and less tidying behaviours than those with Down syndrome. IQ and adaptive ability were negatively associated with repetitive questioning in people with Williams syndrome. The profile of repetitive behaviour amongst individuals with Williams syndrome was similar to the comparison syndromes. The cognitive mechanisms underlying these behaviours in genetic syndromes warrant further investigation.

Prenatal lipopolysaccharide induces hypothalamic dopaminergic hypoactivity and autistic-like behaviors: Repetitive self-grooming and stereotypies.

Previous investigations by our group have shown that prenatal exposure to lipopolysaccharide (LPS), which mimics infection by gram-negative bacteria, induces social, cognitive, and communication deficits. For a complete screening of autistic-like behaviors, the objective of this study was to evaluate if our rat model also induces restricted and repetitive stereotyped behaviors. Thus, we studied the self-grooming microstructure. We also studied the neurochemistry of hypothalamus and frontal cortex, which are brain areas related to autism to better understand central mechanisms involved in our model. Prenatal LPS exposure on gestational day 9.5 increased the head washing episodes (frequency and time), as well as the total self-grooming. However, body grooming, paw/leg licking, tail/genital grooming, and circling behavior/tail chasing did not vary significantly among the groups. Moreover, prenatal LPS induced dopaminergic hypoactivity (HVA metabolite and turnover) in the hypothalamus. Therefore, our rat model induced restricted and repetitive stereotyped behaviors and the other main symptoms of autism experimentally studied in rodent models and also found in patients. The hypothalamic dopaminergic impairments seem to be associated with the autistic-like behaviors.

Relationship between repetitive behaviour and fear across normative development, autism spectrum disorder, and down syndrome.

The present study had two aims: first to compare levels of restricted and repetitive behaviours (RRB) across two groups of typically developing (TD) children, and two disorders: Autism Spectrum Disorder (ASD) and Down syndrome (DS), and second to explore the relationship between fear and repetitive behaviours in these four groups. Parents of 41 offspring with ASD (Mage = 123.39 months, SDage = 27.67), 38 offspring with DS (Mage = 125.37 months, SDage = 45.71), 45 typically developing children matched to the mental age (MA) of the DS group (TD MA; Mage = 51.13 months, SDage = 22.1), and 42 chronological age (TD CA; Mage = 117.93 months, SDage = 22.91) matched TD children, completed measures of RRB and fear. ANOVAs revealed differences across the four groups on the RRB subscale scores: "Just Right" F(3,162) = 16.62, P < 0.001; Rigid Routines F(3,162) = 52.76, P < 0.001; Sensory behaviours F(3,162) = 23.26, P < 0.001. Post-hoc comparisons revealed that children with ASD had the highest RRB levels followed by DS, TD MA, and TD CA children. In children with ASD, higher levels of fear were related to higher RRB levels. Similar, albeit less strong, patterns of associations was found among DS and TD MA children but not in older TD CA children. This study provided evidence of a fear-RRB association in children with ASD, DS, and two groups of TD children. Autism Res 2017, 10: 502-507. © 2016 International Society for Autism Research, Wiley Periodicals, Inc.

Parent-Reported Repetitive Behavior in Toddlers on the Autism Spectrum.

Toddlers with autism spectrum disorder (ASD) were assessed on the Repetitive Behavior Scale-Revised (RBS-R), which we found to have acceptable internal consistency. Stereotypical subscale scores showed a negligible association with cognitive level, but correlated more strongly with adaptive and social indicators. Relative to earlier reported RBS-R scores for older age groups, toddlers' scores trended toward higher stereotyped behavior and lower ritualistic/sameness behavior. Our findings on associations with developmental indicators align with those of researchers who used more resource-intensive repetitive behavior measures. The convergence of these findings with those derived from other measurement methods suggests that the RBS-R, a cost effective parent-report measure, is a viable means of assessing repetitive behavior in toddlers with autism.