Implementing a Uniform Outcome Measurement Approach for Early Interventions of Autism Spectrum Disorders.

OBJECTIVE: Naturalistic developmental behavioral interventions (NDBI) for children with autism spectrum disorder (ASD) show evidence for effectiveness for specific social communication targets such as joint attention or engagement. However, combining evidence from different studies and comparing intervention effects across those studies have not been feasible due to lack of a standardized outcome measure of broader social communication skills that can be applied uniformly across trials. This investigation examines the usefulness of the Brief Observation of Social Communication Change (BOSCC) as a common outcome measure of general social communication skills based on secondary analyses of data obtained from previously conducted randomized control trials (RCTs) of three intervention models, the Early Social Intervention (ESI), Early Start Denver Model (ESDM) and Joint Attention Symbolic Play Engagement and Regulation (JASPER). METHOD: The subset of datasets from the three RCTs was created to examine differences in the BOSCC scores between intervention and control groups over the course of the interventions. RESULTS: Based on 582 videos from 207 caregiver-child dyads, the BOSCC noted significant differences between intervention vs. control groups in broad social communication skills within two of the three intervention models which were longer in duration and focused on a broad range of developmental skills. CONCLUSION: The BOSCC offers the potential to take a uniform measurement approach across different intervention models to capture the effect of intervention on general social communication skills but may not pick up the effects of some brief interventions targeting proximal outcomes.

Examining variability in Naturalistic Developmental Behavioral Intervention strategy use in caregivers of children with autism spectrum disorders.

BACKGROUND: Naturalistic Developmental Behavioral Interventions (NDBIs) for young children with autism spectrum disorder commonly involve caregiver-mediated approaches. However, to date, there is limited research on how caregivers' skills change, and, in turn, impact child outcomes. METHODS: We evaluated the NDBI strategy use of 191 caregivers prior to participation in NDBIs (or control groups) across multiple randomized controlled trials, using the Measure of NDBI Strategy Implementation, Caregiver Change (MONSI-CC). Clustering analyses were used to examine caregiver variability in NDBI strategy use at intervention entry. Generalized Linear Mixed Models were used to examine changes in caregiver strategy use over the course of intervention and its impact on changes in children's social communication. RESULTS: Using clustering analysis, we found that caregivers' baseline skills fit four profiles: limited, emerging, variable, and consistent/high, with few demographic factors distinguishing these groups. Caregivers starting with limited or emerging skills improved in their strategy use with intervention. Caregivers starting with more skills (consistent/high or variable) maintained higher skills over intervention. Children of caregivers in these groups who received target NDBIs improved in their social communication skills. CONCLUSIONS: Results suggested that caregiver skills improve through participation in NDBIs and may directly contribute to their children's outcomes, although more research on mediating factors is needed. Individualized approaches for caregivers and their children starting with differing skill profiles at intervention entry may be warranted.

Anxiety in Turner syndrome: Engaging community to address barriers and facilitators to diagnosis and care.

Turner syndrome (TS), caused by complete or partial loss of the second sex chromosome, is associated with complex medical manifestations. The TS community identifies anxiety as a major contributor to reduced quality of life. The study aimed to improve understanding of anxiety symptomatology, diagnosis, and care in individuals with TS. A mixed methods design integrated community engagement, including community leaders as co-investigators and a community advisory board, an online survey (N = 135), and in-depth interviews (N = 10). The majority of respondents reported that anxiety symptoms occur two or more days per week, with self-advocates reporting more frequent symptoms than caregivers (p = 0.03). Self-advocates reported feeling anxious more often at school/work; both rater groups reported anxiety-related behaviors were most likely to be expressed at home. Insomnia was the most common symptom of anxiety endorsed across age and rater groups (>70%). Anxiety symptoms and triggers changed with age and often were undiagnosed or untreated during childhood. Therapy and medication were reported as helpful by most respondents who had tried these strategies. Qualitative themes included: 'Triggers for anxiety are related to TS', 'Anxiety impacts the whole family', and 'Opportunities for early identification and intervention'.

Implementation of Video Feedback Within a Community Based Naturalistic Developmental Behavioral Intervention Program for Toddlers With ASD: Pilot Study.

Video feedback (VF) is an intervention delivery technique that complements naturalistic developmental behavioral interventions (NDBI) and parent-mediated interventions (PMI) by using caregiver-child interaction videos reviewed with a clinician to facilitate behavioral change in caregivers. Although VF has been implemented in PMI with young children with ASD, examinations of feasibility and acceptability, as well as the potential effectiveness of VF in community settings, have been limited. In this pilot randomized control trial (NCT03397719; https://clinicaltrials.gov/ct2/show/NCT03397719), families were randomized into a state-funded Early Intervention (EI) NDBI program or the NDBI program augmented with VF. Results demonstrated high levels of implementation and acceptability of VF augmenting the community-based EI program in caregivers and clinicians. Both groups showed significant improvements after 6 months in social communication symptoms and some areas of developmental and adaptive skills. Clinical Trial Registration:https://clinicaltrials.gov/ct2/show/NCT03397719, identifier: NCT03397719.

Adaptive Skills in FXS: A Review of the Literature and Evaluation of the PEDI-Computer Adaptive Test (PEDI-CAT) to Measure Adaptive Skills.

As adaptive skills (AS) are dynamic and may indicate the success of an intervention, they are a common domain measured in clinical trials. Typical interview tools for measuring AS are time-consuming, and questionnaire measures often lead to inconsistent information. The present study was designed to evaluate the feasibility, validity and test-retest performance of the Pediatric Evaluation of Disability Inventory Computer Adaptive Test (PEDI-CAT) in Fragile X syndrome (FXS). The PEDI-CAT is administered via tablet and uses the item response theory to efficiently determine the items administered. The PEDI-CAT was administered to 42 individuals with FXS (27 males; 15 females) aged 1.6-50.9 years (M = 14.9; SD = 11.2), followed by the Vineland-3 (VABS-3) interview for comparison. Administration was efficient (M = 21.7 min; SD = 9.5; range 8-45 min; mode = 19). Males and females did not significantly differ on the PEDI-CAT domains, except for daily activities (t(40) = -2.22, p = 0.037). Floor effects were significant for both measures, although the PEDI-CAT showed more floor effects in the mobility (35.7%) and social-cognitive (50%) domains. PEDI-CAT daily activities, mobility, social-cognitive and responsibility domains were all significantly correlated with most of the VABS-3 domains (all rho > 0.5; p < 0.01). Test-rest of the PEDI-CAT was comparable to the VABS-3. Results suggest that the PEDI-CAT is efficient, and minimal training is needed to administer it; however, it lacks specificity and shares a high rate of floor effects with the VABS-3.

The effects of early autism intervention on parents and family adaptive functioning.

This review describes the effects of intervention for young children with autism spectrum disorder (ASD) on parents. Like all children, children with ASD bring both negative and positive experiences for parents and families-from increased resource needs, to higher levels of parenting-related stress, to positive personal growth for family members. It is increasingly recognized that, although children with ASD are the primary targets of early ASD intervention, ASD intervention also impacts parents. From the time emerging developmental concerns begin to be identified, through the process of obtaining a diagnosis and initiating services, parents play a central role in addressing the needs of young children with ASD, including implementing and supporting early intervention. Parents experience the impact of intervention directly, through interaction with providers within the health care and educational systems. Parents also experience indirect impacts of ASD intervention due to accelerated developmental progress of children who are benefitting from services and when children make slower progress than expected or have challenging behaviors. Parental stress and psychological well-being are legitimate targets of intervention and compelling research objectives, needing no additional justification. However, parents are also the major contributors to family adaptive functioning-the activities families employ to support positive outcomes for children with ASD (e.g., family-orchestrated child experiences, parent-child interaction, child health and safety functions; Guralnick, 1997). A parent's ability to carry out adaptive functions is, in part, related to their levels of stress and psychological well-being. Thus, there is a transactional process in which parents are both impacted by and have an impact on ASD interventions for their child. Evaluating the effect of ASD intervention on parents is needed to develop new strategies for helping parents and children with ASD reach their full potential. This review will provide an overview of research on the impact of early ASD intervention on parents. Evidence regarding the impact of three types of intervention (i.e., early intensive behavioral intervention, parent-implemented intervention, and programs directly targeting parent stress) on parent well-being and family adaptive functioning will be reviewed. Potential moderators of the impact of ASD intervention on parents and family adaptive functioning will be discussed. We conclude that research on the impact of ASD intervention on parents of young children with ASD is a promising avenue for improving the lives of children with ASD and their families.

Teaching emotion regulation to children with autism spectrum disorder: Outcomes of the Stress and Anger Management Program (STAMP).

Young children with autism spectrum disorder (ASD) struggle with emotion regulation (ER), which is developmentally preceded by lability/negative affect (L/N), and their parents face unique challenges to parenting and providing assistance. The Stress and Anger Management Program (STAMP) is a cognitive-behavioral treatment designed to address ER deficits in young children with ASD through child skill-building and parent training. The current study evaluated child L/N, ER, and parental confidence outcomes in 4- to 7-year-old children with ASD (N = 23; 19 boys) and their parents randomly assigned to a treatment (n = 12) or a waitlist control group (n = 11). Child L/N decreased, regulation was not significantly changed, and parental confidence regarding the child's ability to manage anger and anxiety increased from pre- to posttreatment in the treatment group, but not in the waitlist group. Implications for future interventions that address ER in children with ASD and their parents are discussed.

Maternal Affect During a Challenging Mother-Child Interaction: The Effects of Broad Autism Phenotype and Respiratory Sinus Arrhythmia Reactivity in Mothers of Children With and Without Autism Spectrum Disorder.

Respiratory sinus arrhythmia (RSA) is proposed to index cognitive and behavioral inflexibility. Broad autism phenotype (BAP) traits are prevalent in family members of children with autism spectrum disorder (ASD). The study investigated whether RSA and BAP traits in mothers of typically developing (TD) children and mothers of children with ASD influence maternal affect. It was hypothesized that these factors would interact to influence mother-child interactions. Twenty-three mother-child dyads participated in a challenging interaction while measuring mother's RSA. Results indicated that mothers of children with ASD show different RSA reactivity than mothers of TD children. Furthermore, preliminary analyses revealed RSA reactivity moderated the relationship between mothers' rigidity and maternal affect during this interaction. Implications for future research and interventions are discussed.

Group Differences in Facial Emotion Expression in Autism: Evidence for the Utility of Machine Classification.

Effective social communication relies, in part, on accurate nonverbal expression of emotion. To evaluate the nature of facial emotion expression (FEE) deficits in children with autism spectrum disorder (ASD), we compared 20 youths with ASD to a sample of typically developing (TD) youth (n = 20) using a machine-based classifier of FEE. Results indicate group differences in FEE for overall accuracy across emotions. In particular, a significant group difference in accuracy of FEE was observed when participants were prompted by a video of a human expressing an emotion, F(2, 36) = 4.99, p = .032, η2 = .12. Specifically, youth with ASD made significantly more errors in FEE relative to TD youth. Findings support continued refinement of machine-based approaches to assess and potentially remediate FEE impairment in youth with ASD.

Marching to the beat of your own drum?: A proof-of-concept study assessing physiological linkage in Autism Spectrum Disorder.

Autism Spectrum Disorder (ASD) is a neurodevelopmental condition characterized by difficulty in dynamically adjusting behavior to interact effectively with others, or social reciprocity. Synchronization of physiological responses between interacting partners, or physiological linkage (PL), is thought to provide a foundation for social reciprocity. In previous work we developed a new technique to measure PL using dynamic linear time series modeling to assess cardiac interbeat interval (IBI) linkage in typically developing same-sex unacquainted dyads (Scarpa et al., 2017). The current article describes a proof-of-concept study with three dyads of young adults with ASD interacting with same-sex unacquainted typically developing (TD) partners. This pilot data is applied to propose potential benefits of using this technique to quantify and assess PL in individuals with ASD, both for basic research and for intervention science. Discussion focuses on applications of this measure to potentially advance knowledge of the biology-behavior link in ASD.

The Under- and Over-Identification of Autism: Factors Associated With Diagnostic Referral.

As reported prevalence and public awareness of Autism Spectrum Disorder (ASD) have grown in recent years, clinicians will likely see increased referrals for suspected ASD. The current study sought to elucidate factors associated with referral for possible ASD, as well as diagnostic outcome among youth referred for suspected ASD. Youth referred for psychological evaluations at an outpatient clinic (N = 69, 6-18 years, 48 male) were categorized into four groups: referred for suspected ASD and diagnosed as such, referred for ASD and not diagnosed as such, not referred for ASD but diagnosed as such, and neither referred for nor diagnosed with ASD. Approximately half of cases referred for suspected ASD did not meet diagnostic criteria. A significant effect of group was found for cognitive ability and anxiety. Youth receiving ASD diagnoses, regardless of whether they were referred for suspected ASD, demonstrated lower cognitive ability than children not receiving ASD diagnoses. Youth neither referred for nor diagnosed with ASD demonstrated lower anxiety than those who were referred and diagnosed. Maternal education significantly differed among the four groups. Although group differences are seen for youth cognitive ability, anxiety, and maternal education, we found no clear indicators differentiating referrals that were "accurate" (i.e., those diagnosed with ASD) and those that were not (i.e., those who did not receive ASD diagnosis). Comorbidity was high in all groups, including those referred primarily for ASD assessment, underscoring the importance of comprehensive assessment regardless of specificity of the referral.

Task Dependency When Evaluating Association Between Facial Emotion Recognition and Facial Emotion Expression in Children with ASD.

The impact of facial emotion recognition (FER) deficits on facial emotion expression (FEE) during interaction with a novel computerized system was investigated in children with ASD (n = 20), in comparison to typically developing (TD) peers (n = 20). Although there was not clear evidence of impaired FEE, children with ASD showed more atypical FEE. In children with ASD, better FER predicted better FEE when the participants were asked to express a labeled emotion (t(18) = - 2.75, p = .01, d = 1.24). The stronger relationship between FER and FEE in children with ASD, relative to controls, suggests that intervention targeting social communication deficits might have maximal effect when both processes are considered.

The Association of the Broader Autism Phenotype with Emotion-Related Behaviors in Mothers of Children With and Without Autism Spectrum Traits.

Broader autism phenotype (BAP) characteristics (pragmatic language deficits, aloofness, and rigidity) are prevalent in families of individuals with autism spectrum disorder (ASD) and may influence emotion-related behaviors. The current study analyzed associations among BAP characteristics with emotion-related behaviors in mothers of children with and without ASD. Twenty-seven mothers completed BAP and emotion regulation (ER) questionnaires. Maternal affect was coded during an interaction task. BAP rigidity negatively correlated with the ER strategy reappraisal. BAP total and pragmatic scores positively correlated with observed negative affect. Associations remained significant in step-wise regressions that controlled for other BAPQ subscale scores. Findings suggest that pragmatic difficulties may interfere with positive mother-child interactions and mothers with high rigidity may benefit from learning adaptive ER strategies.

A Pilot Study of the Effects of Mindfulness-Based Cognitive Therapy on Positive Affect and Social Anxiety Symptoms.

Randomized controlled trials have demonstrated that mindfulness-based cognitive therapy (MBCT) is efficacious in reducing residual depressive symptoms and preventing future depressive episodes (Kuyken et al., 2016). One potential treatment effect of MBCT may be improvement of positive affect (PA), due to improved awareness of daily positive events (Geschwind et al., 2011). Considering social anxiety disorder (SAD) is characterized by diminished PA (Brown et al., 1998; Kashdan, 2007), we sought to determine whether MBCT would reduce social anxiety symptoms, and whether this reduction would be associated with improvement of PA deficits. Adults (N = 22) who met criteria for varied anxiety disorders participated in a small, open-label trial of an 8-week manualized MBCT intervention. Most participants presented with either a diagnosis (primary, secondary, or tertiary) of generalized anxiety disorder (GAD) (N = 15) and/or SAD (N = 14) prior to treatment, with eight individuals meeting diagnostic criteria for both GAD and SAD. We hypothesized participants would demonstrate improvements in social anxiety symptoms, which would be predicted by improvements in PA, not reductions in negative affect (NA). Results of several hierarchical linear regression analyses (completed in both full and disorder-specific samples) indicated that improvements in PA but not reductions in NA predicted social anxiety improvement. This effect was not observed for symptoms of worry, which were instead predicted by decreased NA for individuals diagnosed with GAD and both decreased NA and increased PA in the entire sample. Results suggest that MBCT may be efficacious in mitigating social anxiety symptoms, and this therapeutic effect may be linked to improvements in PA. However, further work is necessary considering the small, heterogeneous sample, uncontrolled study design, and exploratory nature of the study.

Child Autism Spectrum Disorder Traits and Parenting Stress: The Utility of Using a Physiological Measure of Parental Stress.

Caregivers of children with autism spectrum disorder (ASD) report greater stress due to unique parenting demands (e.g.; Estes et al. in Brain Dev 35(2):133-138, 2013). Stress is often studied through self-report and has not been extensively studied using physiological measures. This study compared parenting stress in mothers of children with and without ASD traits. Twenty-seven mother-child dyads participated in an interaction task while measuring mother's heart rate variability (HRV) and mothers self-reported stress levels. Results demonstrated that while self-report and physiological stress measures were not correlated, ASD symptomology did account for HRV change score (i.e., more severe ASD symptoms were positively related to HRV change). This may reflect an atypical coping response. Implications for using physiological indicators for studying parenting stress are explored.

Side by side: Modeling dyadic physiological linkage in strangers.

Physiological linkage (PL) refers to coordinated physiological responses among interacting partners (Feldman, 2012a), thought to offer mammals evolutionary advantages by promoting survival through social groups. Although PL has been observed in dyads who are familiar or have close relationships (e.g., parent-infant interactions, romantic couples), less is known with regard to PL in stranger dyads. The current study used dynamic linear time series modeling to assess cardiac interbeat interval linkage in 26 same-gender stranger dyads (17 female and 9 male dyads; 18-22 years old) while they spoke or wrote about emotional or neutral life events. The estimated coefficients in bivariate regression models indicated small but statistically significant PL effects for both male and female dyads. The PL effect was stronger for female dyads, extending to a lag of 4 seconds. For male dyads, the effect was statistically significant but weaker than for female dyads, extending only to a lag of 1 second. No statistically significant differences in PL were noted for type of task (i.e., baseline, writing, speaking, listening) or with differing task emotional content. Frequency domain analysis based on the estimated dynamic models yielded similar results. Our results suggest that PL can be detected among strangers in this setting and appears to be stronger and longer-lasting in women. Our findings are discussed in terms of the importance of biological synchrony in humans, gender differences, and possible implications for objective measurement of social reciprocity at a physiological level. (PsycINFO Database Record

Comparing oxytocin and cortisol regulation in a double-blind, placebo-controlled, hydrocortisone challenge pilot study in children with autism and typical development.

BACKGROUND: Children with autism spectrum disorder (ASD) show marked impairment in social functioning and poor adaptation to new and changing contexts, which may be influenced by underlying regulatory processes. Oxytocin (OT) and cortisol are key neuromodulators of biological and behavioral responses, show a synergistic effect, and have been implicated in the neuropathological profile in ASD. However, they are rarely investigated together. The purpose of the pilot study was to evaluate the relationship between cortisol and OT in children with ASD under baseline and physiological stress (hydrocortisone challenge) conditions. Arginine vasopressin (AVP), structurally similar to OT, was also examined. METHODS: A double-blind, placebo-controlled, randomly assigned, crossover design was employed in 25 children 8-to-12 years with ASD (N = 14) or typical development (TD, N = 11). A low dose of hydrocortisone and placebo were administered via liquid suspension. Analysis of variance (ANOVA) was used to examine the within-subject factor "Condition" (hydrocortisone/placebo) and "Time" (pre and post) and the between-subject factor "Group" (ASD vs. TD). Pearson correlations examined the relationship between hormone levels and clinical profile. RESULTS: There was a significant Time × Condition × Group interaction F (1.23) = 4.18, p = 0.05 showing a rise in OT during the experimental condition (hydrocortisone) and a drop during the placebo condition for the TD group but not the ASD group. There were no group differences for AVP. Hormone levels were associated with social profiles. CONCLUSIONS: For the TD group, an inverse relationship was observed. OT increased during physiological challenge suggesting that OT played a stress-buffering role during cortisol administration. In contrast for the ASD group, OT remained unchanged or decreased during both the physiological challenge and the placebo condition, suggesting that OT failed to serve as a stress buffer under conditions of physiological stress. While OT has been tied to the social ability of children with ASD, the diminished moderating effect of OT on cortisol may also play a contributory role in the heightened stress often observed in children with ASD. These results contribute to our understanding of the growing complexity of the effects of OT on social behavior as well as the functional interplay and differential regulation OT may have on stress modulation.

Emotion Dysregulation and Anxiety in Adults with ASD: Does Social Motivation Play a Role?

Young adults with ASD and no intellectual impairment are more likely to exhibit clinical levels of anxiety than typically developing peers (DSM-5, American Psychiatric Association, 2013). This study tests a mechanistic model in which anxiety culminates via emotion dysregulation and social motivation. Adults with ASD (49 males, 20 females) completed self-report measures on emotion regulation, caregivers completed measures on ASD severity and both on social anxiety. Results indicated that emotion dysregulation (p < .001; p < .05) and social motivation (p < .05, p < .001) significantly predicted social anxiety as reported by caregivers and young adults respectively. However, social motivation did not appear to play a moderating role in the relationship between emotion regulation and anxiety, even when controlling for social awareness. Significant predictor variables of social anxiety varied based on reporter (i.e. caregiver versus young adult), with difficulty engaging in goal-directed behaviors during negative emotions serving as the only shared predictor.

Peer-mediated theatrical engagement for improving reciprocal social interaction in autism spectrum disorder.

The hallmark characteristic of autism spectrum disorder (ASD) is poor reciprocal social communication. Interventions designed to improve this core deficit are critically needed. Social skills interventions such as direct training, peer mediation, and video modeling have contributed to improvements in various social skills in children with ASD. This paper reviews existing social competence interventions available for children with ASD while highlighting hypothesized critical components for advancing, maintaining, and generalizing skills, which include (1) peer mediation, (2) active learning, and (3) implementation in supportive, natural contexts. As a framework for these approaches, this conceptual paper describes SENSE Theatre, a novel intervention that combines trained peers that facilitate the performance-based theatrical treatment delivered in a supportive, community-based environment. A review of previous research shows early feasibility, setting the stage for more rigorous studies to aid in developing a standardized intervention package.