Promoting early autism detection and intervention in underserved communities: study protocol for a pragmatic trial using a stepped-wedge design.

BACKGROUND: Despite the known benefits of early, specialized intervention for toddlers with Autism Spectrum Disorder (ASD), access to such intervention remains limited. This pragmatic trial examines a novel healthcare delivery model (Screen-Refer-Treat [SRT]), which capitalizes upon existing health care and early intervention (EI) infrastructure to increase community capacity for ASD detection and treatment before age 3, when it is likely to have the greatest impact. This model comprises three components: (1) universal use of Stage 1 ASD screening by primary care providers (PCPs) at 18-month well-child visits (i.e., Screen); (2) immediate referral of positive screens to a community-based EI program (i.e., Refer); and (3) provision of an inexpensive, evidence-based ASD-specialized treatment by EI providers, after verifying ASD risk with a Stage 2 screen (i.e., Treat). This paper describes our research design and the initial successes, challenges, and adaptations made during the early implementation phase. METHOD/DESIGN: A stepped-wedge cluster RCT was used to implement the SRT model sequentially in four diverse Washington State counties ("clusters"). Counties are randomly assigned to the time of receipt of the SRT intervention, which comprises training workshops and technical assistance focused on the use of evidence-based ASD screening and intervention tools. Separate cohorts of families with toddlers (16-35 months old) with and without ASD concerns are recruited before and after the SRT intervention from participating PCP practices and EI programs. PCPs and EI providers complete measures on their screening, referral, and intervention practices before and after the SRT intervention. Each family cohort completes surveys about their well-being, parenting efficacy, health care satisfaction, and toddler's social-communicative behaviors. CONCLUSION: This trial is the first of its kind to work simultaneously with two service delivery systems with the goal of improving early detection and treatment for ASD. Our approach was successful in attaining buy-in from PCPs and EI providers, building and maintaining partnerships with providers, and achieving high levels of retention and survey completion. Fostering provider engagement and problem-solving issues together as partners were integral to overcoming the main challenges. Numerous lessons have been learned thus far, which have applicability for implementation researchers in ASD and those in other fields. TRIAL REGISTRATION: The registration number for this trial is NCT02409303 and it was posted on ClinicalTrials.gov on April 6, 2015.

Longitudinal prediction of language emergence in infants at high and low risk for autism spectrum disorder.

This study used a prospective longitudinal design to examine the early developmental pathways that underlie language growth in infants at high risk (n = 50) and low risk (n = 34) for autism spectrum disorder in the first 18 months of life. While motor imitation and responding to joint attention (RJA) have both been found to predict expressive language in children with autism spectrum disorder and those with typical development, the longitudinal relation between these capacities has not yet been identified. As hypothesized, results revealed that 15-month RJA mediated the association between 12-month motor imitation and 18-month expressive vocabulary, even after controlling for earlier levels of RJA and vocabulary. These results provide new information about the developmental sequencing of skills relevant to language growth that may inform future intervention efforts for children at risk for language delay or other developmental challenges.

Diagnostic stability in young children at risk for autism spectrum disorder: a baby siblings research consortium study.

BACKGROUND: The diagnosis of autism spectrum disorder (ASD) made before age 3 has been found to be remarkably stable in clinic- and community-ascertained samples. The stability of an ASD diagnosis in prospectively ascertained samples of infants at risk for ASD due to familial factors has not yet been studied, however. The American Academy of Pediatrics recommends intensive surveillance and screening for this high-risk group, which may afford earlier identification. Therefore, it is critical to understand the stability of an ASD diagnosis made before age 3 in young children at familial risk. METHODS: Data were pooled across seven sites of the Baby Siblings Research Consortium. Evaluations of 418 later-born siblings of children with ASD were conducted at 18, 24, and 36 months of age and a clinical diagnosis of ASD or Not ASD was made at each age. RESULTS: The stability of an ASD diagnosis at 18 months was 93% and at 24 months was 82%. There were relatively few children diagnosed with ASD at 18 or 24 months whose diagnosis was not confirmed at 36 months. There were, however, many children with ASD outcomes at 36 months who had not yet been diagnosed at 18 months (63%) or 24 months (41%). CONCLUSIONS: The stability of an ASD diagnosis in this familial-risk sample was high at both 18 and 24 months of age and comparable with previous data from clinic- and community-ascertained samples. However, almost half of the children with ASD outcomes were not identified as being on the spectrum at 24 months and did not receive an ASD diagnosis until 36 months. Thus, longitudinal follow-up is critical for children with early signs of social-communication difficulties, even if they do not meet diagnostic criteria at initial assessment. A public health implication of these data is that screening for ASD may need to be repeated multiple times in the first years of life. These data also suggest that there is a period of early development in which ASD features unfold and emerge but have not yet reached levels supportive of a diagnosis.

Using virtual multiteam systems to conduct a multisite randomized clinical trial in the part C early intervention system: Benefits, challenges, and lessons learned.

BACKGROUND/AIMS: COVID-19 necessitated a shift to virtual data collection for many research projects, providing the opportunity for novel approaches to carrying out multi-site clinical trials. Virtual multiteam systems (VMTS) are a type of team structure in which multiple geographically dispersed teams collaborate using technology-mediated communication. The article presents a case study of our use of VMTS, in response to COVID-19, to carry out a multisite randomized hybrid effectiveness-implementation trial of a caregiver-implemented intervention. METHODS: We describe how we modified our team structure from predominantly site-specific, co-located teams to predominantly cross-site, virtual teams. We then present examples of how we have conducted the two primary data collection activities virtually. To demonstrate the feasibility of this approach, we present participant demographic information, the percent of cross-site data collection activities, and fidelity data. RESULTS: In the first 20 months of data collection, we have enrolled 108 EI providers and 132 families, with 17% and 9% attrition respectively. The family sample is highly diverse in terms of race/ethnicity, parent education, and household income. The majority of provider training activities and roughly 50% of family assessment activities have been conducted cross-site. Fidelity is high, with no differences across site. CONCLUSIONS: Our data illustrate the feasibility of using virtual teams, training, and assessment in a multisite clinical trial in the Part C system. We discuss the strengths and challenges of this approach, as well as lessons learned to facilitate the planning of future multisite randomized clinical trials which may benefit from this approach. CLINICAL TRIALS: NCT05114538.

The implementation of the screening tool for autism in toddlers in Part C early intervention programs: An 18-month follow-up.

LAY ABSTRACT: The early detection of autism spectrum disorder can lead to access to autism spectrum disorder-specific services that have been shown to have a large impact on a child's overall development. Although a stable diagnosis of autism spectrum disorder can be made by age 2 years, most children are not diagnosed until much later. To address this issue, this study examined the effectiveness of training Part C Early Intervention providers to use an interactive autism spectrum disorder screening tool, the Screening Tool for Autism in Toddlers. Sixty-nine providers attended a 1-day training workshop on the use of the Screening Tool for Autism in Toddlers. After the workshop, providers reported increased knowledge about recognizing the early signs of autism spectrum disorder, and about 45% of the providers reported using the Screening Tool for Autism in Toddlers with families in their caseloads 18 months after the training. These results suggest that the Screening Tool for Autism in Toddlers is feasible for use within Early Intervention settings. In addition, they suggest that specific providers might serve as a screening "point-person," rather than expecting the Screening Tool for Autism in Toddlers to be used by all providers. Future research should aim to identify specific characteristics of agencies or providers that might be best suited for using the Screening Tool for Autism in Toddlers.

Telehealth Delivery in Part C Early Intervention: Provider and Caregiver Perspectives.

Telehealth is a promising modality for Part C early intervention (EI), services typically implemented face-to-face in home and community settings. Barriers to telehealth in EI reported prior to COVID-19 included lack of training and access to reliable internet. The abrupt telehealth shift at the onset of the pandemic did not permit a phased adoption approach. This mixed-methods study aimed to characterize perspectives of service changes resulting from the telehealth transition. Providers (n = 39) and caregivers (n = 11) completed surveys about perceptions towards the telehealth switch. All providers indicated at least one aspect of services had changed. Approximately half of caregivers reported satisfaction with services decreased and half that satisfaction remained the same. Implications for telehealth in EI beyond the pandemic are discussed.

Separate scoring algorithms for specific identification priorities optimize the screening properties of the Screening Tool for Autism in Toddlers (STAT).

The Screening Tool for Autism in Toddlers (STAT) is a validated stage-2 autism spectrum disorder (ASD) screening measure that takes 20 minutes to administer and comprises 12 play-based items that are scored according to specific criteria. This study examines an expanded version (STAT-E) that includes the examiner's subjective ratings of children's social engagement (SE) and atypical behaviors (AB) in the scoring algorithm. The sample comprised 238 children who were 24-35 months old. The STAT-E assessors had limited ASD experience to mimic its use by community-based non-specialists, and were trained using a scalable web-based platform. A diagnostic evaluation was completed by clinical experts who were blind to the STAT-E results. Logistic regression, ROC curves, and classification matrices and metrics were used to determine the screening properties of STAT-E when scored using the original STAT scoring algorithm versus a new algorithm that included the SE and AB ratings. Inclusion of the SE and AB ratings improved positive risk classification appreciably, while the specificity declined. These results suggest that the STAT-E using the original STAT scoring algorithm optimizes specificity, while the STAT-E scoring algorithm with the two new ratings optimizes the positive risk classification. Using multiple scoring algorithms on the STAT may provide improved screening accuracy for diverse contexts, and a scalable web-based tutorial may be a pathway for increasing the number of community providers who can administer the STAT and contribute toward increased rates of autism screening.

Reducing Barriers to Autism Screening in Community Primary Care: A Pragmatic Trial Using Web-Based Screening.

OBJECTIVE: To determine whether an intervention addressing both logistical and knowledge barriers to early screening for autism spectrum disorder (ASD) increases evidence-based screening during 18-month well-child visits and primary care providers' (PCPs') perceived self-efficacy in caring for children with ASD. METHODS: Forty-six PCPs from 10 diverse practices across four counties in Washington State participated. PCPs attended a 2-hour training workshop on early recognition and care for toddlers with ASD and use of a REDCap-based version of the Modified Checklist for Autism in Toddlers-Revised with Follow-up (webM-CHAT-R/F) that provided automated presentation and scoring of follow-up questions. Data were collected at baseline and 6 months following each county's training window. PCPs' screening methods and rates and perceived self-efficacy regarding ASD care were measured by self-report and webM-CHAT-R/F use was measured via REDCap records. RESULTS: At follow-up, 8 of the 10 practices were using the webM-CHAT-R/F routinely at 18-month visits. The proportion of PCPs reporting routine M-CHAT screening increased from 82% at baseline to 98% at follow-up (16% increase, 95% confidence interval [CI] 3%-28%; McNemar exact P = .02). The proportion using the M-CHAT-R/F follow-up interview questions increased from 33% to 82% (49% increase, 95% CI 30%-68%, exact McNemar test, P < .001). Significant increases in self-efficacy were found for all seven areas assessed (Ps ≤ .008). CONCLUSIONS: This brief intervention increased PCPs' self-reported valid use of the M-CHAT-R/F at 18 months and their self-efficacy regarding ASD care. Combining educational information with a web-based ASD screen incorporating the M-CHAT-R/F follow-up questions may increase universal ASD screening with improved fidelity.

Brief Report: Perceptions of Family-Centered Care Across Service Delivery Systems and Types of Caregiver Concerns About Their Toddlers' Development.

Family-centered care represents a collaborative partnership between caregivers and service providers, and is associated with positive caregiver and child outcomes. This approach may be especially important for caregivers with early concerns about autism, as service providers are often the gateway to appropriately-specialized intervention. Perceptions of family-centered care received from primary care providers (PCPs) and Part C Early Intervention (EI) providers were rated by two groups of caregivers: those concerned about autism (n = 37) and those concerned about another developmental problem (n = 22), using the Measure of Processes of Care (MPOC-20). Ratings did not differ across caregiver groups, but both groups rated EI providers significantly higher than PCPs, which may reflect systems-level differences between primary care and EI.

Caregiver Attributions of Toddlers' Behaviors: A Comparison Between Groups of Children with Differing Developmental Concerns.

Little is known about the attributional patterns of caregivers of autistic children, particularly in relation to caregivers of children with other developmental or behavioral disorders. This study examined differences in caregiver attributions of child behavior between three groups: toddlers with (1) Autism spectrum disorder (ASD) or ASD concerns; (2) Other developmental concerns; and (3) No concerns. Qualitative descriptions of actual child behaviors were coded using a three-stage content analysis. Regression analyses were utilized to determine if group membership predicted types of positive and challenging behaviors caregivers endorsed, as well as their attributions of these behaviors. Caregivers of children with ASD or ASD concerns endorsed similar types of behaviors, but rated their child's positive behaviors as less characteristic of their child and more a function of the particular situation, less stable or permanent, and less controllable as compared to caregivers of toddlers with other developmental or no concerns. Additionally, they rated their child's challenging behaviors as more stable or permanent and less controllable as compared to caregivers of toddlers with other developmental concerns or no concerns. These findings suggest that caregivers of children with ASD and ASD related concerns may be vulnerable to a negative attributional pattern, which can have important implications for child and family functioning and overall quality of life.

Characterizing Available Tools for Synchronous Virtual Assessment of Toddlers with Suspected Autism Spectrum Disorder: A Brief Report.

The COVID-19 pandemic, and associated social distancing mandates, has placed significant limitations on in-person health services, requiring creative solutions for supporting clinicians engaged in the diagnosis of autism spectrum disorder (ASD). This report describes the five virtual instruments available at the time of manuscript development for use by experienced clinicians making diagnostic determinations of ASD for toddlers across the 12- to 36-months age range. We focus on synchronous virtual assessments in which clinicians guide the child's caregiver through a range of assessment activities and observe spontaneous and elicited behaviors. Assessments are compared on dimensions of targeted behavioral domains, specific activities and presses employed, scoring approaches, and other key logistical considerations to guide instrument selection for use in varied clinical and research contexts.

A method for defining the CORE of a psychosocial intervention to guide adaptation in practice: Reciprocal imitation teaching as a case example.

LAY ABSTRACT: Interventions that support social communication include several "components," or parts (e.g. strategies for working with children and families, targeting specific skills). Some of these components may be essential for the intervention to work, while others may be recommended or viewed as helpful but not necessary for the intervention to work. "Recommended" components are often described as "adaptable" because they can be changed to improve fit in different settings where interventions are offered or with different individuals. We need to understand which parts of an intervention are essential (and which are adaptable) when translating interventions from research to community settings, but it is challenging to do this before studying an intervention in the community. This article presents the CORE (COmponents & Rationales for Effectiveness) Fidelity Method-a new method for defining the essential components of evidence-based interventions-and applies it to a case example of Reciprocal Imitation Teaching, an intervention that parents are taught to deliver with their young children with social communication delays. The CORE Fidelity Method involves three steps: (1) gathering information from multiple sources; (2) integrating information from previous research and theory; and (3) drafting a CORE model for ongoing use. The benefits of using the CORE Fidelity Method may include: (1) improving consistency in intervention and research materials to help all providers emphasize the most important skills or strategies; (2) clarifying which parts of the intervention can be adapted; and (3) supporting future research that evaluates which intervention components work and how they work.

A randomized controlled trial of Hanen's 'More Than Words' in toddlers with early autism symptoms.

BACKGROUND: This randomized controlled trial compared Hanen's 'More than Words' (HMTW), a parent-implemented intervention, to a 'business as usual' control group. METHODS: Sixty-two children (51 boys and 11 girls; M age = 20 months; SD = 2.6) who met criteria for autism spectrum disorders (ASD) and their parents participated in the study. The HMTW intervention was provided over 3.5 months. There were three measurement periods: prior to randomization (Time 1) and at 5 and 9 months post enrollment (Times 2 and 3). Children's communication and parental responsivity were measured at each time point. Children's object interest, a putative moderator, was measured at Time 1. RESULTS: There were no main effects of the HMTW intervention on either parental responsivity or children's communication. However, the effects on residualized gains in parental responsivity from Time 1 to both Times 2 and 3 yielded noteworthy effect sizes (Glass's Δ = .71, .50 respectively). In contrast, there were treatment effects on child communication gains to Time 3 that were moderated by children's Time 1 object interest. Children with lower levels of Time 1 object interest exhibited facilitated growth in communication; children with higher levels of object interest exhibited growth attenuation. CONCLUSIONS: The HMTW intervention showed differential effects on child communication depending on a baseline child factor. HMTW facilitated communication in children with lower levels of Time 1 object interest. Parents of children who evidence higher object interest may require greater support to implement the HMTW strategies, or may require different strategies than those provided by the HMTW curriculum.

Web-based training in early autism screening: results from a pilot study.

BACKGROUND: Lack of familiarity with early signs of autism by community service providers has resulted in significant delays in children receiving early intervention services necessary to improve long-term outcomes. The Screening Tool for Autism in Toddlers and Young Children (STAT) was specifically developed to identify early behavioral features of autism. Although STAT training has been available for years, access is limited because of few STAT trainers and geographic concerns. This study evaluated the efficacy and acceptability of Web-based training of the STAT as a means of increasing accessibility to this training. MATERIALS AND METHODS: Thirty professionals from three geographic areas participated. Roughly 1 of 3 had little or no training on autism assessment. The tutorial contains a general overview, administration and scoring conventions, and item-specific content and concepts. Participants completed a pretest and then completed the STAT tutorial at their own pace, followed by a post-test and a user satisfaction questionnaire. RESULTS: Mean scores on STAT concepts significantly improved after taking the tutorial (p<0.001). At pretest, only 1 person (3%) obtained correct scores on at least 80% of the items (a priori cutoff for a "pass"), compared with 22 (73%) at post-test (p<0.001). The majority of trainees enjoyed taking the tutorial, thought it was well organized, relevant, interesting, and useful, and felt it was easy to understand and operate. DISCUSSION: Results support Web-based training as a promising method for promoting early identification of autism and may help overcome problems associated with the critical shortage of autism-screening professionals.

A web-based tutorial for parents of young children with autism: results from a pilot study.

OBJECTIVE: Early intervention can significantly improve long-term outcomes for children with autism. Unfortunately, many children do not receive early intervention services due to a critical shortage of trained professionals in this area. To bridge this gap, we evaluated a Web-based parent training tutorial (Enhancing Interactions), based on evidence-based practices and utilizing the Web-based platform to maximize learning. METHODS: Twenty-three parents with a child between 18 months and 6 years with an autism spectrum disorder participated. Pre- and posttest scores of parents' knowledge were used to evaluate tutorial effectiveness. The system usability scale (SUS) evaluated technical user-friendliness and the user satisfaction questionnaire (USQ), gauged satisfaction with content. RESULTS: The mean number of correct items on the posttest significantly increased, from 12.6 to 20.4, p<0.001. The mean SUS score was 85 (standard deviation=17), corresponding to a score of "excellent." All participants found the tutorial user friendly, well integrated, and 96% (all but one participant) thought it was easy to use, felt confident using the technical features, and would use a tutorial like this again. On the USQ, all participants found that the tutorial was well organized, clearly presented, and easy to understand; that it increased their knowledge about communicating with their child; and that they felt capable of applying these techniques with their child. CONCLUSIONS: The tutorial appears effective in increasing parents' knowledge with high user satisfaction.

The implementation of reciprocal imitation training in a Part C early intervention setting: A stepped-wedge pragmatic trial.

Despite the development of several evidence-based Naturalistic Developmental Behavioral Interventions (NDBIs), very few have been adapted for use in community-based settings. This study examines the implementation of Reciprocal Imitation Training (RIT)-an NDBI-by community Early Intervention (EI; IDEA Part C) providers serving toddlers from birth to 3 years. Of the 87 EI providers enrolled from 9 agencies in 4 counties across Washington State, 66 were included in the current sample. A stepped-wedge design was used to randomly assign counties to the timing of RIT training workshops. Self-report measures of practice and self-efficacy regarding ASD care were collected at baseline (T1, T2) and 6-months and 12-months post-training (T3, T4). At T3 and T4, providers reported on RIT adoption and rated items about RIT feasibility and perceived RIT effectiveness; at T4, they also reported on child characteristics that led to RIT use and modifications. From pre-training to post-training, there were significant increases in providers' self-efficacy in providing services to children with ASD or suspected ASD. At T3 and T4, provider ratings indicated high levels of RIT adoption, feasibility, and perceived RIT effectiveness. At T4, providers indicated that they most commonly: (a) initiated RIT when there were social-communication or motor imitation delays, or an ASD diagnosis; and (b) made modifications to RIT by repeating elements, blending it with other therapies, and loosening its structure. While additional research is needed, RIT may help families get an early start on accessing specialized treatment within an established infrastructure available across the United States. LAY SUMMARY: Reciprocal imitation training (RIT) is an evidence-based treatment for ASD that might be a good fit for use by intervention providers in widely accessible community-based settings. After attending an educational workshop on RIT, providers reported feeling more comfortable providing services to families with ASD concerns, used RIT with over 400 families, and believed that RIT improved important social communication behaviors.

Parent utilization of ImPACT intervention strategies is a mediator of proximal then distal social communication outcomes in younger siblings of children with ASD.

LAY ABSTRACT: Later born siblings of children with autism spectrum disorders (ASD) are at elevated risk for language delay or ASD. One way to manage this risk may be for parents to use techniques taught in Improving Parents as Communication Teachers (ImPACT) with the younger siblings during the period in which language delay and ASD may be too subtle to be diagnosed. ImPACT targets children's play, imitation, and communication skills. Improvement in these skills may reduce the severity of language delays and social communication deficits associated with ASD. In this study, 97 younger siblings of children with ASD and their primary parents were randomly assigned to ImPACT or a control group. We measured whether parents used ImPACT teaching strategies and whether children used the skills that ImPACT targets. We also measured children's later language ability and social communication skills. The results confirmed our predictions that parents' use of ImPACT strategies improves language ability by improving children's motor imitation and communication skills. Use of ImPACT also had a positive effect on children's language delay and ASD symptoms, supporting the clinical value of the findings. The study's methodological strengths make this one of the most rigorous tests of ImPACT and supports one way to manage the risk of language delay and ASD in younger siblings of children with ASD.

For which younger siblings of children with autism spectrum disorder does parent-mediated intervention work?

LAY ABSTRACT: In this second of two primary papers, we examined two pre-intervention characteristics that might describe for whom a parent-implemented intervention, "Improving Parents As Communication Teachers," worked. Investigators randomized 97 high-risk siblings and their primary parent to either the Improving Parents As Communication Teachers or control group, used intent-to-treat analysis, and used assessors and coders who were blinded to group assignment. We predicted that a combined risk score (incorporating young siblings' sex, multiplex status, and behavioral risk) would describe the subgroup for whom Improving Parents As Communication Teachers affected the targeted skills related to reducing communication challenges. We also predicted that pre-intervention level of parents' depressive symptoms would describe the parents whose parenting stress and effectiveness as parents would be improved by learning to use Improving Parents As Communication Teachers. In girls with only one older sibling with autism spectrum disorder and who scored at low risk on an autism spectrum disorder screen, parental receipt of Improving Parents As Communication Teachers training had an indirect effect on children's expressive language ability or autism spectrum disorder diagnosis through earlier effects on high-risk siblings' intentional communication or expressive vocabulary. We did not confirm our prediction regarding Improving Parents As Communication Teachers' effect on parenting-related stress or sense of parenting effectiveness.

DSM-5 symptom expression in toddlers.

LAY ABSTRACT: Children with autism show more social-communication symptoms and repetitive behaviors than children with typical development or those diagnosed with other developmental disorders; however, non-autistic children often show some behaviors that are associated with autism. We compared the behavioral reports from caregivers of children in these three groups to identify the behaviors that were specific to autism. Children with autism were found to show more of these behaviors, and behaviors that are particularly indicative of autism were identified. These behaviors included social symptoms (approaching others to interact, showing things, looking back while showing, responding to an approaching child, spontaneous imitation) and repetitive behavior symptoms (specific, inflexible play, unusual body movements, strong specific interest, carrying around an unusual object, sensory seeking, and sensory hyper-reactivity).These findings may aid professionals in determining the most appropriate diagnosis for a child between the ages of 12 and 36 months.

Early Intervention Providers' Perspectives About Working with Families of Toddlers with Suspected ASD: A Qualitative Study.

Part C early intervention (EI) providers are at the front line of service provision for children under 3 years old with developmental delays or disabilities. As such, they can play a key role in both the early detection of autism spectrum disorder (ASD) and the provision of ASD-specialized treatment. Focus groups were conducted with 25 EI providers from three agencies in a Northwestern U.S. county to understand their role in the identification of ASD and communication about ASD concerns to families. Results revealed the tension that providers experience between maintaining a positive and supportive relationship with families and raising the issue of possible ASD. Cultural influences affecting ASD care and suggestions for desired resources were also discussed.