"Opt-In" Early Intervention Program for Children Waiting to Receive an Autism Diagnosis

BACKGROUND. The increasing prevalence of autism spectrum disorder (ASD), now at 1 in 36 children, highlights an urgent need for early interventions that are both effective and efficient, given the social and economic impact of the challenges experienced by individuals on the autism spectrum and their families. Several clinical trials have documented the benefits of parent-mediated interventions starting early in life, even before a formal autism diagnosis is established. Symptoms of autism manifest across the lifespan and can be diagnosed as early as 12 months of age. Although parents often identify concerns about their child's development during the first two years of life, the median age of diagnosis in the United States continues to be 49 months. Even following formal diagnosis, services often do not begin for another 9 months. These cumulative waiting times often result in children receiving minimal early intervention prior to school entry. Additionally, health disparities disproportionally affect access to services for families from minoritized and socioeconomically disadvantaged backgrounds. Families who are low-income, have low parental educational attainment, or are from minoritized racial and ethnic backgrounds not only experience delays in receiving diagnoses, but are also less likely to receive high-quality services following diagnosis. These inequities have been exacerbated by the COVID-19 pandemic.

Barriers to accessing diagnostic services limit the opportunity to receive autism-specific interventions at the age for which they are shown to be maximally beneficial. Children on the autism spectrum whose needs are not addressed in a timely fashion are at an increased risk of impairments in cognitive, adaptive, and social dimensions of well-being, as well as physical and mental health issues. Further, the 'passive waiting' time in the waitlist is harmful for parental wellbeing and mental health, with research reporting feelings of frustration, worry, and stress in parents experiencing the yearlong 'odyssey' from initial concerns to diagnosis and intervention referral.

Importantly, such harmful delays in accessing autism-specific early interventions are potentially avoidable, as many caregivers of children later diagnosed with autism identify concerns about their child's development before her or his second birthday, often as early as 13 months of age. Many difficulties identified by parents during infancy and toddlerhood can be successfully addressed through evidence-based intervention practices. For example, early emerging differences in verbal and nonverbal communication are amenable to improvements through targeted parent-mediated interventions, with benefits documented across multiple dimensions of wellbeing (i.e., adaptive and social-communicative functioning).

Despite advances in this area, the potential benefits of early interventions remain largely untapped, as wait times to receive a formal diagnosis of autism are such a significant barrier that they have been referred to as a "crisis" in the field, with parents reporting an average wait time of 1.2 years in the US. The American Academy of Pediatrics recommends simultaneous referrals for clinical diagnostic evaluation and early intervention for young children with suspected autism - however, most children without an autism diagnosis receive low-intensity, generic early interventions rather than autism-specific early intervention programs that rely on evidence-based practices. Accordingly, key stakeholders, including caregivers and providers, have voiced the need for facilitating earlier access to intervention options to counteract the harmful impact of the waitlist crisis. Accordingly, the current project focuses on circumventing the roadblock of delayed access to diagnosis and intervention by empowering caregivers to address their children's needs while they are waiting to be seen for a formal diagnosis.

One viable option to accomplish this goal is the delivery of parent-mediated intervention programs that do not require a previous diagnosis or enrollment in formal early intervention services. In particular, recent research has shown the promise of self-directed online intervention programs for caregivers of autistic children. These programs involve independent access to online resources that caregivers can utilize at their own pace and individualize to their own children by selecting content relevant to the specific difficulties experienced by their children. Implementation of the intervention strategies might be exclusively self-directed (i.e., parents deliver the intervention strategy without any assistance from a provider) or based on a hybrid format, whereby a clinician is available to provide guidance and clarification related to the program's content, although without explicit coaching and feedback. Across formats, self-directed programs do not rely on diagnostic and intervention services for eligibility, insurance and billing purposes, providing a potential avenue for circumventing the roadblock of delayed access to diagnosis and intervention. Research has documented that despite not being coached by experienced clinicians, caregivers have expressed a high degree of acceptability and satisfaction with the self-directed format, demonstrated fidelity of implementation, experienced gains in knowledge and their use of the intervention strategies was associated with child improvements following parent completion of programs. However, substantial variation in outcomes in response to different self-directed programs was documented, and existing research only focuses on children who were already diagnosed. Therefore, the potential impact of a self-directed intervention program during families' enrolment in waitlists for a diagnostic evaluation remains unexplored.

Against this background, our project focuses the implementation and evaluation of the "Online Parent Training in Early Behavioral Intervention" (OPT-In-Early), a self-directed online resource for caregivers of children with autism or high likelihood of autism which will be made available during their enrolment in waitlists for a diagnostic evaluation. Key features of Opt-In-Early include: (a) a comprehensive scope (i.e., multiple domains are addressed), (b) the use of a Naturalistic Developmental Behavioral Intervention approach, i.e., evidence-supported strategies that blend behavioral and developmental components to address the needs of autistic children and their families, (c) a combination of required and optional modules, so that caregivers can learn autism-specific strategies and focus on areas most relevant to their specific child's needs, and (d) a hybrid format including access to web-based materials designed for self-directed implementation, as well as optional access to remote consultation with a clinician for advice, clarification and support (e.g., selection of appropriate modules).

APPROACH.

The overarching goal of our project is to evaluate the utility of the OPT-In-Early program for caregivers of toddlers who are on a waitlist for receiving diagnostic services. The OPT-In-Early program centers on the use of naturalistic, developmental, and behavioral principles to increase motivation and engagement, teach skills, and generalize skills across settings. The program emphasizes the importance of utilizing the parent-child relationship, the child's interests, and daily routines to facilitate skill acquisition, and to create developmentally appropriate learning experiences, to value small gains, and to use evidence-based applied behavior analytic principles including task analysis, prompting and prompt fading, and contingent reinforcement. Optional modules focus on specific needs, such as toilet training and picky eating. To evaluate whether the OPT-In-Early program is an acceptable and effective tool for caregivers who are on a waitlist for receiving diagnostic services, the following specific aims will be addressed:

1. Comparing outcomes for 120 toddlers aged 16-48 months whose caregivers are on a waitlist to receive a formal autism diagnosis, randomly assigned to receive either OPT-In-Early or no intervention for six months period. The investigators hypothesize that children randomized to the OPT-In-Early condition will have superior gains. 2. Examining feasibility and acceptability of the intervention (both hypothesized to be high). 3. As an exploratory aim, the investigators will examine theoretically- and empirically- motivated factors associated with intervention engagement and child outcomes.

Participants and Setting. Participants will be 120 toddler-caregiver dyads (with each dyad including one toddler and one caregiver) who are on a waitlist to receive a formal diagnostic evaluation due to autism concerns identified by either parents or professionals. As the study does not involve in-person contact between investigators and participants, participants will be recruited across any geographical area.

Inclusion criteria include (a) age between 16-48 months, (b) parental consent, (c) being on a waitlist to receive a formal diagnostic evaluation at the time of parent consent due to autism concerns, (d) parents speak English, (e) an initial score of 8 or greater or Follow-Up score of 2 or greater on the Modified Checklist for Autism in Toddlers, Revised, with Follow-Up (M-CHAT-R/F) as an indication of autism likelihood.

Exclusion criteria include (a) having already received a formal diagnosis of autism, (b) enrolment in early intervention programs delivering more than 5 h per week of evidence-based autism-specific interventions at the time of consent, (c) child has a gestational age less than 36 weeks associated with a birth weight less than 2500 g, (d) the child is not yet walking, or has severe hearing and/or vision impairment that is uncorrected with aids, or known neurological disorder or genetic syndrome (e.g., Down syndrome).

Research Design. Our aims will be addressed using an intent-to-treat randomized controlled trial, with participants randomized to either the OPT-In-Early intervention or a waitlist control condition, stratified by age, language level and parental education. Data will be collected from each family prior to random assignment (baseline) and following completion of the 6-month trial period (or earlier for families electing to discontinuing their participation from the trial). The investigators will use REDCap, a HIPAA-compliant data management software to store data. To ensure data accuracy, data will be doubled entered by independent research team members, when not collected directly via REDCap.

Study flow. The study flow is articulated in the 5 steps. In step 1 participants will be recruited through our extensive network of hospitals and clinical centers who provide diagnostic evaluations for autism, who will show study flyers in their waiting rooms and websites and inform families enrolled in their waitlist about the opportunity to participate. Interested caregivers will complete a phone screen to learn more about the study and to confirm eligibility.

In step 2 caregivers who are invited and consented to the RCT will complete baseline assessments prior to randomization through an online procedure that includes completing online forms and phone interviews conducted by a clinician blind to group allocation and study hypotheses. Assessment measures will include standardized measures indexing broad adaptive functioning and severity of autism symptoms, as well as an idiographic measure capturing goals specific for each dyad. During the baseline assessment, a detailed description of the specific goals that each participant family wants to achieve with their child (e.g., addressing difficulties with verbal communication, toilet training, food refusal) will be obtained using Goal Attainment Scaling procedures. As part of the process, parents will be also asked to provide 3 to 5 "baseline goal" vid

• Vineland Scales of Adaptive Behavior-3 [Срок оценки: Within 30 days from consent]
• Parenting Stress Index-4, Short-Form [Срок оценки: Within 30 days from consent]
• Autism Impact Measure (AIM) [Срок оценки: Within 30 days from consent]