Enhancing stakeholder roles in autism early interventions in the United States: A stakeholder-driven research agenda

Abstract

This article outlines a stakeholder-created research agenda to guide future early intervention research for autistic children. We collaborated with 10 autism service providers, 10 parents of individuals with autism, and 10 autistic people across a total of 18 small group and 2 inter-group meetings occurring over 2 years. Together, we synthesized results from (1) these stakeholder workshops and (2) a survey completed by 237 autistic adults, autism providers, caregivers, and autism researchers in the United States. The finalized research agenda includes (1) Guiding Principles, (2) Research Priorities, and (3) Systems Implications. The full version of the early intervention autism research agenda is available in Supplemental Material. In this article, we summarize the main points of the research agenda and discuss unique themes highlighted by stakeholders in considering early autism intervention research. Finally, we highlight the need to include stakeholders in decision-making and consultant positions throughout the research process to align future work most directly and optimally with the goals and needs of the autism community. We have gathered guidance directly from our stakeholders and experiences with Project Stakeholders Engaging in Early Intervention Research (STEER) into a researcher workbook which we hope may facilitate these efforts. This workbook is available in Supplemental Material.

Lay abstract

In this article, we outline a stakeholder-driven research agenda to guide future early intervention research for children with autism. Our research team collaborated with autism service providers, parents of individuals with autism, and autistic people to create this research agenda by (1) conducting workshops with community members and (2) distributing a survey to a larger number of community members around the country. The finalized research agenda includes (1) Guiding Principles for current and future research, (2) Research Priorities focused on early intervention for individuals with autism, and (3) Systems Implications to consider in future clinical, research, and policy efforts for early intervention. The full version of the research agenda is available in Supplemental Material. This article lists the main points of the research agenda and discusses unique themes highlighted by the community members. One main conclusion is that researchers need to include community members in decision-making and consultant positions throughout the research process to best meet the needs of the broader autism community. We have created a researcher workbook which we hope may facilitate these community consultation efforts. This workbook is available in Supplemental Material.

Keywords

early intervention neurodiversity research agenda stakeholder consultation

Research regarding early intervention for autistic¹ children is of high priority for investigators, funders, and stakeholders.² Indeed, the first years of life may represent a critical period, with intervention during this time having the strongest impact on brain development, behavior, and long-term outcomes (Webb et al., 2014). Early intervention also represents a substantial investment, both from a clinical and research perspective. In 2018, over US$52 million of public and private funding was devoted to more than 260 autism intervention research projects across the United States (Interagency Autism Coordinating Committee (IACC), 2021). Clinically, the costs of early intervention are even greater—by one estimate, applied behavior analysis (ABA) treatment programs alone constitute a US$1.8 billion annual market in the United States (LaRosa, 2018). Despite the high demand for and utilization of early autism intervention services, autistic adults and other autism community stakeholders have expressed concern about research and practice in this area. Stakeholders express that available services do not fully meet their needs. They are also concerned that much of the research being conducted regarding early autism intervention is inconsistent with their goals, values, and priorities (Gardiner, 2017; McGill & Robinson, 2020; Pellicano et al., 2014). Most notably, some people with autism have raised major concerns about highly researched standard-of-care early intervention programs, such as the potential for intensive behavioral intervention to lead to trauma and increased vulnerability for autistic people (Kupferstein, 2018; McGill & Robinson, 2020). While these concerns have been strongly refuted by some caregivers, autism researchers, and practitioners (e.g. Gorycki et al., 2020; Leaf et al., 2018), others have responded by acknowledging gaps in current research and practice and recognizing the need to involve autistic stakeholders more closely in autism early intervention research (e.g. Bottema-Beutel et al., 2021; Dawson et al., 2022; Leadbitter et al., 2021; Schuck et al., 2022).

Throughout the last decade, there have been growing calls to increase patient involvement in research on physical, mental, and behavioral health and development, including in research agenda setting (Liberati, 2011; Odgers et al., 2018; Richards et al., 2013). Proponents of enhanced stakeholder involvement in research argue that stakeholders are best equipped to understand their own needs, and that stakeholder-guided research is likely to be more relevant, impactful, and equitable. Despite the growing popularity of stakeholder-informed research agendas and other participatory approaches, members of the intellectual and developmental disability (IDD) community (such as autistic people and individuals with intellectual disability) risk being excluded from such efforts. However, with the right supports, individuals with autism and/or other IDDs can understand clinical research goals and methods (Robotham et al., 2011) and contribute to discussions related to Research Priorities (Pellicano et al., 2014). Moreover, people with autism and/or other IDDs both value and expect direct and honest communication with the research community (McDonald et al., 2016). Autistic individuals are also parts of families, communities, and systems of care; therefore, voices from caregivers, clinicians, educators, and administrators offer additional critical perspectives.

In recent years, several notable efforts have been made to engage the autism community in shaping the direction of autism research. For example, the IACC, Department of Defense, Patient-Centered Outcomes Research Institute (PCORI), and others include community members on committees and funding review panels (although they typically form a minority of constituents and the power they hold relative to scientific members is not always clear). Concurrently, research teams have engaged stakeholders to understand the extent to which current research and practice aligns with the values, needs, and preferences of the autism community, including autistic adults (Habayeb et al., 2022; Kuo et al., 2018; Snell-Rood et al., 2020). A few groups have gone even farther, using stakeholder voices to identify Research Priorities (Benevides et al., 2020; Parsons et al., 2020; Pellicano et al., 2014; Shattuck et al., 2018). For instance, Shattuck and colleagues (2018) integrated findings from literature reviews, interviews, meetings, and a Delphi survey with community and agency stakeholders to identify broad priorities for research on the transition to adulthood in autistic people. Indeed, as of 2021, a total of seven published articles focused on articulating Research Priorities of key stakeholders in the autism community (Roche et al., 2021), with several additional articles published since the review such as Benevides and colleagues (2020) work which engaged stakeholders, including autistic adults, parents, representatives from community organizations, and researchers in an online survey, stakeholder meetings, and focus groups to co-produce a list of priorities to guide mental health research and practice in autism.

These efforts have been crucial in articulating both general and specific priorities across several areas of autism research. Across outputs, early identification and early intervention have been noted as an important area of need. Accordingly, several groups have worked to consider key priorities in the early identification and intervention space specifically. The Autism CRC recently published Australian National Guidelines for supporting learning, participation, and well-being of autistic children and their families (Trembath et al., 2022). This work represents an impressive effort to synthesize systematic input from autistic individuals, family members of individuals with autism, clinical practitioners, and researchers to create practice guidelines for autistic children from birth to 12 years of age. While this report offers critical clinical recommendations, it does not explicitly focus on stakeholder-identified Research Priorities to specifically guide investigators and funders to advance the science of autism research.

With the specific goal of informing the direction of autism early identification and early intervention research, Grzadzinski et al. (2021) published research recommendations regarding pre-symptomatic young children who go on to be diagnosed with autism; these recommendations were created by a group of experts. Early intervention was also the focus of a recent Special Interest Group at a major autism conference (International Society for Autism Research (INSAR)), resulting in a set of recommendations for the future of early autism intervention research, again created collectively by this group of research professionals (Vivanti et al., 2018). While these research agenda-setting projects represent important initial steps in the autism early intervention research space, the lack of involvement of stakeholders beyond researchers and clinical experts represents a critical gap that can be addressed by engaging with more diverse stakeholder participant groups. To our knowledge, only one published article has described the process for engaging with stakeholders specifically around the goals of early identification and early intervention research in the field of autism (Fletcher-Watson et al., 2017). These investigators partnered with diverse stakeholders (autistic adults, parents of children with autism, healthcare practitioners, and educational practitioners) across 11 countries in Europe to design and then distribute a survey related to attitudes toward and goals of early identification and early intervention research, finding that the overall areas of highest importance to the community were related to describing the earliest signs of autism and understanding the genetic basis of autism spectrum disorder. Although stakeholders contributed broadly to the development of the survey questions, the final list of Research Priorities was generated by researchers and then vetted by a handful of autistic adults (n = 2), practitioners (n = 3), and parents (n = 3) in the United Kingdom. While this work offers a strong foundation for understanding the perspectives of a variety of stakeholders with respect to autism early intervention research, it is unclear as to whether these priorities would be the same for stakeholders outside of Europe, including in the United States. Further, opportunities to expand on this work include taking a more explicit co-construction approach to identifying Research Priorities key stakeholders, as well as designing and disseminating a formal research agenda that can be used to communicate findings to the autism and research communities at large.

Thus, the goal of this work, Project Stakeholders Engaging in Early Intervention Research (STEER), was to construct and disseminate a stakeholder-driven research agenda, co-created by individuals within the autism community (autistic adults, parents, and community providers serving young children with autism) in the United States. Thirty community stakeholders and four research team members worked collaboratively over a period of 2 years to create and refine this agenda. While this agenda-setting exercise was facilitated by a research team, the content of the research agenda was created primarily by the stakeholder members. We use the reporting guideline for priority setting of health research (REPRISE) to describe our agenda-setting process (Tong et al., 2019).

Methods

Context and scope

The aim of Project STEER was to engage stakeholders as active collaborators in generating Research Priorities for early autism intervention research. The long-term goal of this work was to guide investigators, funders, and policymakers regarding important directions for future research. This work was conducted within two US cities, and likely best represents stakeholder perspectives within US systems of care; however, we expect that a substantial subset of the priorities are likely to be relevant beyond the United States.

Governance and team

Project STEER was initiated by two autism early intervention researchers (K.M.W. and A.L.W.) and two research coordinators (A.R.B. and R.A.G.) based out of autism and developmental disabilities centers. This research team was responsible for developing and guiding the priority setting process, synthesizing qualitative and quantitative data, and developing drafts of outputs for feedback from the stakeholder team.

Framework for priority setting

This project’s Research Priority setting approach was informed by Community-Based Participatory Research methods; more details about specific meeting activities are provided below.

Selection of stakeholders and group characteristics

Stakeholders were recruited in Columbus, Ohio, and Chicago, Illinois, through advertisements in email newsletters, Facebook posts, outreach to community partners, and word of mouth. Interested applicants were invited to complete a brief electronic survey describing their interests, careers, experience working and/or living with autistic people and/or with self-advocacy, as well as their motivation for and logistical ability to participate in this consultation work.

A total of 91 individuals submitted a complete application (15 autistic, 45 parents, and 31 professionals); 79 were invited for interviews and 70 completed phone interviews. Stakeholders were not chosen for an interview if they did not complete the full application, indicated not being able to attend all the stakeholder meetings, or did not indicate a professional background in early intervention (for provider group). After interviews were held, the Project STEER facilitation team selected final members of the group. Individual experiences, background, excitement for the project, and ability to commit to the timeline of the engagement project were all considered. Stakeholder teams were broken into six sub-groups, with each regional team containing five autistic self-advocates, five community-based providers (e.g. clinicians, educators) or policymakers, and five parents of children with autism. At the time of application, stakeholders were asked to select a single group with which to affiliate; however, there were some stakeholders with intersecting identities (e.g. a parent of an autistic child who is also an educator, multiple autistic individuals who also work at early childhood centers that serve autistic children).

Given the limited number of spaces on the stakeholder teams, we recognize that we were unable to capture the full diversity of existing stakeholder experiences on our teams, and that the application process may have missed a subset of individuals (e.g. those who do not access/use the Internet, individuals with low literacy). The application did include several questions about identity and experiences, and we intentionally selected participants with a range of backgrounds and life experiences. Because of our commitment to treating our stakeholder team members as research partners rather than research subjects, we did not seek permission for the individual demographic information collected during the application process to be reported publicly; however, brief descriptions of the range of relevant identity features and experiences disclosed by stakeholders are included below.

Autistic stakeholders

Among autistic stakeholders, five identified as men and five as women. All were young adults (under the age of 40 years). Half identified as non-White (Black or Asian). All had completed high school, and several had completed college. Several received an autism diagnosis in adolescence or adulthood, while others were diagnosed in early childhood. They disclosed a range of co-occurring conditions including mental health challenges and intellectual disability. Several had current or prior experiences as clinicians or educators serving autistic children. All autistic team members used speech as their primary mode of communication. They had a range of past advocacy experience (i.e. some were new to advocacy, while others had engaged in autism or disability advocacy groups at a local, state, or national level). All autistic adults were engaged in vocational training, post-secondary education programs, or employed.

Parents of autistic children

Parents of children with autism were nine biological and one adoptive mother of children between early childhood and young adulthood. Four of the 10 mothers identified as either Black or Hispanic. About 75% of parents had sons, and their children had a range of verbal and cognitive abilities, genetic diagnoses, medical and psychological conditions, and educational environments. Two participating parents had multiple children diagnosed with autism. Several parents identified as neurodivergent but none reported formal diagnoses of autism spectrum disorder.

Community providers

Project STEER included a range in types of community clinicians and educators. These included two speech–language pathologists, one occupational therapist, three special education teachers, a psychologist, an autism school program director, and a healthcare system–based social worker. One clinician discontinued participation partway through the project due to competing time demands. Community providers reported a wide range in years of experience (from less than 5 to over 20 years). Nine of the 10 providers were female and all but one identified as White and non-Hispanic.

Meeting format

Stakeholders met in person (prior to the start of the COVID-19 pandemic) or via Zoom in small stakeholder-specific groups (four to five participants) at each location. Three small group meetings focused on research agenda creation and were conducted in homogeneous groups with regard to stakeholder type (i.e. autistic people, parents, providers) to increase the comfort level of participants in sharing their individualized experiences. For example, many parents expressed great frustration with service systems and providers—something they may have felt less comfortable speaking about in a group that included providers.

Each meeting functioned as an interactive workshop, lasting about 2 h with designated time for topic review, two to four focused activities, questions, and breaks throughout the meeting time. If a stakeholder missed a group meeting, they participated in an individual phone call with a project coordinator to contribute to meeting content and provide project feedback. Activities were designed to engage stakeholders through different modalities and communication styles (e.g. semi-structured large group discussions, written comments on sticky notes or flashcards, email and text communication, and online surveys for in-between meeting activities). Activity materials were provided ahead of the meeting for stakeholders who wished to take additional time to prepare for the activities (although advance preparation was not required). Some individuals who indicated the need for additional participation support also participated in individually arranged pre-group phone calls with a research staff member to explain materials, answer questions, and assist them in preparing their thoughts for the group meetings. Throughout each meeting, notes were taken via poster board or screen-shared documents, and then synthesized across all groups and disseminated back to all project members to encourage sharing of ideas and cohesion across members. See Table 1 for full description of meeting topics, activities, and outputs.

Table 1.

Project meeting topic, activities, and outputs utilized in research agenda development and finalization.

Meeting format and number	Date	Topic	Activities	Outputs
Small Group Meeting 1	August to September 2019	Orientation	● Project team presentation: project goals and structure. ● Individual introductions. ● Individual brainstorming and group discussion: defining stakeholders, engagement, and early intervention. Qualities of a good group member and communicator. ● Group discussion: myths and misunderstandings about autism, early intervention, and research.	● Stakeholder contact and project expectations established. ● Definitions of stakeholder engagement. ● Meeting notes shared with all project members.
Small Group Meeting 2	October to November 2019	Development of Research Agenda Ideas	● Individual activity and group discussion: identifying individual values and goals for change in the autism community. ● Group activity: information, resources, and key players needed to achieve goals for change. ● Individual activity: generating Research Priorities.	● Identified values and goals for the autism community. ● 150 priorities for ASD research. Meeting notes shared with all project members. ● After-meeting survey: ranking of 29 Research Priorities (Table 2)
CCC Meeting 1 (Virtual)	December 2019	CCC Orientation	● Individual introductions. ● Group discussion: insights from individual meetings. ● Project team presentation: research questions and research agendas.	● CCC inter-group contact established. ● Meeting notes shared with all project members.
Small Group Meeting 3	January 2020	Refine and Revisit Research Agenda	● Review of 15 top ranked Research Priorities. ● Individual activity shared with group: assessing alignment of priorities with core values, impact on autism community, and categorization of priority type (overarching core value, Research Priority, policy priority). ● Individual activity: rank top and bottom 5 priorities.	● Consolidated final list of 23 agenda items. ● Establishment of Guiding Principles and Systems Implications. ● Synthesized meeting notes shared with all project members.
CCC Meeting 2	February 2020	Research Agenda Finalization: Drafting and Formulation	● Small group activity and presentations: discussion of Guiding Principle importance to the autism community and drafting of descriptive statements. ● Small group activity and presentations: discussion of Research Priority importance, impact on the autism community, and example research questions. ● Full group activity: drafting and rephrasing of Research Priorities and explanatory statements. ● Full group discussion and activity: Systems Implications priorities, connections to core values, and areas for policy reform. Drafting and rephrasing.	● Draft of 23 research agenda items, descriptions, and example research questions. ● After-meeting survey to all project members: feedback on wording, clarity, and accuracy of 23 draft research agenda items. ● Meeting notes shared with all project members.

ASD: autism spectrum disorder; CCC: Cross-City Collaboration.

Each small group meeting timepoint included six individual meetings of five stakeholders per regional team (parents of individuals with autism, autistic self-advocates, and clinicians or policymakers), facilitated by two project leaders per region. CCC meetings included one representative from each of the six regional teams (six stakeholders) and were facilitated by all four project leaders.

To allow for sharing of ideas across stakeholder types and assist in coming to consensus across groups, a subset of stakeholders had the opportunity to participate in inter-group meetings, named the Cross-City Collaboration (CCC), composed of one representative from each of the six stakeholder groups. Those who were interested in the CCC volunteered and were randomly selected to participate. The CCC then met several times throughout the research agenda creation process, once via a full-day conference in Chicago and once via Zoom meeting.

Group orientation

During their first small group meetings, stakeholders were oriented to the project goals and structure. Members individually brainstormed and then collectively shared their ideas regarding stakeholder engagement and early intervention. A group discussion regarding myths and misunderstandings about autism, research, and early intervention concluded this meeting. This activity highlighted perceived challenges in the research-to-practice gap by identifying barriers between research aims and community member experiences. This meeting also provided project members with background knowledge about research, medical versus social models of disability, and early autism intervention.

Identification and collection of Research Priorities

After this orientation meeting, initial research agenda priorities were solicited during a second round of small group meetings. During Meeting 2, project members first individually reflected on their own values and goals using a guided individual activity. A group discussion then helped stakeholders draw connections between these individual experiences to collectively identify core values central to their role in the autism community. Group members independently journaled about goals for change in the autism community, and then collectively generated a vision for the future of the autism community via group discussion. These values and visions were used to create a list of goals covering a range of topics including increased services and intervention, professional development, media representation, and daily community support. For each identified goal, the group identified perceived knowledge gaps, resources, and “key players/systems,” written on post-it notes and visually presented on poster boards representing respective goals throughout the meeting room. At the end of this activity, stakeholders privately and individually generated the top five topics they believed were most important to be studied in autism early intervention research and wrote them on a note card. This resulted in 150 individual topic ideas.

Prioritization of research topics

The project team then categorized these topic ideas into a list of 28 broad priority areas (as there was a substantial overlap among the 150 written individual topics). In an online survey, stakeholders selected and rank-ordered their Top 10 items of importance for early intervention research. The 15 most frequently endorsed priorities among each stakeholder group (autistic adults, parents, or providers) were distributed back to the groups in advance of the next meeting. In cases where there were “ties” for number of endorsements, we used rank order to determine which were included in the Top 15. See Table 2 for a full list of preliminary priorities appearing in each group’s Top 15 and the number of individuals ranking each of these priorities in their individual Top 10.

Table 2.

Stakeholder prioritization of research topics.

	Topic statement	Number of Top 10 rankings			Final item placement in agenda
	Topic statement	Autistic adults (n = 10)	Parents (n = 7)	Professionals (n = 10)	Final item placement in agenda
1	Improving and standardizing diagnostic services to increase quality and reduce misdiagnosis.	6^a	4^a	5^a	Research Priority #5
2	Increasing access to existing resources through improved service navigation, linking, and/or care coordination.	4^a	5^a	6^a	Systems Needs
3	Increasing access to services and supports across the lifespan.	7^a	3^a	4^a	Guiding Principle #6; Systems Needs
4	Increasing treatment access and service availability for all families through changes in policy, financial constraints, wait lists, etc.	4^a	5^a	8^a	Guiding Principle #2; Systems Needs
5	Understanding how to best teach communication skills to children with autism.	3^a	4^a	6^a	Research Priority #2
6	Considering autistic culture, voices, preferences, and goals in the development and evaluation of treatment models and methods.	4^a	3^a	3	Guiding Principle #1; Systems Needs
7	Understanding how cultural differences impact diagnosis and treatment, including research on racial/ethnic minorities, women/girls, children who were adopted, and those from multiple minority groups (intersectionality).	5^a	3^a	0	Guiding Principle #4; Research Priority #7
8	Better understanding the impact of medical comorbidities, such as GI issues, sleep, immune dysregulation, genetics, etc. on autism symptoms and behaviors.	6^a	2	6^a	Research Priority #6
9	Developing and testing individualized interventions to better understand how to select and modify treatments.	4^a	1	5^a	Research Priority #10
10	Improving communication between treatment providers, families, and autistic individuals regarding goal setting, intervention needs, and progress monitoring.	6^a	2	6^a	Guiding Principle #3; Research Priority #11
11	Daily life support, including crisis support.	5^a	1	3	Research Priority #4 (Mental Health)
12	Enhancing family quality of life.	4^a	2	4	Items 12, 13, and 14 Integrated with #31 Below (Well-Being)
13	Increasing social and friendship opportunities for children with autism and their families.	4^a	1	3
14	Peer support, such as mentors or support groups.	5^a	1	0
15	Reducing stigma and building community awareness of autism.	4^a	1	1	Guiding Principle #7
16	Development/testing of interventions for improving emotion regulation in children with ASD.	1	4^a	6^a	Research Priority #3
17	Early identification and diagnosis via education, resources, and training for parents or providers.	1	5^a	7^a	Research Priority #5
18	Establishing a “standard of care” or “gold standard” for early autism intervention.	3	5^a	6^a	Not included
19	Providing more effective training for educators and other stakeholders.	2	3^a	7^a	Research Priority #1
20	Understanding how “key players” on the child’s treatment team (e.g. physicians, educators, and therapists) can most effectively collaborate and how this impacts outcomes.	2	4^a	4^a	Guiding Principle #3; Research Priority #11
21	Understanding how to best prepare autistic children for school/group settings.	3	4^a	4^a	Not included
22	Understanding how to provide the most effective support and education for parents and caregivers, especially immediately following diagnosis.	3	4^a	4^a	Research Priority #5
23	Research on service delivery models, including appropriate intensity and frequency of services and integration of services into community settings.	2	3^a	3	Integrated with Research Priority #10
24	Providing treatment and support for developmental delays.	3	2	3	Not included
25	Access to/understanding of how more individualized supports (smaller groups, 1:1 support) impacts treatment outcomes.	2	2	1	Not included
26	Increased understanding of resiliency factors and the impact of strengths-based approaches.	2	1	1	Not included
27	Understanding the impact of general and specific trauma on both short- and long-term outcomes for children with autism.	3	0	0	Integrated with Research Priority #8, #4
28	Conducting high-quality research on interventions that are available in the community, but not yet evidence-based.	2	1	3	Integrated Across Intervention-Related Research Priorities
29	How can early intervention research be made more accessible to stakeholders?	Emerged from discussions in subsequent group meetings			Guiding Principle #5; Research Priority #9
30	What are the potential harms of specific early intervention techniques or programs?	Emerged from discussions in subsequent group meetings			Research Priority #8
31	How can we improve overall well-being for individuals with autism?	Emerged from discussions in subsequent group meetings			Research Priority #12

ASD: autism spectrum disorder; GI: gastrointestinal.

Inclusion in group’s Top 15.

The Top 15 items for each group had substantial inter-group overlap, but also some clear differences; a total of 23 different priorities were represented among the Top 15 of the different groups. Five items were included in the Top 15 for all three groups; these included items related to improving diagnostic services, increasing access to existing resources, access to services and supports across the lifespan, policy changes that would increase service access for all families, and teaching communication to children with autism. Two items were shared between autistic adults and parents, but not providers; three items were shared by autistic adults and providers, but not parents; and seven items were endorsed by parents and providers, but not adults with autism. Five items appeared in the Top 15 for the autism group only, and one item appeared in the Top 15 for the parent group only.

Review of these priorities during Meeting 3 revealed that while many of the priorities were related to specific research questions, others were more oriented toward guiding the research/stakeholder process (referred to as Guiding Principles) or goals for larger policy and systems (referred to as Systems Implications). Thus, stakeholders individually categorized these priorities into Guiding Principles, Research Priorities, or Systems Implications, and estimated the impact of each priority on the broader autism community via a reflective journaling activity. Project members shared these individual reflections with each other to elicit additional considerations and gauge how the priority reflected goals and values of the group. At the end of the meeting, stakeholders again ranked their top five priorities and identified five priorities that they viewed as least important to early intervention research. These ranked priorities, in conjunction with ideas synthesized from group discussions, were consolidated by the project team into a final list of 23 items broken down into three categories (Guiding Principles, Research Priorities, and Systems Implications). See Table 2 for final placement of priority items in agenda.

The 23 items were discussed in detail during the daylong, in-person CCC meeting attended by six stakeholder representatives and facilitated by the four research team members. Small and large group discussions focused on (1) the potential impact of a given item on the autism community, (2) developing explanatory statements for each item, and (3) refining wording used in the items. Small groups comprised one member of each stakeholder type first discussing the importance and impact of Guiding Principles, drafting descriptive statements, and presenting them to the larger group. Next, project members formed new small groups to discuss the topics further and identify example research questions for further study. Finally, the full team worked collaboratively to draft priority item statements and descriptions using a projector. This activity was done to directly and intentionally capture the words of project members in real time, rather than deferring to research team synthesis or interpretation. The meeting concluded with a full group discussion of Systems Implications and ideas for policy-wide changes, again. Group members agreed that Systems Implications were best explained in a narrative written format, rather split into separate sub-points, due to the interconnected nature of policy and system-wide challenges for early intervention.

Output

After completing the CCC-drafted edits to the agenda items, the project team made additional edits to clarify language, eliminate redundancy, and incorporate information about extant research in the priority area. All project members then completed a survey to rate the clarity and accuracy of items via a Likert-type scale and were invited to provide suggestions for editing agenda items via a text box. With this feedback, the Project STEER research team developed a draft version of the research agenda. See Supplemental Material for the full research agenda.

Evaluation and feedback

As a final step, the draft agenda was distributed via an online survey, which was distributed across the United States. The primary purpose of this survey was to “cross-check” the priorities developed by our stakeholder teams with the opinions of a larger, more geographically diverse group to ensure that the identified priorities were also endorsed by the broader US autism community. This survey research was approved by the Ohio State University Institutional Review Board (IRB). Survey participants were identified through (1) the ResearchMatch database, (2) autism, autistic community, and clinician/provider organizations serving the United States, and (3) targeted lists of researchers for direct contact. ResearchMatch is a US-based recruitment database funded by the National Institutes of Health (NIH) that has a wide geographic and demographic spread. Recruitment emails were sent through ResearchMatch in multiple batches of 1500 interested participants per batch with a demographic spread across the United States. Autistic individuals and parents/caregivers were recruited by sending recruitment announcements to various autistic and autism community self-advocacy networks with broad reach across the United States (e.g. Autistic Self Advocacy Network, Multicultural Autism Action Network, Parent to Parent USA, and Family Voices). Autism providers were recruited through online networks, with a focus on US-wide rather than local or statewide networks (e.g. Division for Early Childhood, American Speech–Language–Hearing Association, National Association of Special Education Teachers, American Academy of Pediatrics, and many others). Autism researchers were recruited by compiling a targeted list of early intervention researchers through autism research networks such as the INSAR and NIH contact lists; recruitment emails were sent directly to these researchers.

The survey started with a brief demographic questionnaire followed by a description of Project STEER’s research agenda. Participants were asked to identify as an autistic person, parent of a child with autism, early intervention autism provider, or autism researcher. Participants could select multiple categories to account for intersecting identities. After this introductory material, each agenda item and description was listed individually, followed by four questions: (1) Would answering this research question positively impact individuals with autism, their families, and the autism community? (2) Does this research question address a gap or need in current research or clinical practice? (3) Does this research question reflect my values as a member of the autism community? and (4) Is conducting research in this area a priority? Respondents answered similar questions for the Guiding Principles and for a paragraph synthesizing Systems Implications. Respondents were given the opportunity to provide qualitative feedback after each item.

A total of 429 survey responses were collected. Of these, 192 were excluded from the final analysis due to (1) short duration for survey completion (less than 7 min, when the survey took an average of 28 min to complete) (n = 41), (2) partial completion of survey (less than 76% complete) (n = 70), and (3) duplicate responses (multiple surveys were either started at the exact same time or reported exact same responses in open text boxes) (n = 81). Final reported data are from 237 respondents, including 88 adults with autism, 63 autism providers, 91 parents of individuals with autism, and 35 autism researchers (30 participants identified with multiple groups; see Table S1). Autistic respondents were disproportionately male (64%), while respondents from other categories were more likely to be female (parents 65%; providers 84%; and researchers 74%). The majority of respondents across groups identified as White (autistic = 56%; parents = 74%; providers = 89%; and researchers = 86%), with the second most frequent racial identification being Black or African American (autistic = 23%; parents = 11%; providers = 3%; and researchers = 11%). About one-third (37%) of autistic participants identified as Hispanic, with lower percentages of Hispanic participants in the other groups (parents = 12%; providers = 8%; and researchers = 15%). Participants were incentivized through a randomized drawing to receive 1 of 106 US$20 gift cards. See Table S2 in Supplemental Material for additional demographic information from the survey respondents.

Following the survey, items in the research agenda were revised by the project team one final time to address and integrate ideas and suggestions provided by survey respondents in their open-ended responses. In accordance with initial goals to increase the accessibility of research for stakeholder communities, Project STEER developed a community-facing version of this research agenda, in addition to this article. The community version is included as Supplemental Material. Additional information about the project can be accessed at https://nisonger.osu.edu/project-steer/.

Funding and conflict of interest

This project was funded by a PCORI Engagement Award to the first and senior authors. The first and senior author are both trained and licensed as clinical psychologists. They have both been trained to implement Naturalistic Developmental Behavioral Intervention (NDBI) models in their clinical practice, and both have received consulting fees for conducting workshops regarding implementation of specific NDBI programs (Reciprocal Imitation Training and Project ImPACT). The second author has also participated in implementation of Reciprocal Imitation Training in a research setting, supervised by the first author. The first author oversees provision of the Project ImPACT program in a clinical setting. The third author was previously trained in and delivered ABA therapy to individuals with autism. All four authors approach their work in part from a neurodiversity framework, honoring and supporting diverse thinking, communication, and behaviors of autistic people as valuable and not necessarily in need of change (although have also been trained in more medical conceptualizations of autism), and infused this perspective into this project (e.g. providing information about the neurodiversity model to stakeholders during orientation). Stakeholder members approached their work from a variety of different perspectives and frameworks. Clinical perspectives and training of providers included speech–language pathology, special education, occupational therapy, and ABA therapy. Stakeholders were compensated US$50 per meeting for their participation and were reimbursed for travel. Additional compensation of US$100 and travel reimbursement was provided to the six CCC team members who traveled for the CCC meeting.

Community involvement statement

Community members co-created all elements of this project.

Results

Research agenda

Project STEER stakeholders felt strongly that it was important to establish shared values and goals at the forefront of the research agenda. Thus, Guiding Principles serve as a keystone to ground recommendations for future early intervention research within the values of the autism stakeholder community. The Research Priorities section includes 12 broad research questions that stakeholders identified as important for guiding the direction of future autism early intervention research. Finally, Systems Implications represent a set of considerations for continued efforts at the policy and systemic level, to ensure future early autism intervention research has direct and lasting impacts. As one STEER stakeholder explained, “more, higher quality research is not enough.” Guiding Principles and research questions identified by the Project STEER team are included in Table 3 and the full research agenda is available in Supplemental Material.

Table 3.

Project STEER Guiding Principles and Research Priorities.

Guiding Principles
1. It is necessary to consider autistic culture, voices, preferences, and goals in all activities related to the autism community.
2. Everyone has a right to a range of high-quality supports and services that are culturally and financially accessible.
3. Communication and collaboration among families, people with autism, and service providers is an essential component of effective care.
4. People with autism come from many different backgrounds; we must work to build mutual understanding and respect for autistic people who identify with a range of cultural, family, and community backgrounds.
5. Stakeholders should have access to current, accurate, and understandable information about autism research.
6. Early intervention must take a lifespan perspective.
7. Community members and environments must be willing to change to be more supportive of people with diverse needs.
Research Priorities
1. How do we best train professionals and community members who deliver services, care, support, and education for young children with autism?
2. How can we ensure that all autistic people are able to effectively communicate their wants, needs, goals, and preferences in their daily lives?
3. How can we support the development of effective coping skills, emotional understanding, and emotional expression in young children with autism?
4. How can we support mental health in autistic individuals, starting early in development?
5. How do we ensure autistic children receive accurate and timely diagnoses, and support families through the diagnostic process?
6. How can we best provide proper interventions for co-occurring medical problems in autistic people?
7. How can we better understand the intersection of autism with other social and cultural identities a person or family may hold?
8. What are the potential harms of specific early intervention techniques or programs?
9. How can early intervention research be made more accessible to stakeholders?
10. How can we make early interventions individualized to better assist each person with autism?
11. What are the most effective strategies for improving coordination among medical, therapeutic, and educational service systems that provide care for young autistic children?
12. How can we improve overall well-being for individuals with autism?

Survey results

The Guiding Principles, Research Priorities, and Systems Implications were each rated on a 5-point scale from “Strongly Disagree” to “Strongly Agree” on several items (e.g. positive impact on the autism community, reflective of community values, important, reflective of current gaps/needs). For Guiding Principles, proportion agreement (rating of “Agree” or “Strongly Agree”) ranged from 84.39% to 93.25% when all groups’ responses were combined. For Research Priorities, agreement ranged from 81.86% to 92.83% when all groups’ responses were combined. For Systems Implications, respondents largely agreed that these considerations addressed needs in the current systems of autism care (percent agreement = 87.80% for autistic adults; 89.89% for parents; 89.47% for providers; and 86.67% for researchers) and would positively impact members of the autism community (percent agreement = 85.37% for autistic adults; 93.26% for parents; 91.38% for providers; and 96.77% for researchers). See Supplemental Tables S3 and S4 for full survey results broken down by item and respondent group.

Discussion

The Project STEER research agenda represents a critical step forward in aligning stakeholder goals and values with early autism intervention research. The project yielded several clear takeaways regarding future research and initiatives for autism. However, of note, the process to reach this agenda was not linear, and the perspectives of involved stakeholders were by no means monolithic. Several takeaways, challenges, limitations, and future directions of this work are described below.

Elevation of autistic voices

Ultimately, Project STEER demonstrates the critical importance of elevating and prioritizing stakeholder voices in generating change. In particular, Guiding Principle #1 highlights the need to appoint autistic people in decision-making positions, with the goal of placing their voices and preferences at the forefront of future research initiatives. This includes supporting the careers of autistic researchers, placing autistic community members in decision-making positions, and engaging autistic people as well as other members of the autism community as collaborators and consultants in a larger number of research studies. Moreover, consistent with neurodiversity conceptualizations of autism (i.e. autism as a set of neurodevelopmental differences that should be celebrated and supported rather than “cured”), our stakeholders identified the need for communities, and society at large, to be willing to accept some of the burden of change to be more accepting of autistic people. In many ways, these recommendations are unsurprising. They dovetail with findings from multiple other autism-related research agendas published in the last 10 years (Pellicano et al., 2014; Shattuck et al., 2018). In particular, Shattuck et al. (2018) identify increasing involvement of people on the spectrum as an ethical responsibility. These Guiding Principles also align with efforts from prominent autism research organizations such as INSAR, which has recently launched efforts to facilitate stakeholder-researcher consultation, as well as created an Autistic Researchers Committee.

Yet, despite the clear alignment of these principles with other recent work in the autism field, it took time, sharing, and effort for this mutual understanding to develop across our stakeholder groups. Interestingly, the Guiding Principle regarding involvement of autistic voices was not initially included in the Top 15 ranked priorities for our group of professionals, although it emerged as a strong priority in both the parent and autistic groups. Moreover, parent participants entered the project with varying degrees of exposure to neurodiversity ideas and to autistic adults; some already fully embraced neurodiversity perspectives with regard to their young children and were strongly linked to autistic self-advocate communities. Others had never met an autistic adult and had their first experience speaking with autistic adults at our inter-group social event held early in the project. By the time that the research agenda was finalized, there was strong inter-group agreement that the first Guiding Principle should be the necessity of autistic voices in research spaces. Interestingly, while our parent members embraced and found value in pushing community members to become more accepting of autistic people, they also expressed feeling a tension between holding communities accountable for this change and having a responsibility to best prepare their children for the world they currently live in (which may be less accepting than is desirable), rather than the world the parents wished they lived in.

It is worth noting that, despite the focus of the Project STEER research agenda on early childhood—an age group in which the children being directly impacted in real time are clearly not able to engage directly in research consultation efforts—engagement with autistic people was still highly emphasized. The Project STEER experience has demonstrated that engagement with autistic individuals should not be limited to research with particular age groups or topic areas, but rather we should aspire to include autistic voices in all research about autism. Examples of how autistic people might provide valuable insight into early intervention research are also beginning to emerge in the literature; one recent example is a commentary on the fit between a neurodiversity perspective and NBDIs (Schuck et al., 2022).

Group process and consensus-building

As is evident in Table 2, and consistent with prior related efforts (Fletcher-Watson et al., 2017), initial priorities of the different groups diverged considerably. Notably, stakeholders with autism were more likely to hold unique priorities (i.e. not shared by the other stakeholder groups) in comparison with parents and professionals, with 5 of their 15 initial top priorities not being shared by another group. In subsequent discussions, it emerged that many of these unique priorities were related to the idea of positive overall well-being (e.g. support, positive self-concept, reduced stigma, social opportunity). In addition to observed between-group differences, there was also substantial within-group variability with regard to priorities. Each of the initial 28 priorities appeared in the Top 10 of at least one individual. This speaks to the need to include a range of stakeholder voices when considering research direction; it is likely that just one or two stakeholders from a particular group will be unable to adequately represent the range of perspectives present in stakeholder communities.

Finally, it is essential to emphasize that this series of priority setting exercises was a dynamic process. For example, gaining a better understanding of cultural differences in autism was initially among the highest ranked priorities for both autistic individuals and parents, but not a single provider included this in their Top 10. Yet as providers engaged in discussions with parents and people with autism during the CCC meetings, they were strongly supportive of including this priority in multiple places in the agenda and recognized this as a key oversight, resulting in its ultimate inclusion as both a Guiding Principle (#4) and a Research Priority (#7). In another example of consensus-building, both parents and providers initially ranked “Establishing a ‘standard of care’ or ‘gold standard’ for early autism intervention” in their Top 10. However, as discussions of intervention needs unfolded, the groups ultimately concluded that the need for individualized intervention exceeded the need for a single standard of care and chose to emphasize the former in the final outputs. The need to understand which interventions work for whom, when, and under what conditions has been echoed in prior research agendas as well (e.g. Benevides et al., 2020).

The slow consensus-building relied on open-mindedness from all of our participants—including those new to neurodiversity ideas, as well as autistic individuals willing to field questions and comments from parents and professions less familiar with neurodiversity-informed ideas about autistic culture and competency. This was one clear advantage of our longer, multi-meeting process; unlike more workshop-style, single-meeting exercises, the longer process allowed time for personal interactions and rich discussions that ultimately led to greater group understanding and consensus.

Need for a lifespan perspective

Another takeaway from this research agenda-setting process is the need to consider a lifespan perspective within the early intervention space. This is reflected in both Guiding Principle #5 and in several research questions (e.g. #4, #6). STEER members discussed this lifespan perspective as working in multiple directions. First, stakeholders identified the need to better assess and consider long-term outcomes and developmental trajectories in early intervention efforts. In addition, they highlighted that early intervention researchers should be using information known from older children and adults—such as information about elevated rates of certain physical and mental health concerns—to inform efforts to identify and prevent these co-occurring conditions sooner. Despite the fact that mental health is rarely explicitly discussed within early autism intervention research, STEER members emphasized the need to identify and address precursors of mental health concerns during early childhood (Research Question #4), and to help young children develop effective coping and regulation strategies (Research Question #3). This emphasis on taking a lifespan perspective is echoed in Shattuck et al.’s (2018) agenda about transition of autistic youth; their stakeholder teams noted that experiences and support in early childhood “set the stage” for much of what impacts transition success.

Diversity of needs across the autism community

Another area emphasized by stakeholders and represented in both the Guiding Principles (#7) and research questions (#7) is the need to better address and honor the intersection of autism with other social and cultural identities. Many of the Project STEER conversations took place concurrently with larger social justice movements across the country, which spurred even richer conversations about the need for autism early intervention research to consider issues of representativeness and equity. Further, stakeholders emphasized that autism is a key aspect of identity and that future research efforts need to better acknowledge autistic identity, as well as how this identity intersects with other identity features (e.g. race, ethnicity, and gender). Many of these discussions also emphasized the role of service systems (e.g. health care systems and funders) in perpetuating inequities for diverse communities.

Stakeholders also identified a need for research studies that can address multiple levels of impact—including systems, community, and person-level experiences and outcomes. Stakeholders were certainly supportive of research that focused on child-level supports for building particular skill sets—especially in the areas of communication (Research Question #2), coping skills (Research Question #3), and overall well-being (Research Question #12). While researchers have emphasized the need to focus on communication, regulation, and cognitive and behavioral flexibility as pivotal intervention outcomes (e.g. Grzadzinski et al., 2021), there appears to be increasing consensus across stakeholder-engaged outputs such as research agendas (e.g. Benevides et al., 2020) and strategic plans (IACC, 2021) that understanding and maximizing well-being and quality of life for autistic individuals and their families is of paramount importance.

Concurrently, Project STEER members also recognized that findings from such studies would have limited impact without a better understanding of how to effectively disseminate this knowledge (Research Question #9) and train personnel to deliver these programs (Research Question #1). These ideas echo Shattuck and colleagues’ (2018) Research Priorities of increased focus on community and systemic factors that influence outcomes in research on transitions to adulthood in autism. This project similarly affirms the importance of measuring contextual factors beyond person-level outcomes, and increasing research and funding efforts that tackle policy and systemic barriers (Shattuck et al., 2018).

Limitations and future directions

As a truly stakeholder-driven research agenda, this project represents an important step toward better aligning stakeholder goals and needs with early autism intervention research. However, as described above, priorities across the autism community are extremely diverse, and this agenda likely does not represent the goals and needs of all parts of the autism community. While items on the agenda recognize that different members of the autism community have differing needs (e.g. those living in poverty, racial and ethnic minority communities), there was limited representation from these communities in the creation of the research agenda. While our adults with autism and parents were relatively diverse with regard to race and ethnicity (with about half being non-White), our providers were less diverse and the majority of our participants across groups were relatively well-educated and middle to upper class. Several lived in relatively rural areas of Ohio (1–2 h outside Columbus); however, all had the resources required to travel to in-person meetings in major cities. Among our survey respondents, parents and professionals were also predominately White, non-Hispanic, and well-educated. It is clear that specific considerations relevant to individual communities (e.g. immigrant communities, families living in poverty) as well as considerations for autistic children living outside the United States (particularly those living in low- and middle-income countries) are likely to be influenced by unique needs and concerns. Future work that specifically engages members of underserved communities to tailor research questions and needs to their experiences is essential.

In addition, the items on this research agenda represent broad priority topics, rather than clearly defined research questions that can be answered in a single study. Additional collaborative research planning using frameworks that help stakeholders identify isolated and specific research questions that can be answered in a specific study is also needed; one promising framework is the Stakeholder Engagement in Question Development (SEED) method, which guides stakeholders through a planning process specifically designed to frame research questions using the patient, intervention, comparison, outcome, and time (PICOT) framework (Zimmerman & Cook, 2021).

Conclusion

This research agenda is a call to researchers, policymakers, and community members to put action to the words described by Project STEER—to wholeheartedly make the experiences and perspectives of the autism community a top priority when making decisions about early intervention research and supports for autistic children. We urge researchers to consider these Guiding Principles and Research Priorities in their own work, and to develop relationships within their own stakeholder communities that will help to guide, enrich, and enhance the impact of their research. We have gathered guidance directly from our stakeholders and experiences with Project STEER into a researcher workbook which we hope may facilitate these efforts. This workbook is available in Supplemental Material and at www.go.osu.edu/steer_consultation. We recognize a need to continue and extend this work through consideration of other research topic areas, inclusion of a wider variety of autistic perspectives (including non-speaking autistics), and continued input as goals and norms evolve in our communities. We hope that decision-makers will attend to these priorities, and consider how researchers, community members, and policymakers can work together to support the well-being of autistic members of our community throughout the lifespan.

Supplemental Material

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Supplemental Material

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Footnotes

Acknowledgements

The members of Project Stakeholders Engaging in Early Intervention Research (STEER) include the following: Alexandra Maher, Amy Wood, Angie Ali, Anne Weisgerber, Annette Lugo, Brandon Adjoumani, Brandon Simpson, Cara Brennan, Carter Sanders, Cassidy Anderson, Chelsey Grimes, Christine Eichelberger, Dairia Edmunds, Douglas DeVito, Elizabeth Waterman, Faith Burmania, Jada Thompson, Jodi Kempner Collins, Laura Gambill, Lindsay Stonecipher, Michelle Hurtt, Nicholas Brown, Regina Parker, Samantha Leahy, Sara Hedrick and Rufus the Service Dog, Shannon Martin, Tema Krempley, Viviana Figueroa, and William Taylor.

Author contributions

K.M.W. and A.L.W. conceptualized and designed the work, secured funding for the work, developed the engagement methodology, assisted with moderation of engagement activities, supervised the work, drafted the initial article, and reviewed and revised the article. A.R.B. and R.A.G. assisted with development of engagement methodology, assisted with moderation of engagement activities, managed and coordinated the project, drafted the initial article, and reviewed and revised the article. All authors approved the final article as submitted and agreed to be accountable for all aspects of the work.

Declaration of conflicting interests

The author(s) declared the following potential conflicts of interest with respect to the research, authorship, and/or publication of this article: K.M.W. and A.L.W. receive fees for conducting workshops regarding implementation of specific autism early intervention programs (Reciprocal Imitation Training and Project ImPACT).

Funding

The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: Research reported in this article was funded through a Patient-Centered Outcomes Research Institute (PCORI) Award (14089-TOSU). The statements in this article are solely the responsibility of the authors and do not necessarily represent the views of the PCORI, its Board of Governors, or Methodology Committee. This project was supported, in part, by the National Center for Advancing Translational Sciences of the National Institutes of Health under Grant Number UL1TR002733. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.

ORCID iD

Katherine M Walton

Supplemental material

Supplemental material for this article is available online.

Notes

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