In the United States, autistic individuals experience disproportionate physical and mental health challenges relative to non-autistic individuals, including higher rates of co-occurring and chronic conditions and lower physical, social, and psychological health-related quality of life. The Autism Intervention Research Network on Physical Health (AIR-P) is an interdisciplinary, multicenter research network for scientific collaboration and infrastructure that aims to increase the life expectancy and quality of life for autistic individuals, with a focus on underserved or vulnerable populations. The current paper describes the development of the AIR-P Research Agenda.
Development of the research agenda involved an iterative and collaborative process between the AIR-P Advisory Board, Steering Committee, and Autistic Researcher Review Board. The methodology consisted of 3 phases: (1) ideation and design, (2) literature review and synthesis; and (3) network engagement.
Six core research priorities related to the health of autistic individuals were identified: (1) primary care services and quality, (2) community-based lifestyle interventions, (3) health systems and services, (4) gender, sexuality, and reproductive health, (5) neurology, and (6) genetics. Specific topics within each of these priorities were identified. Four cross-cutting research priorities were also identified: (1) neurodiversity-oriented care, (2) facilitating developmental transitions, (3) methodologically rigorous intervention studies, and (4) addressing health disparities.
The AIR-P Research Agenda represents an important step forward for enacting large-scale health-promotion efforts for autistic individuals across the lifespan. This agenda will catalyze autism research in historically underrepresented topic areas while adopting a neurodiversity-oriented approach to health-promotion.
In the United States, autistic individuals experience disproportionate physical and mental health challenges relative to non-autistic individuals, including higher rates of co-occurring and chronic conditions and lower physical, social, and psychological health-related quality of life.
This research agenda represents an important step forward for enacting large-scale health-promotion efforts for autistic individuals across the lifespan. This agenda will catalyze autism research in historically underrepresented topic areas while adopting a neurodiversity-oriented approach to health-promotion.
In the United States, autistic individuals experience disproportionate physical and mental health challenges relative to non-autistic individuals, including higher rates of co-occurring and chronic conditions and lower physical, social, and psychological health-related quality of life.1–5 Funded by the Health Resources and Services Administration, Maternal and Child Health Bureau, the Autism Intervention Research Network on Physical Health (AIR-P) seeks to establish and maintain a research network to enhance the physical health and well-being of autistic children, adolescents, and adults, particularly for underserved and vulnerable populations. The current paper describes the AIR-P Research Agenda, developed through collaborations with researchers, practitioners, stakeholders, and autistic individuals; literature reviews; and consensus-building. This research agenda will be iteratively revised as the field continues to advance to ensure that ongoing efforts within the network reflect state-of-the-art research, priorities, and practice.
Methods
Sample
The research agenda development process involved a collaborative process among the AIR-P Advisory Board (n = 33), Steering Committee (n = 14), and Autistic Researcher Review Board (ARRB) (n = 8). The Advisory Board is composed of researchers and practitioners across the United States who have expertise in autism and support research efforts within the network. The Steering Committee is composed of interdisciplinary researchers, practitioners, and other stakeholders with professional expertise, as well as personal experience working with or caring for autistic individuals across the lifespan. The ARRB is composed of autistic researchers and scholars in the United States, United Kingdom, and Canada who leverage their unique personal experiences to inform research that seeks to enhance the lives of autistic individuals. The network engagement phase of the methodology (described below) included a subsample (n = 24) from the Advisory Board (n = 13; 54%), Steering Committee (n = 6; 25%), and ARRB (n = 5; 21%). This subsample included individuals with diverse levels of training and education (PhD: n = 10, 41%; MD/PhD: n = 5; 21%; MD: n = 4, 17%; students and residents: n = 3, 13%; and master’s level: n = 2, 8%). The sample also included those who identified as autistic (n = 5; 21%), those with an autistic family member or close friend (n = 12; 50%), and caregivers (n = 5; 21%).
Methodology
The methodology consisted of 3 phases:
1. Ideation and design: through a series of planning meetings, the Steering Committee identified preliminary priority domains based on their areas of expertise and background knowledge of the literature;
2. Literature review and synthesis: the Steering Committee created a targeted annotated bibliography of quantitative and qualitative research in the identified domains and synthesized the findings in a literature review to refine the domains; and
3. Network engagement: experts across the network (n = 24) engaged in a 2-step process via Qualtrics surveys to identify priority research topics within each domain.
In the first step, the sample provided open-ended responses regarding the highest-priority research topics within each of the research domains identified during the series of planning meetings and review of the literature. The open-ended responses were organized utilizing Dedoose coding software6 and coalesced into 6 to 10 priority research areas within each domain. In the second step, participants were asked to participate in a follow-up survey to identify the top 3 priority research areas from the options within each domain, with consideration of the following criteria:
1. Need/urgency (ie, the influence of the research topic on the physical health and well-being of autistic individuals; severity of consequences [eg, number of autistic individuals affected by the issue]; and presence of critical gaps in knowledge);
2. Research impact (ie, the ability of the knowledge gained from this research topic to translate to meaningful progress in 5 to 10 years and the potential to lead to improvements in physical health status, quality of care, public health, or policy over the longer term); and
3. Person/family-centeredness (ie, how well the research topic reflects the experiences and priorities of autistic individuals and their families).
The research topics most frequently endorsed in participants’ top 3 rankings across all categories were identified. Cross-cutting research domains—that is, research areas that apply to all domains and topic areas—emerged throughout all steps of the process.
Results
The AIR-P Research Agenda, along with the highest-ranking research topic areas identified in the network engagement phase, are outlined in Table 1. The research agenda consists of 6 core research priority domains, each with critical implications for autistic individuals’ physical health, and 4 cross-cutting priorities that applied across all domains.
Research Priority/Top 3 Research Topicsa . | n (%)b . |
---|---|
Primary care services and quality: research that enhances primary care models that support autistic individuals across the lifespan | |
Autistic individuals’ self-determination (ie, capacity for autonomy, self-efficacy, and decision-making) in health care | 19 (79) |
Primary care provider training and specialized protocols for working with autistic individuals | 18 (72) |
Care and management around stress, anxiety, and/or suicidal ideation; referrals for psychiatric care | 14 (58) |
Community-based lifestyle interventions: research that enhances lifestyle interventions that support autistic individuals across the lifespan | |
Lifestyle interventions that engage communities, families, children, and peers to promote social connectedness and enhance relationships | 17 (71) |
Lifestyle interventions that build self-advocacy capacities and important skills for development and independence | 16 (67) |
Engaging families and individuals in physical activity and nutrition, increasing community supports, and strengthening the evidence base for lifestyle interventions | 12 (50) |
Health systems and services: research that facilitates health care services and systems, particularly transitions and care coordination for autistic individuals | |
Health care navigation for adolescents and adults and models of effective transition practices | 16 (67) |
Training primary care providers in the adult health care system to serve autistic individuals | 14 (58) |
Educating families and individuals about transitions and adult care | 14 (58) |
Gender, sexuality, and reproductive health: research that promotes sexual health among autistic individuals and addresses systemic barriers for women and gender nonconforming autistic individuals | |
Developing sexual health curricula for autistic individuals and promoting awareness and knowledge | 17 (71) |
Understanding and addressing individual, caregiver, and family education and needs pertaining to sexual health | 14 (58) |
Understanding and supporting youth in areas related to intersectionality, LGBTQ+ issues, and gender and sexual identity | 12 (50) |
Self-determination around sexual behavior | 12 (50) |
Neurology: research that develops and tests the neurologic and developmental outcomes of motor-based interventions for autistic individuals | |
Co-occurring conditions, (eg, motor dysfunction, sensory issues, seizures, and sleep) and corresponding accommodations, supports, and interventions | 18 (75) |
Understanding developmental neurologic trajectories and adult outcomes | 12 (50) |
Neurologic care coordination with other health care providers | 12 (50) |
Genetics: research that facilitates access to genetic testing as a means to enhance physical health among autistic individuals and their families | |
Genetics counseling and physician–patient communication | 15 (63) |
Identifying genes linked to co-occurring conditions or treatment responses | 13 (54) |
Addressing the autistic community’s concerns about genetics research and moving away from cure/cause research toward increasing quality of life | 12 (50) |
Research Priority/Top 3 Research Topicsa . | n (%)b . |
---|---|
Primary care services and quality: research that enhances primary care models that support autistic individuals across the lifespan | |
Autistic individuals’ self-determination (ie, capacity for autonomy, self-efficacy, and decision-making) in health care | 19 (79) |
Primary care provider training and specialized protocols for working with autistic individuals | 18 (72) |
Care and management around stress, anxiety, and/or suicidal ideation; referrals for psychiatric care | 14 (58) |
Community-based lifestyle interventions: research that enhances lifestyle interventions that support autistic individuals across the lifespan | |
Lifestyle interventions that engage communities, families, children, and peers to promote social connectedness and enhance relationships | 17 (71) |
Lifestyle interventions that build self-advocacy capacities and important skills for development and independence | 16 (67) |
Engaging families and individuals in physical activity and nutrition, increasing community supports, and strengthening the evidence base for lifestyle interventions | 12 (50) |
Health systems and services: research that facilitates health care services and systems, particularly transitions and care coordination for autistic individuals | |
Health care navigation for adolescents and adults and models of effective transition practices | 16 (67) |
Training primary care providers in the adult health care system to serve autistic individuals | 14 (58) |
Educating families and individuals about transitions and adult care | 14 (58) |
Gender, sexuality, and reproductive health: research that promotes sexual health among autistic individuals and addresses systemic barriers for women and gender nonconforming autistic individuals | |
Developing sexual health curricula for autistic individuals and promoting awareness and knowledge | 17 (71) |
Understanding and addressing individual, caregiver, and family education and needs pertaining to sexual health | 14 (58) |
Understanding and supporting youth in areas related to intersectionality, LGBTQ+ issues, and gender and sexual identity | 12 (50) |
Self-determination around sexual behavior | 12 (50) |
Neurology: research that develops and tests the neurologic and developmental outcomes of motor-based interventions for autistic individuals | |
Co-occurring conditions, (eg, motor dysfunction, sensory issues, seizures, and sleep) and corresponding accommodations, supports, and interventions | 18 (75) |
Understanding developmental neurologic trajectories and adult outcomes | 12 (50) |
Neurologic care coordination with other health care providers | 12 (50) |
Genetics: research that facilitates access to genetic testing as a means to enhance physical health among autistic individuals and their families | |
Genetics counseling and physician–patient communication | 15 (63) |
Identifying genes linked to co-occurring conditions or treatment responses | 13 (54) |
Addressing the autistic community’s concerns about genetics research and moving away from cure/cause research toward increasing quality of life | 12 (50) |
Research topics reflect topics that were identified in the top 3 individual rankings. Rankings were obtained separately for need or urgency, research impact, and person or family-centeredness, and the topics with the highest rankings across all categories are displayed.
This column reflects the number and percentage of experts across the network (n = 24) who ranked each research topic in their top 3 rankings of either need or urgency, research impact, or person or family-centeredness.
Core Research Priorities Identified
Primary Care Services and Quality
Relative to their non-autistic counterparts, autistic children and adults have significantly more physician visits and medications prescribed and higher medical costs and total expenditures per treated patient.7–10 Nonetheless, they create a smaller burden on health insurers because of their relatively low receipt of necessary treatment and services.11–14 In addition, there are pronounced racial, ethnic, and gender health inequities in access to quality medical services.13,14
Of particular concern, only half of autistic individuals receive comprehensive primary care consistent with the medical home model as recommended by the American Academy of Pediatrics. This model is defined as care from a personal provider that is preventive, family-centered, compassionate, culturally appropriate, accessible, comprehensive, and coordinated.15 A range of patient, provider, and systemic obstacles to an optimal medical home for autistic individuals has been cited in the literature.16–19 These obstacles perpetuate unmet health care needs related to physical and mental health, prescription medications, and preventive care, and increase emergency service utilization.20 Specific research priorities that emerged included promoting autistic individuals’ self-determination (ie, capacity for autonomy, self-efficacy, and decision-making) in health care (n = 19, 79%), enhancing primary care provider training (n = 18, 72%), and mental health and psychiatric care (n = 14, 58%).
Community-Based Lifestyle Interventions
Although many autistic individuals experience challenges related to lifestyle, including nutrition, gastrointestinal issues, food selectivity,21 obesity and associated chronic health conditions,16,22, physical activity and motor performance,23 sleep,24–26 stress and anxiety,27,28 social connectedness,29,30 and substance use,31 these challenges are variable in the autistic population. In addition, findings on the effectiveness of interventions have been inconclusive because of reliance on small sample sizes and subsequent failure to obtain robust effect sizes on physical health outcomes.22,25 Research priorities that emerged included lifestyle interventions that promote social connectedness (n = 17, 71%), build self-advocacy capacities (n = 16, 67%), and engage families and communities (n = 12, 50%).
Health Systems and Services
Although health systems and services emerged as an important priority, a focus on transitions and coordination appeared to be particularly salient. Autistic individuals and their families characterize their experiences as a “lifetime of difficult transitions.” They report difficulty using services, lack of source of care, inadequate insurance coverage, and lack of shared decision-making and care coordination. There are also a limited number of specialized service providers and resources that are often highly fragmented and governed by stringent and restrictive program and funding criteria.32–35 Further, autistic individuals frequently encounter a poor person–environment fit in health care and service settings and uncertainty about the roles of parents in facilitating transitions.34,35 Transition challenges are heightened for Black and low-income youth and those with comorbid conditions.36,37 Access to a medical home is an important predictor of receiving health care transition services because autistic youth with a medical home are almost 3 times as likely to receive health care transition services as youth without a medical home.38 Specific research priorities that emerged included supporting health care navigation and models of transition practices (n = 16, 67%), training primary care providers in the adult health system (n = 14, 58%), and educating individuals and families about transition care (n = 14, 58%). In recognition of the need for further research on health care transitions, the Maternal and Child Health Bureau has prioritized research, performance measurement, and data analysis around transitions. As an example, they invested in the Autism Transitions Research Project for Youth and Young Adults with Autism Spectrum Disorders. The AIR-P will collaborate with the Autism Transitions Research Project over the course of the grant.
Gender, Sexuality, and Reproductive Health
Mounting evidence suggests that gender-biased diagnostic and clinical criteria, along with actual sex and gender differences, may lead to the delayed recognition of autism in females.39 Barriers to service access for autistic women may continue into adulthood, particularly for health services related to reproductive health and pregnancy.40 Autistic adults are less likely to be gender-conforming than non-autistic adults. Belonging to a sexual or gender minority group is associated with higher rates of unmet health care needs among autistic people and, in turn, poorer physical health.41 Indeed, autistic individuals often receive insufficient support regarding their sexuality and gender identity.42,43 Further compounding these challenges, autistic individuals are at higher risk of sexual victimization and abuse compared with non-autistic people. These experiences are associated with suicidal or self-injurious behavior and psychiatric hospitalizations.44 Autistic adults report less perceived and actual sexual knowledge, which may increase the risk of sexual victimization.45 Specific research priorities that emerged included developing sexual health curricula for autistic individuals (n = 17, 71%), promoting sexual health education among individuals and families (n = 14, 58%), understanding intersectional identity issues (n = 12, 50%), and promoting self-determination (n = 12, 50%).
Neurology
Although autistic individuals have been found to experience motor impairments, including impairments related to gross and fine motor, postural control, and imitation and praxis, such impairments are not universal nor specific to autism.46,47 The current research base surrounding motor-based interventions has shown some promising results, but is largely based on case studies and individuals without intellectual disability, or have yielded small-effect sizes.48–50 Rigorous studies that include autistic individuals with heterogeneous behavioral and intellectual abilities are needed to objectively evaluate (1) motor dysfunction, and (2) motor-based interventions that target fundamental motor skills, social communication, and proprioceptive awareness.51–53 Specific research priorities that emerged were understanding and addressing co-occurring neurologic conditions (n = 18, 75%) and neurologic developmental trajectories and adult outcomes (n = 12, 50%), as well as coordinating neurologic care (n = 12, 50%).
Genetics
Although autism has been found to be highly heritable,54 genotyping is particularly complex due to frequently co-occurring conditions. Although a proportion of autism is attributable to common variants, rare copy-number variants and protein-disrupting, single-nucleotide variants have also been shown to significantly contribute to the etiology.55 The diagnostic relevance of genomewide small common and rare copy-number variants continues to provide evidence of the high diagnostic yield of microarray for genetic testing in autistic children.56 These findings support the utility of enhancing access to genetic testing for autistic individuals. Research priorities that emerged were genetic counseling (n = 15, 63%), identifying genes linked to co-occurring conditions or treatment responses (n = 13, 54%), and addressing the autistic community’s concerns about genetics research and moving away from cure and cause research toward increasing quality of life (n = 12, 50%).
Cross-Cutting Research Priorities Identified
Neurodiversity-Oriented Care
The neurodiversity movement challenges deficit-oriented conceptualizations of disability, reframing the disability as a valuable minority identity that does not require corrective treatment.57 The neurodiversity movement has proliferated in recent decades, primarily across college campuses, as a means to promote the success of diverse learners. However, the majority of literature on neurodiversity has been largely conceptual, with limited efforts to adapt a neurodiversity approach to health care.58 This perspective encourages the promotion of self-determination, advocacy, and decision-making among autistic individuals and is informed by autistic individuals themselves.
Facilitating Developmental Transitions
Research with nationally representative samples of autistic youth reveal profound developmental, educational, and health-related struggles in the transition to adulthood38,59 As previously mentioned, these challenges are often because of, in part, systemic factors that impede their capacity to succeed and thrive.60 Transition-related challenges occur across the lifespan for autistic individuals and represent important opportunities for intervention and support.
Methodologically Rigorous Intervention Studies
Our review of the literature revealed that translating research on physical health for autistic individuals into interventions is constrained by several factors. These include research with small samples, a limited quantity of existing interventions that target physical health promotion, and a lack of measurement strategies that capture multidimensional developmental outcomes.46,61
Addressing Health Disparities
There is a well-established research base regarding the need for research and interventions that address health and health care disparities among marginalized and underserved autistic populations.7,14,36 This includes a range of populations, including women, racial and ethnic minorities, and individuals from low socioeconomic backgrounds.
Discussion
This paper presents the development of the AIR-P Research Agenda. Our agenda catalyzes autism research in currently underrepresented topic areas (eg, gender, sexuality, and reproductive health), particularly for autistic populations that experience disproportionate disparities. Our agenda adopts a neurodiversity orientation to health promotion for autistic individuals. This model contrasts with the traditional deficit-oriented medical model that emphasizes identifying a cure or pinpointing the etiology of autism at the expense of promoting well-being and thriving among autistic individuals in accordance with their self-reported needs, experiences, and priorities.
This agenda lays the foundation for research conducted within the AIR-P Network. Moving forward, the ARRB will collaborate with the Steering Committee in the continuous development and refinement of the overall research domains. In addition, there will be systematic efforts to catalog research that emerged during the first year of the AIR-P to ensure that priorities are representative of the current research and emerging trends. There will also be continuous consensus-building efforts to ensure alignment within the network.
Conclusions
The AIR-P Research Agenda represents an important step forward for enacting large-scale health-promotion efforts for autistic individuals across the lifespan. The AIR-P looks forward to addressing these critical research priorities and continuing to advance this research agenda as the network spurs research and innovation.
The Autism Intervention Research Network on Physical Health National Coordinating Center and the Steering Committee created a targeted annotated bibliography of quantitative and qualitative research in identified domains and drafted the initial manuscript; the National Coordinating Center, Steering Committee, and Autistic Researcher Review Board participated in the qualitative research and reviewed and revised the manuscript; the Health Resources & Services Administration Maternal and Child Health Bureau contributed to the conception and design of the work, participated in revising the manuscript, and provided guidance and overarching feedback to this work; and all authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.
FUNDING: Supported by the Health Resources and Services Administration of the US Department of Health and Human Services under the Autism Intervention Research Network on Physical Health, grant UT2MC39440.
References
Abbreviations
Competing Interests
CONFLICT OF INTEREST DISCLOSURES: The authors have indicated they have no potential conflicts of interest to disclose. The information, content, and/or conclusions are those of the authors and should not be construed as the official position or policy of, nor should any endorsements be inferred by, the Health Resources and Services Administration, the US Department of Health and Human Services, or the US government.
Comments