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Brief Report: A Survey of Autism Research Priorities Across a Diverse Community of Stakeholders

  • Autism Speaks
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Inclusion of stakeholder voices in the allocation of research funding can increase the relevance of results and improve community engagement in research. We describe the results of an online survey that gathered input from community stakeholders regarding autism research priorities. A demographically diverse sample of respondents (N = 6004; 79.1% female; 72.5% ages 30–59; 86.4% USA) completed the survey. Results indicated a preference for applied relative to basic science topics, though both basic and applied science areas were rated as important. Respondents gave their highest ratings to research focused on co-occurring conditions, health and well-being, adult transition, and lifespan issues. These results can guide decision-making by public and private funders when developing science funding priorities and engaging in science dissemination activities.
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Journal of Autism and Developmental Disorders (2018) 48:3965–3971
Brief Report: ASurvey ofAutism Research Priorities Across aDiverse
Community ofStakeholders
ThomasW.Frazier1· GeraldineDawson2· DonnaMurray1· AndyShih3· JessicaSnyderSachs4· AngelaGeiger4
Published online: 15 June 2018
© Springer Science+Business Media, LLC, part of Springer Nature 2018
Inclusion of stakeholder voices in the allocation of research funding can increase the relevance of results and improve com-
munity engagement in research. We describe the results of an online survey that gathered input from community stakeholders
regarding autism research priorities. A demographically diverse sample of respondents (N = 6004; 79.1% female; 72.5% ages
30–59; 86.4% USA) completed the survey. Results indicated a preference for applied relative to basic science topics, though
both basic and applied science areas were rated as important. Respondents gave their highest ratings to research focused on
co-occurring conditions, health and well-being, adult transition, and lifespan issues. These results can guide decision-making
by public and private funders when developing science funding priorities and engaging in science dissemination activities.
Keywords Autism· Research priorities· Caregivers· Funding· Stakeholders· Adult transition
Over the last two decades, research has identified and
begun to explain the etiologic and phenotypic heteroge-
neity of autism (de la Torre-Ubieta etal. 2016; Georgia-
des etal. 2014). Increases in public and private/non-profit
research funding have been an important part of promot-
ing autism research advances. From 2008 to 2015, autism
research funding in the US (public and private) expanded
from $222million to $343million, with projected increases
to $496million by 2020 (source: NIH Reporter) (National
Institutes of Health 2017). Expanded funding has paralleled
increases in the number of autism publications, from < 500
in 2000 to > 3500 in 2015 (source: US National Library of
Medicine PubMed database search) (Ncbi Resource Coor-
dinators 2017). Similarly, the number of patent applications
relevant to autism increased from 4 in 2001 to almost 60
in 2014 (source: US Patent and Trademark Office AppFT)
(US Patent and Trademark Office 2017). While not every
research area has received strong funding, investigations
have proceeded on a wide front, with topics ranging from
genetics and basic neuroscience to cognition and behavior
to clinical trials and quality of life. As the cohorts of young
children diagnosed during the expansion of autism aware-
ness have progressed to adolescence and early adulthood,
research into understudied topics such as adult transition has
also increased. However, absolute funding levels for services
and lifespan research—areas important to the quality of life
of many individuals and families—lag behind (Interagency
Autism Coordinating Committee (IACC) October 2017).
Understanding the wide range of needs and desires of
people that are directly affected by autism, including per-
sons on the spectrum and families, as well as providers
(clinicians and educators) and researchers, is important in
setting priorities regarding autism research funding. (Pel-
licano etal. 2014a, b). Including viewpoints of those with
autism and their families as well as those who work directly
with persons on the spectrum, such as therapists and teach-
ers, in decision-making can increase the benefits that future
research delivers to individuals and families, iteratively
Electronic supplementary material The online version of this
article (https :// 3-018-3642-6) contains
supplementary material, which is available to authorized users.
* Thomas W. Frazier
1 Science Department, Autism Speaks, 1 East 33rd Street 4th
Floor, NewYork, NY10016, USA
2 Department ofPsychiatry andBehavioral Sciences, Duke
University, Durham, NC, USA
3 Global Autism Public Health, Autism Speaks, NewYork,
4 Autism Speaks, NewYork, USA
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
... Nineteen studies reported the number of engaged patients with a median of 47 (3-4885) ( Table 2a). The study engaging the largest number of patients did so through a priority setting questionnaire [23]. Fourteen studies reported on at least one demographic feature of patient partners (i.e. ...
... The second theme acknowledges challenges associated with engaging a small sample of patient partners and how this may limit perspectives brought to a research project. Conversely, it was suggested engaging multiple In vitro G Explicit statement of funding received to support patient engagement Russell et al. [21] United Kingdom Autism In vivo G, F, I N/R Tamagnini et al. [22] United Kingdom Alzheimer's and dementia In vivo, Ethics of animal research F Explicit statement of funding received to support patient engagement Frazier et al. [23] United States Autism N/R N/R Talebizadeh et al. [24] United patient partners may help ensure diverse perspectives are considered. ...
... Three themes outlined lessons to consider before engaging patient partners. These included 'providing educational resources to team members' covering sufficient background information and rationale for the project, planning to 'recognize patient partner contributions' through compensation or acknowledgement for example, and the importance of 'timing patient Rheault et al. [18] x x x N/R Pre-conference and conference events (NR) van den Berg et al. [19] x x x x N/R Face-to-face workshop (2-days) Boenink et al. [20] x N/R One-time engagement (N/R) Russell et al. [21] x x x x 66 2 events (1-year apart) with email follow-up (N/R) Tamagnini et al. [22] x x x 3 N/R Frazier et al. [23] x x x x 4885 One-time engagement (N/R) Talebizadeh et al. [24] x x x 12 Attendance at 6 sessions over a 12-month period McDonnell et al. [25] x 523 One-time engagement (N/R) Parsons et al. [26] x 63 One focus group (90 min) Zoeller [27] x x 71 Attendance at meetings (Two weekends) Filocamo [28] x N/R Attendance at several meetings and workshops (N/R) Black and Brockway-Lunardi [29] x x N/R N/R Godard et al. [30] x 1,568 One-time engagement (N/R) *Haga et al. [31] x 159 8 group sessions (N/R) *O'Daniel et al. [32] x 159 8 group sessions (N/R) Terry et al. [33] x N/R 2-year project Pulver et al. [34] x 53 One-time engagement (N/R) Arturi [35] x x N/R N/R Baart and Abma [36] x x 16 1-year project Boon and Broekgaarden [37] x x N/R N/R Van Olphen et al. [38] x x x 9 N/R Haddow et al. [39] x x x N/R N/R Riter and Weiss [40] x x x x x 12+ N/R Mollan et al. [42] x x x x 122 18-month project Costello and Dorris [43] x ...
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Background: 'Patient engagement' involves meaningful collaboration between researchers and 'patient partners' to co-create research. It helps ensure that research being conducted is relevant to its ultimate end-users. Although patient engagement within clinical research has been well documented, the prevalence and effects of patient engagement in translational preclinical laboratory research remain unclear. The aim of this scoping review is to present current patient engagement activities reported in preclinical laboratory research. Methods: MEDLINE, Embase, and grey literature were systematically searched from inception to April 2021. Studies that described or investigated patient engagement in preclinical laboratory research were included. Patient engagement activities where patients (i.e. patients, family members, caregivers or community members) provided input, or consultation on at least one element of the research process were eligible for inclusion. Study characteristics and outcomes were extracted and organized thematically. Findings: 32 reports were included (30 primary studies, 1 narrative review, and 1 researcher guide). Most studies engaged patients at the education or priority setting stages (n=26). The most frequently reported benefit of patient engagement was 'providing a mutual learning opportunity'. Reported barriers to patient engagement reflected concerns around 'differences in knowledge and research experience' and how this may challenge communication and limit meaningful collaboration. Interpretation: Patient engagement is feasible and beneficial for preclinical laboratory research. Future work should focus on assessing the impacts of patient engagement in this area of research.
... (6) Acknowledge that we are at the beginning of studying autistic adults. (7) Advocate for more funding for research in autistic adults. ...
... Whether it be the proliferation of the autism-vaccine hoax, 1 the use of social media and internet forums to foster community among autistic adults, 2,3 a resource for autistic adults, caregivers, and parents seeking advice and support, 4 or a medium to provide intervention to underserved areas, 5 the internet has played a considerable role in the development and growth of the autism community. From the research perspective, the increasing need to focus on adult outcomes 6,7 and the burgeoning online autistic adult community 8 has led to a boom in internet-based surveys that can reach a population of autistic adults, often overlooked in research studies. 9 To illustrate, the Autism Science Foundation, a nonprofit research funder in the United States, alone has posted upward of 20 active online research surveys one can elect to participate in at the time of our writing. ...
Internet-based online surveys are a crucial tool for researchers to learn about the understudied and often overlooked population of autistic adults. The recruitment and administration of online surveys can be cheaper, quicker, and have a wider reach compared with more traditional in-person methods. As online surveys become more prevalent, it is important to place strengths in the context of limitations and biases that can arise when recruiting and administering surveys online. In this perspective, we discuss two common issues that often appear in studies that use online tools to recruit and administer surveys to autistic adults and nonautistic volunteers: selection bias and sample identifiability. Selection bias is the distortion in effect estimates (e.g., relative risk, risk ratio, incidence rate) resulting from the factors that influence why a person chose to participate or how the researcher recruits and selects participants in a study. Sampling identifiability is the ability (or inability) to quantify and define the population of interest. We use a case example of an online survey study of suicidal ideation in autistic adults and describe how issues in selection bias and sample identifiability arise and may lead to challenges unique to studying autistic adults. We conclude with recommendations to improve the quality and utility of online survey research in autistic adults. Using online resources to recruit and collect data on autistic adults is an incredible tool with great potential; yet, authors need to consider the limitations, potential biases, and tools to overcome systematic error at each stage of the study. Lay summary: What is the purpose of this article?: Our purpose was to describe challenges in conducting and analyzing data from surveys of autistic adults, recruited and completed online.What is already known on the topic?: Health outcomes for autistic adults are understudied by crucial areas of autism research. While researchers are interested in the outcomes of autistic adults, this type of research is difficult because many autistic adults are not formally diagnosed, so not available to recruit for studies through clinic registries. Furthermore, study participation can be a long, inconvenient, and stressful process. It is not surprising then that we are seeing internet surveys of autistic adults become a popular tool to reach this population. We wanted to offer an overview and recommendations of these issues to researchers and people who read research about topics pertaining to autistic adults.What are the perspectives of the authors?: We are epidemiologists at Boston University and the University of Wisconsin-Madison. We both conduct research centered in improving health and well-being for autistic people across the life span. As people who study research methods, we have seen a lot of new research using survey methods. This research is intriguing, but all too often the articles need more information so we can be sure that the research is high quality. We want to share ways to improve this type of research and to help people in understanding the strengths and limitations of online survey research.What do the authors recommend?: We offer a few considerations for researchers working in this area. (1) Make the steps you took to do the research as clear as possible. (2) Be specific about who you intend to study and who you ended up studying. (3) Present the demographics and characteristics of the participants. (4) If possible, consider using data analysis methods to account for selection bias and sample identifiability issues. (5) Do not make statements that are not supported for your study results. (6) Acknowledge that we are at the beginning of studying autistic adults. (7) Advocate for more funding for research in autistic adults.How will these recommendations help autistic adults now or in the future?: Online surveys are an important tool for researchers to generate hypotheses and connect with a wider range of participants. However, online surveys have unique methodological challenges. We hope that this perspective raises the topic of bias and misinterpretation in online surveys and researchers continue to produce valid and meaningful work that is crucial to improving lives of autistic adults.
... To try to capture this reality, recent investigations in different countries have sought to compare the interests of autistic people themselves with the interests of autism researchers (e.g. Benevides et al., 2020;Cusack & Sterry, 2016;Frazier et al., 2018;Pellicano et al., 2014). In Australia, this has led to a recent publication of a report by the Australian Autism Research Council (AARC), a group of community members from different stakeholder groups, including autistic people, parents and carers and researchers, which sits under the auspices of the Cooperative Research Centre for Living with Autism (AARC, 2021b). ...
... It is not, surprising, then, that the vast preponderance of autism research worldwide focuses on the underlying genetic causes and biology of autism (den Houting & Pellicano, 2019;Office of Autism Research Coordination, 2019;Pellicano, Dinsmore, & Charman, 2014b;Singh, Illes, Lazzeroni, & Hallmayer, 2009). This focus is in sharp contrast to the stated research priorities of community membersautistic people, their family members, educators, clinicians and other professionalswho have consistently called for research on areas that are of more immediate, practical concern or for basic science research that may be more straightforwardly translated into applications (Frazier et al., 2018;Jose et al., 2020;Pellicano et al., 2014b;Robertson, 2010;Roche, Adams, & Clark, 2021;Warner, Parr, & Cusack, 2019). In the words of one parent, 'when it comes down to it, it's not real life . . . ...
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Since its initial description, the concept of autism has been firmly rooted within the conventional medical paradigm of child psychiatry. Increasingly, there have been calls from the autistic community and, more recently, nonautistic researchers, to rethink the way in which autism science is framed and conducted. Neurodiversity, where autism is seen as one form of variation within a diversity of minds, has been proposed as a potential alternative paradigm. In this review, we concentrate on three major challenges to the conventional medical paradigm – an overfocus on deficits, an emphasis on the individual as opposed to their broader context and a narrowness of perspective – each of which necessarily constrains what we can know about autism and how we are able to know it. We then outline the ways in which fundamental elements of the neurodiversity paradigm can potentially help researchers respond to the medical model’s limitations. We conclude by considering the implications of a shift towards the neurodiversity paradigm for autism science.
... The concerns around RRBI intervention design echo broader concerns about research on ASD, with calls to focus on issues that more directly impact on autistic people's lives (Frazier et al., 2018;Roche et al., 2021). For example, Roche et al. (2021) conducted a systematic review of studies that reported on stakeholders' priorities for research. ...
Conference Paper
Aim: Restricted and Repetitive Behaviours and Interests (RRBIs) are a diagnostic feature of Autism Spectrum Disorder (ASD). The literature on RRBIs interventions has been critiqued for focusing on symptom reduction instead of broader outcomes which are meaningful to autistic Children and Young People (CYP). Given the ecosystemic and CYP-centred framework within which most EPs practice, the extent to which EP practice and research can inform each other is unclear. The aim of this research was to bridge research and practice on RRBIs, using Bronfenbrenner’s bioecological model as a framework for discussion. Method: Part 1: A systematic scoping review (SSR) of RRBI intervention design that examined how outcomes are defined, by whom, and whether interventions are effective in achieving broader outcomes. The SSR included 564 studies. Part 2: Focus groups with 7 EPs and 3 Trainee EPs to elicit their views on how they practice with regard to RRBIs. Findings: The SSR indicated that the reasons for intervention were not clearly reported, and the intended broader outcomes were not consistently measured. CYP views were rarely reported. Parent/caregiver and teacher views on RRBIs were largely summarised in short statements of negative impact. There were few reports of collaborative approaches to setting outcomes. A discussion of a subset of studies that defined and measured reasons for intervention suggested that changes in RRBIs sometimes, but not always, are associated with changes in broader outcomes. The EP focus groups emphasised understanding the meaning / function of behaviour and advocating against normalisation. EPs suggested that they avoid the RRBI term because it implies deficit and because of its limitations in understanding behaviour. Conclusion: Current intervention literature is limited in informing when and for what purpose intervention on RRBIs may be effective. Barriers to meaningful collaboration between research and practice are discussed. EPs can contribute to reframing RRBI research with a focus on meaningful outcomes for autistic CYP and their families.
... Examining and addressing co-occurring mental health conditions, mental health service needs, evidence-based mental health treatments, and capacity building of mental health services for autistic individuals is an important priority of stakeholders, including autistic individuals, family members, providers, organization and system leaders, and researchers [18,19]. In this review, we summarize recent advances and continued gaps in quality mental healthcare for autistic individuals across the lifespan. ...
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Purpose of Review This synthesis of recent mental health services research with autistic individuals presents significant advances, current gaps, and recommendations for improving mental healthcare for this population. Recent Findings Recent advances include improved understanding of co-occurring mental health conditions among autistic individuals, a growing evidence base for interventions to address them, the development and implementation of new service models to support mental health for this population, and a substantial increase in mental health services and implementation research focused on autism. Ongoing challenges include a lack of mental health interventions designed for community implementation with autistic individuals, limited workforce capacity, complex and disconnected service systems, and racial, ethnic, and socioeconomic disparities in accessibility and quality of mental health services. Summary Despite the advances in our understanding of mental health needs and mental health services for autistic individuals, several critical gaps remain. We encourage future efforts to develop and test interventions that can be used in community settings, train and incentivize the workforce to provide them, realign policies and funding with best practice, and embrace an equity-focused approach to autism research and care.
... Researchers should work together with individuals with autism/their families, advocacy groups, health system leaders, and policymakers to design longitudinal studies, address stakeholder interests, and inform relevant policies/programs. 113,114 These studies should include populations from across the globe, in Table 1. Recommendations to improve healthcare access for individuals with ASD. ...
Most individuals with autism spectrum disorder (ASD)—a complex, life-long developmental disorder—do not have access to the care required to address their diverse health needs. Here, we review: (1) common barriers to healthcare access (shortage/cost of services; physician awareness; stigma); (2) barriers encountered primarily during childhood (limited screening/diagnosis; unclear referral pathways), transition to adulthood (insufficient healthcare transition services; suboptimal physician awareness of healthcare needs) and adulthood (shortage of services/limited insurance; communication difficulties with physicians; limited awareness of healthcare needs of aging adults); and (3) advances in research/program development for better healthcare access. A robust understanding of barriers to accessing healthcare across the lifespan of autistic individuals is critical to ensuring the best use of healthcare resources to improve social, physical, and mental health outcomes. Stakeholders must strengthen healthcare service provision by coming together to: better understand healthcare needs of underserved populations; strengthen medical training on care of autistic individuals; increase public awareness of ASD; promote research into/uptake of tools for ASD screening, diagnosis, and treatment; understand specific healthcare needs of autistic individuals in lower resource countries; and conduct longitudinal studies to understand the lifetime health, social, and economic impacts of ASD and enable the evaluation of novel approaches to increasing healthcare access. Despite the growing body of evidence, our understanding of barriers to healthcare encountered by individuals with ASD remains limited, particularly beyond childhood and in lower resource countries. We describe current and emerging barriers to healthcare access encountered by individuals with ASD across the lifespan. We recommend that stakeholders develop evidence-informed policies, programs, and technologies that address barriers to healthcare access for individuals with ASD and consider broad, equitable implementation to maximize impact.
... Expertise about autistic people has been created by and situated in non-autistic professionals. In service DRAFT provision and research, this has begun to change with an investment in seeking stakeholder views and more participatory action research studies (Frazier et al., 2018). Research organizations such as Autistica include autistic people in research about autism and support research undertaken by autistic people. ...
For many families, having a child with autism can be a long-term worry and involvement. Parents worry how their children will cope beyond the family home and they worry about the impact on their siblings. For parents, siblings, partners and children of people with an Autism Spectrum Condition, there are challenges which can impact on their own wellbeing. Exhaustion, stress, worry and the misunderstandings of others are some of the main issues for both autistic people and their families. Health and social care professionals are often undereducated in the impact of autism in people’s lives and this affects whether and how people with autism and their families feel about approaching services. Because autism is a hidden disability, you can't always tell if someone is autistic. People with autism and their families are often subject to stigma and discrimination, misunderstanding and criticism. Family therapists have a great deal to bring to the world of autism. We specialize in communication, coordination, working with people’s strengths and interests. We work collaboratively with family members to find new ways of going on in our everyday relationships and co-construct a safe reflective space that can lead to change without compromising people’s identities and values. In this chapter, we argue the case for systemic family therapy to be included in the resources offered to autistic people and their families. First, we offer some background information on autism. We discuss trends in autism theory, diagnosis, research and treatment methods and look at how autism can impact on people’s lives. Then, we introduce new systemic theory and practice for working with the impact of autism in families through sharing examples illustrating how systemic family therapy improves communication. Finally, we highlight areas for developing systemic family therapy training, practice and research on autism.
Background: The preferences of autism stakeholders regarding the top priorities for future autism research are largely unknown. Objective: This study had two objectives: First, to examine what autism stakeholders think new research investments should be and the attributes of investment that they consider important, and second, to explore the feasibility, acceptability and outcomes of two prioritization exercises among autism stakeholders regarding their priorities for future research in autism. Design: This was a prospective stakeholder-engaged iterative study consisting of best-worst scaling (BWS) and direct prioritization exercise. Setting and participants: A national snowball sample of 219 stakeholders was included: adults with autism, caregivers, service providers and researchers. Main outcome measures: The main outcomes measures were attributes that participants value in future research investments, and priority research investments for future research. Results: Two hundred and nineteen participants completed the exercises, of whom 11% were adults with autism, 58% were parents/family members, 37% were service providers and 21% were researchers. Among stakeholders, the BWS exercises were easier to understand than the direct prioritization, less frequently skipped and yielded more consistent results. The proportion of children with autism affected by the research was the most important attribute for all types of stakeholders. The top three priorities among future research investments were (1) evidence on which child, family and intervention characteristics lead to the best/worst outcomes; (2) evidence on how changes in one area of a child's life are related to changes in other areas; and (3) evidence on dietary interventions. Priorities were similar for all stakeholder types. Conclusions: The values and priorities examined here provide a road map for investigators and funders to pursue autism research that matters to stakeholders. Patient or public contribution: Stakeholders completed a BWS and direct prioritization exercise to inform us about their priorities for future autism research.
Lay abstract: In 2017, an advisory board consisting of autism researchers and community members recommended that funders of autism research prioritize research projects on: (1) treatments/interventions, (2) evidence-based services, and (3) lifespan issues. To describe funding in these areas since this recommendation was made, we searched the databases of the three largest federal funders of autism research in the United States. We found that the largest portion of federal funding during 2017-2019 was awarded to research on the biology of autism (32.59%) and treatments and interventions for autism (22.87%). Less funds were awarded to research areas that are high funding priorities by the Interagency Autism Coordinating Committee budget recommendation including services (5.02%) and lifespan issues (2.51%). Our findings emphasize that autism research funding is not consistent with the Interagency Autism Coordinating Committee budget recommendation to increase funding particularly to services and lifespan issues. We recommend that funding patterns should shift to better align with these priorities so that autism research may better serve the needs of the autism community.
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Engagement with stakeholders is an essential part of the research process. This is particularly the case for early autism research with infant cohorts and their families, where a range of ethical issues are pertinent. Here, we report on a large survey of parents who have a child on the autism spectrum (n = 1040) which specifically probed attitudes to early autism research. The large majority of parents showed positive attitudes overall, and these were associated with greater access to services, higher service quality ratings and higher rates of intellectual disability among their children. Parents valued the scientific goals of research, but half of parents also reported that an intervention component would be an essential prerequisite for them to participate in research. If enrolled in a study, parents were positive about most commonly used measures though less favourably disposed towards brain scans for children. They valued direct contact with the research team and openness in data sharing. We interpret our findings in terms of lessons for the early autism research community and for stakeholder engagement projects.
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The National Center for Biotechnology Information (NCBI) provides a large suite of online resources for biological information and data, including the GenBank® nucleic acid sequence database and the PubMed database of citations and abstracts for published life science journals. The Entrez system provides search and retrieval operations for most of these data from 37 distinct databases. The E-utilities serve as the programming interface for the Entrez system. Augmenting many of the Web applications are custom implementations of the BLAST program optimized to search specialized data sets. New resources released in the past year include iCn3D, MutaBind, and the Antimicrobial Resistance Gene Reference Database; and resources that were updated in the past year include My Bibliography, SciENcv, the Pathogen Detection Project, Assembly, Genome, the Genome Data Viewer, BLAST and PubChem. All of these resources can be accessed through the NCBI home page at
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Investigation into the earliest signs of autism in infants has become a significant sub-field of autism research. This work invokes specific ethical concerns such as use of ‘at-risk’ language, communicating study findings to parents and the future perspective of enrolled infants when they reach adulthood. This study aimed to ground this research field in an understanding of the perspectives of members of the autism community. Following focus groups to identify topics, an online survey was distributed to autistic adults, parents of children with autism and practitioners in health and education settings across 11 European countries. Survey respondents (n = 2317) were positively disposed towards early autism research, and there was significant overlap in their priorities for the field and preferred language to describe infant research participants. However, there were also differences including overall less favourable endorsement of early autism research by autistic adults relative to other groups and a dislike of the phrase ‘at-risk’ to describe infant participants, in all groups except healthcare practitioners. The findings overall indicate that the autism community in Europe is supportive of early autism research. Researchers should endeavour to maintain this by continuing to take community perspectives into account.
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Identifying biomarkers for autism can improve outcomes for those affected by autism. Engaging the diverse stakeholders in the research process using community-based participatory research (CBPR) can accelerate biomarker discovery into clinical applications. However, there are limited examples of stakeholder involvement in autism research, possibly due to conceptual and practical concerns. We evaluate the applicability of CBPR principles to biomarker discovery in autism and critically review empirical studies adopting these principles. Using a scoping review methodology, we identified and evaluated seven studies using CBPR principles in biomarker discovery. Results and conclusions The limited number of studies in biomarker discovery adopting CBPR principles coupled with their methodological limitations suggests that such applications are feasible but challenging. These studies illustrate three CBPR themes: community assessment, setting global priorities, and collaboration in research design. We propose that further research using participatory principles would be useful in accelerating the pace of discovery and the development of clinically meaningful biomarkers. For this goal to be successful we advocate for increased attention to previously identified conceptual and methodological challenges to participatory approaches in health research, including improving scientific rigor and developing long-term partnerships among stakeholders.
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There has been a substantial increase in research activity on autism during the past decade. Research into effective ways of responding to the immediate needs of autistic people is, however, less advanced, as are efforts at translating basic science research into service provision. Involving community members in research is one potential way of reducing this gap. This study therefore investigated the views of community involvement in autism research both from the perspectives of autism researchers and of community members, including autistic adults, family members and practitioners. Results from a large-scale questionnaire study (n = 1,516) showed that researchers perceive themselves to be engaged with the autism community but that community members, most notably autistic people and their families, did not share this view. Focus groups/interviews with 72 participants further identified the potential benefits and remaining challenges to involvement in research, especially regarding the distinct perspectives of different stakeholders. Researchers were skeptical about the possibilities of dramatically increasing community engagement, while community members themselves spoke about the challenges to fully understanding and influencing the research process. We suggest that the lack of a shared approach to community engagement in UK autism research represents a key roadblock to translational endeavors.
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Participatory research aims to increase the relevance and broaden the implementation of health research by involving those affected by the outcomes of health studies. Few studies within the field of neurodevelopmental disorders, particularly autism spectrum disorders, have involved autistic individuals as partners. This study sought to identify and characterize published participatory research partnerships between researchers and individuals with autism spectrum disorder or other neurodevelopmental disorders and examine the influence of participatory research partnerships on the research process and reported study outcomes. A search of databases and review of gray literature identified seven studies that described participatory research partnerships between academic researchers and individuals with autism spectrum disorder or other neurodevelopmental disorders. A comparative analysis of the studies revealed two key themes: (1) variations in the participatory research design and (2) limitations during the reporting of the depth of the partner's involvement. Both themes potentially limit the application and generalizability of the findings. The results of the review are discussed in relation to the use of evaluative frameworks for such participatory research studies to determine the potential benefits of participatory research partnerships within the neurodevelopmental and autism spectrum disorder populations.
Should we continue to refer to autism as a ‘disease’ or ‘disorder’, or is the framework of ‘neurodiversity’ a more humane and accurate lens through which to view people with autism? Evidence at the genetic, neural, behavioural and cognitive levels reveals people with autism show both differences, and signs of disability, but not disorder. Disability requires societal support, acceptance of difference and diversity, and societal “reasonable adjustment”, whilst disorder is usually taken to require cure or treatment. These are very different frameworks. It will be important to see how the concept of neurodiversity is applied to the 300 diagnoses in DSM-5, and if it revolutionizes both the science and the practice of psychiatry.
Objective: To illustrate the process of community-partnered participatory research (CPPR) to develop and evaluate interventions for children with autism in urban school districts METHODS: We formed partnerships of school personnel, parents, and researchers to guide the project. We then conducted focus groups, key informant interviews, and town halls to explore how public schools currently serve students with autism. We used findings from these activities to adapt interventions for public schools. We then tested interventions in randomized clinical trials (RCTs). Results: Community input indicated a particular need for interventions to improve children's social interaction and instructional supports to promote their success throughout the day. Based on this input, we adapted two interventions: Remaking Recess for improving peer engagement during social times; and Schedules, Tools, and Activities for Transition (STAT) for facilitating successful transitions between activities throughout the daily routine. Results of the RCT of Remaking Recess are not yet available. The RCT of STAT involved 150 children and 56 teachers. Teachers reported high buy-in and increased their proficiency at implementing STAT; children with ASD reduced their disruptive behavior and made progress toward teacher-nominated goals. However, teachers' implementation remained inconsistent, and children did not reliably improve in academic engagement or independence. Conclusions: The findings suggest that, although CPPR has limitations, it can assist in selecting interventions to address community priorities and produce some favorable outcomes for children with autism in public schools. An important next step is to evaluate the sustainability of the interventions introduced in this project.
Progress in understanding the genetic etiology of autism spectrum disorders (ASD) has fueled remarkable advances in our understanding of its potential neurobiological mechanisms. Yet, at the same time, these findings highlight extraordinary causal diversity and complexity at many levels ranging from molecules to circuits and emphasize the gaps in our current knowledge. Here we review current understanding of the genetic architecture of ASD and integrate genetic evidence, neuropathology and studies in model systems with how they inform mechanistic models of ASD pathophysiology. Despite the challenges, these advances provide a solid foundation for the development of rational, targeted molecular therapies.
The latent class structure of autism symptoms from the time of diagnosis to age 6 years was examined in a sample of 280 children with autism spectrum disorder. Factor mixture modeling was performed on 26 algorithm items from the Autism Diagnostic Interview - Revised at diagnosis (Time 1) and again at age 6 (Time 2). At Time 1, a "2-factor/3-class" model provided the best fit to the data. At Time 2, a "2-factor/2-class" model provided the best fit to the data. Longitudinal (repeated measures) analysis of variance showed that the "2-factor/3-class" model derived at the time of diagnosis allows for the identification of a subgroup of children (9 % of sample) who exhibit notable reduction in symptom severity.