Assessing the influence of researcher–partner involvement on the process and outcomes of participatory research in autism spectrum disorder and neurodevelopmental disorders: A scoping review

Abstract

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.

Keywords

autism neurodevelopmental disorders participatory research partnerships

The growing emphasis on participatory research (PR) over the past decade reflects the belief that research developed through active partnerships between academic researchers (hereinafter referred to as “researchers”) and those affected by the issues being studied (hereinafter referred to as “partner”) leads to higher quality research that is more relevant and applicable (Boote et al., 2002; Cargo and Mercer, 2008; Israel et al., 1998; Macaulay et al., 2011). Unlike conventional research paradigms, where decision-making rests solely with researchers (Macaulay, 2007), PR paradigms specify how partners are involved in research (Boote et al., 2002; Cargo and Mercer, 2008). This is ideal as partners’ real-world knowledge and lived experiences can complement the researchers’ theoretical and methodological expertise, thereby enhancing the process and products of research (Cargo and Mercer, 2008; Viswanathan et al., 2004). The decision to use PR is generally determined by the researcher and/or the parameters of the study, rather than by explicitly defined variations in the approach (Abma et al., 2009; Cornwall and Jewkes, 1995; McKevitt et al., 2010). At one end of the spectrum, partners may only be involved in one stage of the research study as consultants or collaborators. In its fullest expression, however, the researcher–partner cooperative involves collaborative decision-making, from developing the question, to collecting, analyzing and interpreting the data, developing conclusions, and disseminating results (Green and Mercer, 2001; Macaulay et al., 2007; Oliver et al., 2004).

There is a very rich history of PR applications, particularly on the involvement of partners with learning disabilities (LDs; Walmsley, 2001). There has been a diverse range of roles for LD partners, from autobiographers (Atkinson and Walmsley, 1999), interviewers (Williams, 1990), advisors (Atkinson and Williams, 1990; Macadam and Sutcliffe, 1996; Stalker, 1998), and partners (Ham et al., 2004; March et al., 1997; Powers et al., 2007). March (1997) particularly highlights the importance of PR; being coauthored by three individuals with LDs, the article chronicles their experiences with understanding what research is and why it was important for the authors to complete the research themselves (March et al., 1997). Building upon the groundwork from PR in LD, PR approaches now have a growing number of applications, particularly in communities defined by geographic, racial, and/or ethnic groups (Cargo and Mercer, 2008; Gauld et al., 2011; Keightley et al., 2009; Macaulay et al., 2007; Marcus et al., 2004; Royle and Oliver, 2004; Viswanathan et al., 2004), in addition to other populations, including those affected by breast cancer (Yonas et al., 2006), diabetes (Giachello et al., 2003), and caregivers of children with autism spectrum disorders (ASDs; Brookman-Frazee et al., 2012).

An emerging field of PR includes individuals with neurodevelopmental disorders as research partners. One such study (Powers et al., 2007) includes youth with disabilities as partners, who have a range of diagnoses, including cerebral palsy, epilepsy, spinal cord injury, Down syndrome, and ASD. Unfortunately, it is not clear which of the youth participated as a partners. Additionally, PR partnerships have been conducted with a variety of stakeholders, including parents and caregivers (Brookman-Frazee et al., 2012), yet, relatively few PR studies have included people with ASD or other neurodevelopmental disorders as research partners (Gilbert, 2004; Kiernan, 1999; Pellicano and Stears, 2011; Walmsley, 2004). Neurodevelopmental disorders encompass a broad range of impairments associated primarily with the growth and development of the brain or central nervous system. As such, individuals with ASD and other neurodevelopmental disorders experience varying degrees of challenges with speech and language, social relationships, motor skills, behavior, memory, and learning that have implications for PR designs. Although such challenges may limit the degree to which such individuals can be involved in certain activities (e.g. tasks that require a high degree of abstract thinking), it has been demonstrated that people with neurodevelopmental disorders can participate as partners in the development of research (Gilbert, 2004; Walmsley, 2004). Accordingly, the aims of this scoping review are to (1) identify and characterize the current PR partnerships between researchers and individuals with ASD or other neurodevelopmental disorders and (2) examine the influence of PR partnerships between researchers and partners with ASD or other neurodevelopmental disorders on the research processes and outcomes reported in the primary literature.

Methods

This scoping review was conducted to characterize the depth and breadth of research involvement of persons with neurodevelopmental disorders and ASD in published PR partnerships, using the methodological approach defined by Arksey and O’Malley (2005). Accordingly, this review followed five steps.

1. Development of research questions. Three questions guided the selection of relevant sources for this review: (1) What is the nature of the research activity of the partner in PR focused on ASD or neurodevelopmental disorders? (2) What is the breadth and depth of PR partnerships, which have been formed between researchers and partners with ASD or neurodevelopmental disorders? (3) How are the processes and outcomes of PR partnerships between researchers and partners with ASD or neurodevelopmental disorders reported?

2. Identifying relevant publications. Preliminary search terms were developed by the research team to reflect a number of core concepts pertinent to PR. These related to characterizing specific PR approaches that have been utilized, the role of individuals with neurodevelopmental disorders and ASD, and the outcomes of PR partnerships. To cover the breadth of the PR field, the following terms were included along with their synonyms: community-based PR, action research, inclusive research, participatory action research (PAR), participative evaluation, and emancipatory evaluation (Jagosh et al., 2011). Theoretical and conceptual work that provides an important backdrop to the scoping review is referenced throughout this article; however, only primary studies of PR conducted with individuals with neurodevelopmental disorders are reported in the findings.

To ensure comprehensiveness in identifying relevant primary studies, literature was sourced through electronic databases and reference lists of relevant literature (Arksey and O’Malley, 2005). We located publications in multiple steps, beginning with electronic literature databases. Once relevant material was selected from each of these databases, we searched relevant websites, URLs, and reference lists of selected studies to increase our capture of relevant material. Scoping reviews generally consult only a few main health research databases for literature; as such, we searched five electronic databases: MEDLINE^®, EBSCO, EMBASE, ERIC, and PsycINFO. This selection was designed to cover health, psychology, and education literature. The final search, undertaken with the help of a health science librarian, was implemented in September 2013. In all databases, terms related to PR/research participation/advocacy were combined with terms related to autism/neurodevelopmental disorders. For each set of terms, both controlled vocabulary and text words were utilized. No restrictions on publication date were applied. Duplicate references were filtered out as each subsequent database was searched. Appendix 1 contains the search strategy for the MEDLINE database; identical or slightly variant versions were employed in the remaining databases. A review of gray literature was also conducted which included web searches, reference lists, and conversations with stakeholders and authors via email.

3. Screening and selection of publications. The screening process of studies was guided by four criteria. Included studies were required to have (1) involved adults with neurodevelopmental disorders as research partners, (2) been reported in English, (3) reported or referred to primary empirical research, and (4) contained a description of how PR partnerships were established and implemented and/or described the outcomes of PR partnerships on neurodevelopmental disorder research and/or described barriers and/or facilitators to the establishment and implementation of PR partnerships (Jagosh et al., 2011). With the help of a multidisciplinary research team, one reviewer completed independent screening of study abstracts and extracted the data. A second reviewer checked for completeness and accuracy. Given that the aim of the study was to examine PR partnerships implemented and reported in research, articles that were not primary studies, such as news items, letters, editorials, book reviews, and articles appearing in newsletters or magazines, were excluded.

4. Organizing publications. Included studies were reviewed and summarized using a framework adapted from Oliver et al. (2004). Accordingly, descriptive study characteristics were collated in a detailed Microsoft Excel spreadsheet (version 14.0.6112.5000; Microsoft Corporation, Santa Rosa, CA, USA). These characteristics included the country of the first author, the year the study was published, the study population, the study’s aim, the method of involving partners, and key findings including PR-related outcomes, processes, contexts, challenges, as well as barriers and facilitators.

5. Reporting the results. A narrative and descriptive account was generated for each reported PR partnership. A comparative analysis focusing on the context for the study, the breadth and depth of the partner’s involvement throughout the research process, and the outcomes attributed to the partner’s involvement was conducted. Main findings were merged into three broader themes that served as a guide for organizing the discussion: (1) variations in the PR design of the studies, (2) limitations in the reporting of the depth of the partner’s involvement across studies, and (3) clarification of PR versus research assistant with a disability.

Results

Figure 1 provides an overview of the search and retrieval process. Our search strategy yielded 1431 citations for initial screening. An additional 10 citations from website searching and reference lists were reviewed. After duplicates were removed, 636 unique citations remained. Of these, 586 abstracts were excluded because they did not describe PR in individuals with ASD or other neurodevelopmental disorders, yielding a total of 60 articles. Of these, 54 articles were excluded because they did not report or refer to empirical research (n = 37), or discuss how PR partnerships were implemented, or the outcomes of partnerships (n = 16). Thus, seven studies met inclusion criteria and were included in this review (see Figure 1). The included studies were published between 2006 and 2013: three in Australia or New Zealand, three in the United States, and one in the Netherlands. Five studies described PR partnerships with individuals with intellectual disability (ID) and two studies focused on a study population with ASD. The involvement of partner(s) in specified stages of the study development within included studies is summarized in Table 1. The context, depth, breadth, and outcomes of each of the seven studies are described below.

Figure 1.

Flow diagram.

Table 1.

Summary of breadth of participatory research studies involving individuals with ASD and other neurodevelopmental disorders.

Authors	Identifying research question	Informing study methodology and methods	Collecting data	Analyzing data	Disseminating research findings
Bigby and Frawley (2010)			✓	✓	✓
Timmons et al. (2011)			✓	✓
Conder et al. (2011)			✓	✓	✓
McVilly et al. (2006)	✓			✓
Nierse and Abma (2011)			✓	✓	✓
Nicolaidis et al. (2011)	✓	✓	✓	✓	✓
Nicolaidis et al. (2013)	✓	✓	✓	✓	✓

ASD: autism spectrum disorder.

Checkmarks indicate the involvement of partner(s) in specified stage of the study development.

Bigby and Frawley (2010)

The authors reported on the study, “Making Life Good in the Community,” which examined factors involved in achieving positive outcomes for people with a disability transitioning from institutional care to a group home. The study comprised qualitative and quantitative components, and data were collected through ethnography and PR using interviews and surveys. The study team included a partner with an ID, who was involved in collecting, analyzing, and disseminating the data.

While it was intended that the partner would be integrally involved as part of the research team, no expectations were reported regarding his role or how he would be supported. The researchers initially treated the partner “as [they would] any other new relatively inexperienced research assistant” (Bigby and Frawley, 2010: 56) and provided him with training on research methodology and included him in meetings (Bigby and Frawley, 2010). A mentor, identified as having both research skills and experience working with people with an ID, was hired to provide practical support to the partner. As the study unfolded, the partner and mentor were contracted to (1) manage field visits to group homes, where they collected, recorded, and transcribed observations about the comfort of different houses; (2) analyze data; and (3) present study findings at various conferences. While the authors noted that conference presentations delivered by the partner enhanced the impact and authenticity of dissemination efforts, no formal evaluation was conducted.

Upon study completion, the research team evaluated the process of involving the partner as part of the research team using Walmsley and Johnson’s (2003) framework of inclusive research. Because the partner was hired a year into the study, and after funding for the study had been awarded, he had no influence on shaping the research questions, methodology, or study design. The study authors noted that the short timelines imposed by grant funding cycles prevented the partner from maintaining a degree of control over the study, although they also acknowledged concerns about maintaining rigor, and their group’s lack of experience in participatory approaches. The authors also noted that the partner’s perspectives were influenced by significant input from the research mentor. As part of the study, during the data collection stage, the partner contributed to a report summarizing his observations of studied group homes. While the research mentor felt that the ideas captured in the report were a product of a collaborative partnership, the unique intellectual contribution of the partner was not delineated. Based on the components outlined, the authors concluded that the study did not conform to an ideal type of PR, as it was neither initiated directly by people with ID, owned by them, nor reflective of their interests and experience (Bigby and Frawley, 2010).

Timmons et al. (2011)

The authors reported on a study aimed at identifying factors that influenced the employment-related decision-making of individuals with intellectual and other developmental disabilities and examined whether individuals’ preferences were consistent with existing employment options and choices. A methodological goal of the study was to fully involve an individual with an ID as a partner (Timmons et al., 2011).

After orienting the partner to the study, the research team developed a training and support plan to integrate her as part of the larger team. With the support of a research mentor, the partner contributed to various stages of the study, including assisting with recruitment of study participants, contributing to data collection by developing the consent procedure, and conducting face-to-face interviews with participants alongside one of the study authors. In addition, the partner participated in data analysis by coding transcripts and creating memos with members of the research team. The authors did not address the extent to which the research mentor supported the partners’ involvement, nor how her specific perspectives were integrated into the overall interpretation of study findings.

Conder et al. (2011)

This article examined a mixed methods study seeking to develop a quality-of-life questionnaire for individuals with ID. The study authors recognized that people who use services are most knowledgeable about how the services work. Using the participatory action framework, they invited six coworkers with an ID (partners) to focus groups identifying quality-of-life indicators for the questionnaire. They also involved the partners in other stages of the research (Conder et al., 2011), such as training partners in research methods and orienting them to the chosen quality-of-life framework. In addition to conducting focus groups, partners were involved in selecting a name for the project, reviewing the consent process with each participant, analyzing data, developing the questionnaire, and disseminating findings via a report of the study findings and a plain language summary. Given the range of tasks led by the partners, a support team provided practical assistance, including booking flights and accommodations for travel to collect data for the study, arranging meetings to answer any questions, and ensuring that the partners were comfortable with their research roles.

Because the partners were involved in the study after the terms of reference, timelines, and budget had been agreed upon, they did not have the opportunity to inform the action steps or the quality-of-life framework selected by the research team. A tight study time frame also limited the research team’s ability to act on suggestions articulated by the partners. Consequently, the partners and other members of the research team acknowledged that the capacity for participation in the study was less for the partners. The study authors noted that the partners contributed to team discussions, particularly in the latter stages of the project, and that their ideas influenced the final product. However, the authors do not provide specific examples of intellectual contributions made by the partners, nor how the partners’ ideas contributed to the overall interpretation of the data. The study authors concluded that the team achieved their goal of having individuals with IDs author a quality-of-life questionnaire, thus ensuring that individuals for whom the instrument was designed would be involved in its development.

McVilly et al. (2006)

The authors conducted a large, multi-method study that, in part, examined the friendship experiences and aspirations of adults with an ID. While the study was not intentionally established within a PR framework, the authors discuss many characteristics of PR that were included in the study’s design—researchers were considered to be experts on the technical process of research, whereas self-advocate partners and study participants were considered to be experts on the subject of friendship.

Self-advocates were initially involved in formulating the topic of inquiry and later took part in the review and analysis of the data. The data analysis stage involved an expert group of 11 self-advocates with an ID who reviewed findings from three phases of data collection (McVilly et al., 2006). The study authors noted that the dynamics of the expert group provided an environment in which participants could debate among themselves issues of relevance to them, rather than entering into a “dyadic discourse with a researcher and consequently needing to negotiate the power imbalances inherent in such a discourse and the potential influence of this power imbalance on the findings” (McVilly et al., 2006: 705). Many of the recommendations for action and further research were from the expert group’s review of study findings.

The study authors noted that the expert group enabled the research team to “assess, evaluate and refine findings based on the views and priorities of people with intellectual disabilities themselves” (McVilly et al., 2006: 706). By following this process, the authors mentioned that they could ensure their own perspective did not exert too much influence over the outcomes of the study and that they could test the validity of the research team’s interpretation of the findings using the collective expertise of a group of adults with IDs. The authors concluded that convening the expert panel was an effective way to review and analyze data and ensure that panel members “had the last say on the topic of inquiry” (McVilly et al., 2006: 706). They also concluded that engaging self-advocates in the expert review group was crucial to the overall investigation and contributed to the depth, breadth, and rigor of the research process.

Nierse and Abma (2011)

The authors reported on a study that aimed to develop a research agenda for people with an ID. Recognizing that the current research did not incorporate the needs of people with IDs or their parents, the project involved two adult partners with a mild ID and the mother of a child with severe ID, who also participated as a partner on the research team. A responsive methodology was used in the agenda-setting process. In the context of this methodology, specific research activities were not determined beforehand, but were developed in consultation with the stakeholders. This allowed the research team to adapt to the issues that emerge from the subjective experiences and stories of stakeholders (Nierse and Abma, 2011). The decision to involve research partners meant that academic researchers “did not act as experts, but as supporters and assistants of the research partners” (Nierse and Abma, 2011: 413). However, the partners were not involved in identifying the research question (funding was secured prior to their involvement), but rather were involved in data collection and in the analysis and dissemination of the results. The research partners recruited participants through personal networks, prepared topic lists for interviews, interviewed participants, moderated or co-moderated focus groups, analyzed transcripts, and participated in the construction of the study’s questionnaire.

The study authors reported that “the active involvement of people with IDs as research partners was important for the entire process” and that it resulted in “co-ownership of the research agenda and empowered both people with ID, who reported benefits, such as improved knowledge and skills, self-esteem and meaningfulness” (Nierse and Abma, 2011: 419). The research team, however, did not pursue a formal evaluation, which might have supported this claim. The study authors also noted that a particular strength among the research partners that enhanced research outcomes was that they were able to engage people in conversation, feel “more empathy,” encourage “much more authentic” responses from participants, and help “participants to share their intimate voice” (Nierse and Abma, 2011: 419). However, the authors did not provide details (e.g. specific criteria or measures) about how these processes were evaluated. Although the authors noted that adults with ID and parents prioritized research differently, no distinction regarding their involvement in the research process was made.

Nicolaidis et al. (2011)

This article reported on a collaborative initiative to build an academic–community partnership that uses community-based participatory research (CBPR) to improve the lives of people with ASD. The misalignment between researchers’ priorities and those of the autistic community, as well as the exclusion of individuals with ASD from the research process, led to the development of the Academic Autistic Spectrum Partnership in Research and Education (AASPIRE). AASPIRE investigates the healthcare experiences and well-being of ASD adults and is composed of health services disability researchers, self-advocates, healthcare providers, disability service professionals, and family members (Nicolaidis et al., 2011).

Nicolaidis et al. (2011) reported that the philosophy espoused by AASPIRE was academic researchers, individuals with ASD, and other community partners (e.g. family members) bring unique and complementary professional and personal expertise and thus should serve as equal partners throughout the entire research process. Community partners ensure that the research is “respectful, accessible and socially relevant,” and academic researchers ensure that research is “scientifically sound and academically relevant, that the work has the proper rigor, and that it advances academic goals” (Nicolaidis et al., 2011: 145). Members of AASPIRE pursue research projects, which align with priority areas identified by individuals with ASD and family members. Furthermore, individuals with ASD are actively involved throughout the development of study and contribute to “designing protocols, developing and adapting instruments and consent materials, recruiting participants, collecting and analyzing data, and disseminating findings” (Nicolaidis et al., 2011: 145).

Individuals with ASD and family members also “inform the group about potentially offensive or unclear language or assumptions” and are involved in selecting and adapting study instruments (protocols, recruitment and consent materials, and interview guides) to ensure they are accessible to participants (Nicolaidis et al., 2011: 146). The collaborative approach offers strategies for conducting CBPR with ASD self-advocates. These strategies are designed to integrate text-based online media and consensus building processes to “successfully equalize power and accommodate the diverse communication and collaboration needs” of self-advocates (Nicolaidis et al., 2011: 143).

Nicolaidis et al. (2013)

This article reported on a collaborative initiative to adapt a previously validated health services questionnaire to assess the healthcare experiences of adults with ASD using a CBPR approach, allowing both researcher and partners to collaborate as equal partners. This article builds upon their collaborative process, AASPIRE, previously detailed in Nicolaidis et al. (2011; see above).

Nicolaidis et al. (2013) report that during the development of an Internet-based survey to understand healthcare experiences for adults with ASD, “academic and community partners collaboratively identified which constructs to measure, chose assessment instruments, and adapted instruments to be more accessible or relevant to autistic adults” (p. 762). The collaborative PR approach led to the first study that “directly obtain[ed] data from autistic adults about their perceptions of unmet healthcare needs, patient-provider communication, and healthcare self-efficacy” (Nicolaidis et al., 2013: 765).

Discussion

The present scoping review revealed that researchers are beginning to implement PR approaches in adults with neurodevelopmental disorders or ASD in a wide range of contexts, using a variety of methodologies. Of the seven studies identified, five described PR partnerships between researchers and partners with IDs, and two studies described partnerships between researchers and partners with ASD. An evaluation of the partnerships revealed that (Table 1) (1) five of the partnerships were initiated by the research team and two were described as co-initiated by researchers and partners; (2) in all but two studies, the partner was recruited after the study was funded and the research question and methodology had been selected; (3) four of the studies used a mixed-method design and the remaining three studies used a qualitative design; (4) in seven of the studies, partners were involved in analyzing data; (5) only two studies listed the partners; and (6) the number of partners involved in the research study ranged from 1 to 11. Overall, it is clear that the extent of the partner’s involvement during the research process varied among the studies reviewed here. The limited involvement of partners in five of these studies (see Table 1) equated to their role as assisting researchers to gain access to certain populations and carrying out tasks determined by study authors, resulting in less opportunity for the partner to inform the study question and design.

Proponents of PR have argued that gathering input from community partners is critical to producing valid and ethical scientific information that is inclusive of the partner’s perspectives (Aman, 2006; Ham et al., 2004; Koch et al., 2001; Kottorp et al., 2003). There are additional benefits to PR, including the facilitation of recruitment of people with neurodevelopmental disorders, the adaptation of measurements and clarification of language to be easily accessible to research participants, and the dissemination of results to members of the partner’s community (Kiernan, 1999; Nicolaidis et al., 2013; Powers et al., 2007). Nevertheless, application of PR can have distinct challenges, as Voegtle (2008) observes, “communication problems and differing goals, values, and work styles” (p. 4) can produce tensions between members of a developing partnership, and the “process is time-consuming” (p. 4; also Stalker, 1998). Other problems may arise as well, such as the ethical considerations of paying partners (Burke et al., 2003; Walmsley and Johnson, 2003), researchers themselves losing objectivity and becoming activists for the partners’ disability (Kiernan, 1999), the expectation of continuing friendships upon completion of the research project (Stalker, 1998), as well as the limited involvement of only those partners who are able to be involved in, and contribute to, the research process (i.e. high-functioning ASD as partners; Kiernan, 1999).

A comparative analysis of the studies revealed three key themes, related to potential limitations in the use and generalizability of the research reviewed. First, there were differences in the extent to which partners were involved in the development of the studies and a lack of clarity regarding the role of the partner in the research process. This concern highlights the importance of defining criteria for evaluating whether a study is truly participatory. To illustrate, of the studies reviewed, only two report that partners were involved throughout the entire research process (Nicolaidis et al., 2011, 2013; see Table 1). Being included as equal collaborators resulted in the examination of relevant questions that were reported within a context that is easily understood by the partners’ population; this is the ideal for PR partnerships. In contrast, one report (Timmons et al., 2011) only included partners during data collection and analyses. The lack of involvement of the partner during the development of the research question, and arguably the more important step of identifying and adapting research assessments for ease of participant consumption, may result in participant confusion and/or lack of correct response; this is the less-than ideal for PR partnerships. The potential for partners to enhance the process and outcomes of research by being involved in selecting methods has been reported in the broader PR literature. More specifically, the participation of partners in methods selection has been reported to increase the validity of a study by increasing the depth and variety of data collected, establishing congruence between a research question and a local reality, and adapting study methodology to specific contexts (Macaulay et al., 1999; Sandoval et al., 2012; Viswanathan et al., 2004).

A second theme identified was the limited reporting of the depth of the partners’ involvement across studies, including a lack of clarity on how multiple perspectives (researchers and partners) were integrated into the decision-making process. There were varying efforts across the studies to involve partners in the research process (Table 1); however, it was generally not made explicit how the multiple perspectives from “around the table,” including that of the partner, were integrated into study decision-making. With the exception of two studies, it was unclear how dissenting views were integrated into an overall perspective and how consensus was reached in decision-making throughout the course of the studies. Because of the lack of reporting, the unique intellectual contributions of the partners at various stages of the research studies were not explicitly acknowledged. Moreover, this review revealed a lack of clarity in the extent to which a partner influenced the overall development of a study, informed conclusions, or contributed to the research study outcomes.

A final theme that emerged in this review is the question of what differentiates a “partner” from a research assistant. More specifically, in five of the studies, where the partners were assigned tasks by researchers, the boundary was not clearly drawn in terms of what is considered PR and what might be considered hiring someone with a disability to assist with a study. Whereas a research assistant may carry out technical aspects of a study, previous literature on PR (Jagosh et al., 2011; Macaulay et al., 1999) suggests that a partner, as part of the investigative team, should be involved in framing questions, interpreting results, and putting them into context (Nind, 2008). With the exception of two studies, there was an emphasis on the partners’ involvement in operational tasks (e.g. data collection) rather than in higher order aspects of the research process (e.g. analyzing data), which speaks to a hierarchy within the research team and a narrow view of “participation.” Wider use of evaluative tools including the framework we used to assess the breadth, depth, and outcomes of PR partnerships included in this review (Figure 2) may provide a fuller understanding of the degree to which the partner is contributing to every step, ultimately determining how genuine the partnership is (Blackstock et al., 2007). Furthermore, increasing the responsibility for researchers to specify how partners will be involved throughout a study in funding applications may help to prevent researchers from citing “funding already in place” as a rationale for not involving partners from the outset (e.g. Bigby and Frawley, 2010; Nierse and Abma, 2011).

Figure 2.

Framework for assessing breadth, depth, and outcomes of PR partnerships with individuals with ASD and other neurodevelopmental disorders.

Because neurodevelopmental disorders, including ASD, encompass a broad range of impairments, including impairments to speech and language, social relationships, memory, and learning, there are challenges to including them as partners in PR. These impairments may limit the degree to which individuals can be involved in certain activities (e.g. tasks that require abstract reasoning). Nevertheless, previous research has demonstrated that people with neurodevelopmental disorders and ASD can participate as partners in research (Gilbert, 2004; Walmsley, 2004). The research reviewed here highlights the benefits and drawbacks of including individuals with ASD and other neurodevelopmental disorders in PR. For example, Bigby and Frawley (2010) note that presentations delivered by the partner enhanced the impact and authenticity of the research results; the inclusion of partners allowed the assessment, evaluation, and results to reflect the views and priorities of people with ID (McVilly et al., 2006); and partners help to ensure that research is “respectful, accessible and socially relevant” to the group being examined (Nicolaidis et al., 2011: 145). Nevertheless, it is important to ensure that partners are involved from the beginning, as lack of partner involvement in the development of the research question and methodology employed results in the outcomes not being owned by them, nor reflect their interests or experiences (Bigby and Frawley, 2010; Conder et al., 2011). The two reports by Nicolaidis et al. (2011, 2013) demonstrate how PR research should be completed, allowing for a feeling of authenticity and relevance of the research and exemplifying the necessity of adults with ASD or other neurodevelopmental disorders to be included as partners in PR.

There are limitations to this scoping review. Ultimately, our findings are limited by the information available in the published literature, and in particular, how the roles of partners were described and evaluated. Another limitation is the lack of perspectives and experiences of the PR partnership as described by the partner. Given the communication delays common in neurodevelopmental disorders and ASD, further exploration is warranted addressing how to support such individuals in articulating their perspective on research so as to potentially improve and/or optimize PR partnerships in the field. As such, this review calls for future research in this field to more specifically evaluate the contributions of partners with ASD or other neurodevelopmental disorders, and for the creation and testing of tools to assess participatory structures of research engagement for this population. Ultimately, PR research may contribute to improvement in research outcomes, but that remains to be demonstrated.

Conclusion

Several studies over the past decades have attempted to involve individuals with neurodevelopmental disorders or ASD as partners in research. Although this inclusion of partners in research is appealing from an empowerment perspective, this review revealed gaps in how the process and outcomes of such PR partnerships are reported. More specifically, further clarity on the extent to which partners were involved in the development of the study, the roles and responsibilities they undertook relative to other research partners, how their perspectives were integrated into decision-making, and how the partnerships were evaluated is called for. This review highlights the need for an evaluative framework in PR studies, both for the populations reviewed here and beyond. A balance needs to be developed, allowing partners to provide the necessary expertise to aide in the development and implementations of the overarching academic agenda.

Footnotes

Appendix 1 Funding

This research is supported by CIHR and Autism Speaks Canada. LZ is supported by the Stollery Children’s Hospital Foundation Chair in Autism Research and an Alberta Innovates-Health Solutions Scholar Award. JJ and LRS were supported by a CIHR Autism Research Training Program Fellowship award.

References

Abma

Nierse

Widdershoven

GAM

(2009) Patients as partners in responsive research: methodological notions for collaborations in mixed research teams. Qualitative Health Research 19(3): 401–415.

Aman

(2006) Reactions to “ethical challenges and complexities of including people with intellectual disability as participants in research” by Dr. Teresa Lacono. Journal of Intellectual & Developmental Disability 31(3): 180–182.

Arksey

O’Malley

(2005) Scoping studies: towards a methodological framework. International Journal of Social Research Methodology 8: 19–32.

Atkinson

Walmsley

(1999) Using autobiographical approaches with people with learning difficulties. Disability & Society 14(2): 203–216.

Atkinson

Williams

(1990) Know Me as I Am: An Anthology of Prose, Poetry, and Art by People with Learning Difficulties. Sevenoaks: Hodder & Stoughton.

Bigby

Frawley

(2010) Reflections on doing inclusive research in the “Making life good in the community” study. Journal of Intellectual & Developmental Disability 35(2): 53–61.

Blackstock

Kelly

Horsey

(2007) Developing and applying a framework to evaluate participatory research for sustainability. Ecological Economics 60(4): 726–742.

Boote

Telford

Cooper

(2002) Consumer involvement in health research: a review and research agenda. Health Policy (Amsterdam, the Netherlands) 61(2): 213–236.

Brookman-Frazee

Stahmer

Lewis

(2012) Building a research-community collaborative to improve community care for infants and toddlers at-risk for autism spectrum disorders. Journal of Community Psychology 40(6): 715–734.

10.

Burke

McMillan

Cummins

(2003) Setting up participatory research: a discussion of the initial stages. British Journal of Learning Disabilities 31(2): 65–69.

11.

Cargo

Mercer

(2008) The value and challenges of participatory research: strengthening its practice. Annual Review of Public Health 29: 325–350.

12.

Conder

Milner

Mirfin-Veitch

(2011) Reflections on a participatory project: the rewards and challenges for the lead researchers. Journal of Intellectual & Developmental Disability 36(1): 39–48.

13.

Cornwall

Jewkes

(1995) What is participatory research? Social Science & Medicine 41(12): 1667–1676.

14.

Gauld

Smith

Kendall

(2011) Using participatory action research in community-based rehabilitation for people with acquired brain injury: from service provision to partnership with Aboriginal communities. Disability and Rehabilitation 33(19–20): 1901–1911.

15.

Giachello

Arrom

Davis

(2003) Reducing diabetes health disparities through community-based participatory action research: the Chicago Southeast Diabetes Community Action Coalition. Public Health Reports 118(4): 309–323.

16.

Gilbert

(2004) Involving people with learning disabilities in research: issues and possibilities. Health & Social Care in the Community 12(4): 298–308.

17.

Green

Mercer

(2001) Can public health researchers and agencies reconcile the push from funding bodies and the pull from communities? American Journal of Public Health 91(12): 1926–1929.

18.

Ham

Jones

Mansell

(2004) “I’m a researcher!” Working together to gain ethical approval for a participatory research study. Journal of Learning Disabilities 8(4): 397–407.

19.

Israel

Schulz

Parker

(1998) Review of community-based research: assessing partnership approaches to improve public health. Annual Review of Public Health 19: 173–202.

20.

Jagosh

Pluye

Macaulay

(2011) Assessing the outcomes of participatory research: protocol for identifying, selecting, appraising and synthesizing the literature for realist review. Implementation Science 6: 24.

21.

Keightley

Ratnayake

Minore

(2009) Rehabilitation challenges for Aboriginal clients recovering from brain injury: a qualitative study engaging health care practitioners. Brain Injury 23(3): 250–261.

22.

Kiernan

(1999) Participation in research by people with learning disability: origins and issues. British Journal of Learning Disabilities 27: 43–47.

23.

Koch

Marks

Tooke

(2001) Evaluating a community nursing service: listening to the voices of clients with an intellectual disability and/or their proxies. Journal of Clinical Nursing 10(3): 352–363.

24.

Kottorp

Bernsang

Fisher

(2003) Validity of a performance assessment of activities of daily living for people with developmental disabilities. Journal of Intellectual & Developmental Disability 47(Pt 8): 597–605.

25.

Macadam

Sutcliffe

(1996) Still a Chance to Learn? A Report on the Impact of the Further and Higher Education Act (1992) on Education for Adults with Learning Difficulties. Leicester: National Institute for Continuing Adult Education.

26.

Macaulay

(2007) Promoting participatory research by family physicians. Annals of Family Medicine 5(6): 557–560.

27.

Macaulay

Commanda

Freeman

(1999) Participatory research maxmises community and lay involvement. North American Primary Care Research Group. British Medical Journal 319(7212): 774–778.

28.

Macaulay

Ing

Salsberg

(2007) Community-based participatory research: lessons from sharing results with the community: Kahnawake Schools Diabetes Prevention Project. Progress in Community Health Partnerships: Research, Education, and Action 1(2): 143–152.

29.

Macaulay

Jagosh

Seller

(2011) Assessing the benefits of participatory research: a rationale for a realist review. Global Health Promotion 18(2): 45–48.

30.

McKevitt

Fudge

Wolfe

(2010) What is involvement in research and what does it achieve? Reflections on a pilot study of the personal costs of stroke. Health Expectations: An International Journal of Public Participation in Health Care and Health Policy 13(1): 86–94.

31.

March

Steingold

Justice

Mitchell

(1997). Follow the yellow brick road! People with learning difficulties as co-researchers. British Journal of Learning Disabilities 25: 77–80.

32.

McVilly

Stancliffe

Parmenter

(2006) Self-advocates have the last say on friendship. Disability & Society 21(7): 693–708.

33.

Marcus

Walker

Swint

(2004) Community-based participatory research to prevent substance abuse and HIV/AIDS in African-American adolescents. Journal of Interprofessional Care 18(4): 347–359.

34.

Nicolaidis

Raymaker

McDonald

(2011) Collaboration strategies in nontraditional community-based participatory research partnerships: lessons from an academic-community partnership with autistic self-advocates. Progress in Community Health Partnerships: Research, Education, and Action 5(2): 143–150.

35.

Nicolaidis

Raymaker

McDonald

(2013) Comparison of healthcare experiences in autistic and non-autistic adults: a cross-sectional online survey facilitated by an academic-community partnership. Journal of General Internal Medicine 28(6): 761–769.

36.

Nierse

Abma

(2011) Developing voice and empowerment: the first step towards a broad consultation in research agenda setting. Journal of Intellectual Disability Research 55(4): 411–421.

37.

Nind

(2008) Conducting qualitative research with people with learning, communication and other disabilities: methodological changes. Project Report, ESRC National Centre for Research Methods, Southampton, November.

38.

Oliver

Clarke-Jones

Rees

(2004) Involving consumers in research and development agenda setting for the NHS: developing an evidence-based approach. Health Technology Assessment (Winchester, England) 8(15): 1–148.

39.

Pellicano

Stears

(2011) Bridging autism, science and society: moving toward an ethically informed approach to autism research. Autism Research: Official Journal of the International Society for Autism Research 4(4): 271–282.

40.

Powers

Garner

Valnes

(2007) Building a successful adult life: findings from youth-directed research. Exceptionality: A Special Education Journal 15(1): 45–56.

41.

Royle

Oliver

(2004) Consumer involvement in the health technology assessment program. International Journal of Technology Assessment in Health Care 20(4): 493–497.

42.

Sandoval

Lucero

Oetzel

(2012) Process and outcome constructs for evaluating community-based participatory research projects: a matrix of existing measures. Health Education Research 27(4): 680–690.

43.

Stalker

(1998) Some ethical and methodological issues in research with people with learning difficulties. Disability & Society 13(1): 5–19.

44.

Timmons

Hall

Bose

(2011) Choosing employment: factors that impact employment decisions for individuals with intellectual disability. Intellectual and Developmental Disabilities 49(4): 285–299.

45.

Viswanathan

Ammerman

Eng

(2004) Community-based participatory research: assessing the evidence. Evidence Report/Technology Assessment (Summary) 99: 1–8.

46.

Voegtle

(2008) A Systematic Review of Community-Based Participatory Research Interventions in Pediatric and Adolescent Populations (1457569 M.P.H.). Ann Arbor, MI: The University of Texas School of Public Health. Available at: http://search.proquest.com/pqdtft/docview/304471741/1416BAA1A0C21E614F1/1?accountid=14474

47.

Walmsley

(2001) Normalisation, emancipatory research and inclusive research in learning disability. Disability & Society 16(2): 187–205.

48.

Walmsley

(2004) Involving users with learning difficulties in health improvement: lessons from inclusive learning disability research. Nursing Inquiry 11(1): 54–64.

49.

Walmsley

Johnson

(2003) Inclusive Research with People with Learning Disabilities: Past, Present, and Futures. London: Jessica Kingsley.

50.

Williams

(1990) Networks, Workbook 2 of Learning Disability: Changing Perspectives. Buckingham: Open University.

51.

Yonas

Jones

Eng

(2006) The art and science of integrating Undoing Racism with CBPR: challenges of pursuing NIH funding to investigate cancer care and racial equity. Journal of Urban Health 83(6): 1004–1012.