Patients play an essential role in health research, given their unique lived experiences and expertise, which can significantly enhance the quality, relevance, and impact of research by aligning it with patient needs, values, and preferences [1,2]. The overarching objective of patient engagement in health research is to generate research that contributes to improved health care service delivery, clinical outcomes, and population health [3]. Recent research has highlighted the myriad benefits and positive outcomes associated with patient engagement for patient partners, investigators, and research endeavors [4-8]. For example, engaging patients has influenced initial research priorities, study designs, interpretation of findings, health care interventions, and knowledge mobilization strategies, resulting in outcomes that more closely align with patient perspectives [7,9]. When patient engagement is characterized as meaningful and authentic, it provides a rewarding experience for patients and researchers alike [10].

However, other recent research has highlighted various challenges and risks associated with patient engagement in health research. These challenges, which include tokenism [11-14], power imbalances/dynamics [12,15], limited accessibility to patient-friendly training for patient partners [16], and the absence of equitable engagement tools [17], can have lasting effects on patient partners, including mental and physical exhaustion, deteriorating health, doubts about the value of engagement, and a sense of having personally failed both the team and the broader patient community [18]. A recent article by patient partners led by Richards et al [18] on how patient engagement can falter highlighted key themes, including “patient partners as a checkmark,” “unconscious bias towards patient partners,” “lack of support to fully include patient partners,” and “lack of recognizing the vulnerability of patient partners” [18].

To address these challenges and risks, various patient engagement models, theories, frameworks, and guiding principles (eg, the strategy for patient-oriented research [SPOR], the patient engagement in research [PEIR] framework, and “ways community members can participate in the stages of research” from the Ontario Brain Institute) have been developed and adopted within the research community [10,19,20]. For example, the PEIR framework [10] includes eight key components, which collectively contribute to meaningful patient engagement in research: (1) procedural requirements, (2) convenience, (3) contributions, (4) support, (5) team interaction, (6) research environment, (7) feeling valued, and (8) benefits. A subsequent measure of meaningful patient engagement in research from the patient perspective has been developed, the Patient Engagement in Research Scale (PEIRS-22) [21,22]. This measure is designed to be completed by adult patients and family caregivers who partner with researchers on projects.

It is important to acknowledge that existing frameworks often predominantly emphasize the benefits of patient partners to the research project and team, sometimes overlooking the reciprocal benefits that may occur between the research team and patient partners, especially youth partners [23]. This skewed approach fails to fully consider the potentially extractive nature of research collaboration, as aptly described by Metz and Damschroder [23], where the research process can unintentionally exploit the knowledge and contributions of youth without adequately reciprocating in terms of personal and professional development opportunities. This issue takes on particular significance when crafting an engagement framework tailored for youth, as research teams have the potential to foster skill development, positively impact their life and career trajectories, and contribute to their holistic growth [23].

Despite the significant contribution of the PEIR framework [10] and the PEIRS-22 [21,22], they lack the incorporation of a comprehensive review of the evidence on existing frameworks, and their development was based on participants with limited diversity in terms of gender, race, education, primary diagnosis (all had arthritis), and age [10,21,22]. These limitations impede the transferability of this framework and measure to youth- and family-specific engagement in health research.

Furthermore, there is a model of engagement specifically designed for use with youth called the McCain model of youth engagement [24]. However, it was developed solely in the context of youth mental health systems research for youth and young adults aged 15 to 29 years [24], limiting transferability (ie, across different contexts and family members) and highlighting the need to develop a broader framework. Accordingly, our research seeks to directly address these specific gaps in existing engagement models, theories, and frameworks by developing a youth and family-specific engagement in research framework, the UNITE framework.

This proposal has two objectives: (1) to determine the extent of literature on the application of patient engagement models, theories, frameworks, and guiding principles in the context of youth- and family-specific research; and (2) to understand how meaningful the key components and constructs of these models, theories, frameworks, and guiding principles are to youth and their family members. Collectively, these findings will be foundational to the development of the UNITE framework and a subsequent validated measure.

This study will be conducted in 2 phases, with phase 1 consisting of a scoping review and phase 2 consisting of a qualitative descriptive study.

Integrated knowledge translation (iKT) is defined as a collaborative relationship between researchers and relevant knowledge users as partners that facilitates mutually beneficial decision-making related to a study or research program [25]. Youth aged 10 to 24 years in Canada and their family members who have engaged in health research (ie, as patient partners) in the last 3 years were recruited as iKT panel members via Holland Bloorview Kids Rehabilitation Hospital’s Youth Advisory Council and Family Leader Program, professional networks, social media pages, and email lists. A sample of diverse youth advisors who have participated in our previous research on best practices in youth engagement were also approached, including youth with disabilities or developmental differences [26]. A total of 6 youths and 3 family members were recruited to be part of the iKT panels. The lead youth representative will lead the iKT panels. The lead youth representative and research team decided to hold separate iKT panels (ie, having a youth panel and a family panel with the opportunity to mix panels when needed). This approach will ensure that both groups feel comfortable and can freely express their unique perspectives. Activities of the panels have included planning study activities, and may include participant recruitment, data collection and analyses, and knowledge mobilization. Panel discussions will be conducted in a manner that respects diverse perspectives and experiences. A reflective exercise on equity, diversity and inclusion developed by the Strategy for Patient-Oriented Research Evidence Alliance will be conducted with iKT panel members to encourage dialogue and understanding around equity, diversity and inclusion [27].

The methodology for the scoping review will follow the methodological frameworks of the Joanna Briggs Institute (JBI) [28] and Khalil et al [29]. The scoping review protocol has been registered on the Open Science Framework Registries [30] and was guided by and reported according to the Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P) [31]. The completed PRISMA-P checklist can be found in Multimedia Appendix 1. The results of the scoping review will be reported using the Preferred Reporting Items for Systematic Reviews and Meta-Analysis extension for Scoping Reviews (PRISMA-ScR) checklist [32].

The findings from this study will allow us to identify what is currently known about the application of patient engagement models, theories, frameworks, and guiding principles, which are often designed for adults, in the context of youth-specific research, and to understand the importance of the components that make up these models, theories, and frameworks from the perspective of youth and their families.

We will use a variety of passive and active end-of-grant knowledge mobilization approaches to disseminate our findings, which will be codeveloped with our iKT panels. We will ensure youth and family voices are heard to develop flexible communication plans that will suit diverse needs. Traditional knowledge translation will include dissemination through meetings locally, nationally, and internationally (eg, PxP and For Patients, By Patients) and publications in peer-reviewed journals. Members of the research team are affiliated with and situated within pediatric institutions where the UNITE framework will be disseminated and implemented. We will codevelop plain language summaries with youth and family partners with clear, simple, and individualized messages for patients and family and community service organizations to augment the accessibility of the information. Finally, members of the research team will also discuss and distribute the UNITE framework within their expansive training curriculum and a planned youth-focused engagement in research course.

This project lays the foundational work for developing a patient engagement framework called the UNITE framework, which will include equity, diversity, and inclusion considerations, and the subsequent development and validation of an associated measure of engagement. Future research will involve the implementation of the UNITE framework and a subsequent measure in a learning health system context within pediatric institutions that our research teams are associated with. The UNITE framework and associated measure will contribute to meaningful and sustained engagement of youth and their families in health research by addressing gaps in current patient engagement frameworks.

A strength of this study is the inclusion of gray literature (eg, reports and policy literature) for phase 1 (the scoping review). This will provide a more comprehensive understanding of the concept (patient engagement in research) and context (models, theories, frameworks, and guiding principles) that we are interested in, as well as mitigate publication bias [56]. In addition, the search strategy for phase 1 has been reviewed by the iKT panel and undergone peer review using the PRESS statement checklist [34], which further strengthens the relevance, comprehensiveness, and quality of the search strategy. One limitation of this project is the potential for selection bias (specifically for phase 2), where individuals who had either very positive or very negative experiences with patient engagement in research may be more likely to participate in the study, which may limit the applicability of the study’s findings. However, the adoption of the iKT approach and having the iKT panels (consisting of youth and their families) assist in the recruitment for phase 2 of the study should result in the recruitment of a variety of individuals with diverse interests.

The current proposal will lead to the development of a youth- and family-specific engagement in research framework, UNITE, with future research focused on the development of an associated validated measure. The UNITE framework and measure will lay the foundation for meaningful and sustained engagement in health research by youth and their families, ultimately contributing to enhanced health care service delivery, improved clinical outcomes, and increased overall well-being and quality of life for youth and their families [1-3].