Development of a Patient-Centered Pathway of Care for Retinoblastoma Patients: A Mixed-Methods Pilot Study.

Introduction Retinoblastoma treatment and follow-up is complex and varies between patients. Pathways of care can enhance quality of care, patient outcomes, safety, satisfaction, and resource optimization. Developing a pathway of care for retinoblastoma was identified as a top research priority by the retinoblastoma community. This study aimed to co-design and pilot a pathway of care called the "Retinoblastoma Journey Map" tailored for caregivers of newly diagnosed children with retinoblastoma. Methods A working group of patients, health professionals and researchers used human-centred design to ideate, prototype and refine the Retinoblastoma Journey Map. Caregivers of affected children were recruited to use and evaluate the Map. Mixed-methods data was collected on feasibility, acceptability, usability and perceived impact on communication, self-efficacy, anxiety, depression, and the quality of physician-patient interaction. Results The Retinoblastoma Journey Map consisted of an illustrated roadmap with 25 child-friendly stickers covering clinical treatment, medical education and milestones. Quantitative analysis revealed that the Map was feasible, acceptable, and usable; however, no significant effect on communication, self-efficacy, anxiety, depression or quality of physician-patient interaction was observed. Qualitative analysis identified 6 themes: Primary Use, Challenges, Impact, Limitations, Feasibility, Acceptability and Usability, and Unmet Needs. Conclusion A pathway of care for retinoblastoma, co-designed by researchers, health professionals and patients, was usable, acceptable, and feasible by caregivers of children with retinoblastoma. While significant effects on communication and physician-patient interaction were not observed, 'legacy building' - documentation of the pathway of care by families for later education of their child - emerged as an unanticipated yet important use of the Retinoblastoma Journey Map.

EMPOWER Retinoblastoma: Engaging Patient Partners in Solving the Top 10 Priorities for Eye Cancer Research in Canada.

While it is recognized that research priorities should reflect and integrate the perspectives and needs of patients along with those of health professionals and researchers, it remains challenging to actualize such priorities into tangible research projects. Targeted dissemination is required to catalyze research on these priorities. To create awareness of and inspire action toward actualizing the top 10 retinoblastoma research priorities in Canada, Canadian Retinoblastoma Research Advisory Board (CRRAB) members developed a wide range of dissemination tools and processes. These resources, co-produced with patients, were instrumental to CRRAB sharing the top 10 priorities internationally to mobilize action toward solving them.

The Retinoblastoma Research Booklet: A Catalyst for Patient Involvement in Retinoblastoma Research.

Peer-to-peer recruitment efforts are important in generating interest and participation of patients as partners in research but difficult to sustain when face-to-face interactions are limited. The Retinoblastoma Research and You! booklet, co-developed by patients, researchers and health professionals, serves as a guide for patient engagement in research while retaining an element of personalization. The Retinoblastoma Research and You! booklet was developed through two virtual workshops to iterate and finalize the booklet design and content. The booklet outlines how individual patients' lived experiences and skills can influence retinoblastoma research and highlights real-world examples of patient-partnered research activities at different stages of the research process.

The Canadian retinoblastoma research advisory board: a framework for patient engagement.

PLAIN ENGLISH SUMMARY: Retinoblastoma is a rare eye cancer that occurs in one or both eyes of infants and young children as a result of errors in the RB1 gene. There are approximately 2000 retinoblastoma survivors in Canada. Those with the heritable form of the disease are at risk of passing the gene to the next generation and developing a second cancer. Many retinoblastoma survivors and families therefore interact with the healthcare system throughout their lives.The retinoblastoma community has a longstanding history of engaging patients in research, however without any formal process. The literature establishes benefits to patient engagement such as research results which are more applicable, credible, and transparent. Building on the established interest among stakeholders, the Canadian Retinoblastoma Research Advisory Board (CRRAB) was established in 2016 to foster sustainable and meaningful collaboration between patients (survivors and family members), advocacy groups, healthcare professionals, and researchers in the retinoblastoma community.The aim of this study was to evaluate the utility of CRRAB in fostering patient engagement in research. Members of CRRAB were surveyed to uncover their attitudes towards and experience with patient engagement in research. Participants perceived CRRAB to provide diverse and accessible opportunities for patient engagement in research and perceived their participation to have a meaningful impact. The results suggest that CRRAB promotes patient engagement in retinoblastoma research, and provides direction to sustain and enhance future patient engagement. ABSTRACT: Background The Canadian Retinoblastoma Research Advisory Board (CRRAB) is a multidisciplinary group, including patients (survivors and family members), advocacy groups, healthcare professionals, and researchers, which aims to establish and sustain patient engagement in retinoblastoma research. The purpose of this study was to describe the development of CRRAB and to uncover members' understanding of and attitudes towards patient engagement in research. As well, to determine their level of engagement.Methods Retinoblastoma patients, healthcare professionals, and researchers provided leadership to co-develop CRRAB. CRRAB members were surveyed by pre- and post-test questionnaire at the 2016 Annual General Meeting to assess experience with, understanding of, and attitudes towards patient engagement in research. A second questionnaire was administered before the 2017 CRRAB meeting to assess awareness and perceived impact of CRRAB activities, and individual engagement in research. Data were analyzed by descriptive statistics and paired t-test (for pre/post-test). Thematic analysis of chart board discussions at both meetings revealed the joint goals of CRRAB and reasons for and barriers to patient engagement.Results In 2016, 21 individuals participated and self-identified as patients (11, 52%), healthcare professionals (6, 29%), and/or researchers (7, 33%) (participants could overlap stakeholder groups). Overall, participants believed that research is relevant to all stakeholders and that patients can have meaningful impact on research. In 2017, 35 individuals participated and identified as patients (21, 60%), healthcare professionals (9, 26%), and/or researchers (8, 23%). 94% of participants were aware of CRRAB initiatives and 67% had participated in at least one over the previous year. Participants perceived that CRRAB provides diverse opportunities and increases accessibility for patient engagement in research, and perceived patient engagement to have meaningful impact on retinoblastoma research. Chart board discussions revealed that participants wanted to be part of CRRAB to increase knowledge, support innovation and patient engagement, and be part of a community. Members most commonly faced barriers including time and cost restraints.Conclusions The results of this study suggest that CRRAB has supported the engagement needs of patients affected by retinoblastoma, and has provided an opportunity for engaging patients in retinoblastoma research. CRRAB will continue to be used as a framework for patient engagement, with improvements based on participant feedback.