Plain language summary: A national study of patients with retinoblastoma (childhood eye cancer) and their family members and friends, clinicians and researchers was conducted in Canada to answer the question "What are the top 10 retinoblastoma research priorities in Canada?" The method used was an adaptation of the James Lind Alliance Priority Setting Partnership process, commonly used in such joint priority-setting initiatives. The top priority identified related to early diagnosis of retinoblastoma. Advocacy groups, research teams and funding agencies are encouraged to align their practices with the identified retinoblastoma research priorities. R esearch priority setting identifies the research most relevant to and valued by stakeholders. This is neces sary given that most funded research does not reflect the priorities of patients and clinicians, potentially reducing its impact. 1,2 There are several established methods for research priority setting; the James Lind Alliance Priority Setting Part nership (PSP) method 3 is arguably the most popular. The James Lind Alliance PSP method involves patients and clin icians equally in setting a top 10 list of research priorities. Retinoblastoma is a cancer of the infant retina usually caused by a biallelic RB1 gene mutation. 4 About 45% of patients with retinoblastoma have the heritable form, meaning they carry a constitutional RB1 mutation that confers risk of second cancers later in life and can be passed on to offspring. Each year, 8000 children are newly diagnosed with retinoblas toma globally, about 24 of whom are in Canada. 4
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.
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