Shaping Retinoblastoma Research Development Through Patient Engagement

Sunday November 11, 2018

Parents and survivors have valuable lived experience that can shape retinoblastoma research to improve health outcomes. Kaitlyn Flegg reviews progress of the innovative Canadian Retinoblastoma Research Advisory Board, where parent and survivor priorities are setting the research agenda.

The Key Role of Patients, Parents and Survivors

The important role of patients in research, beyond merely serving as research subjects, started to be acknowledged in the late 1970s.1 Today, researchers are encouraged to partner with patients in all areas of the knowledge translation process.

A full circle links six labelled blocks. Inside the circle, a blue clockwise arrow labelled “knowledge creation” transitions into a pink clockwise arrow labelled “knowledge action”. Blocks 1-4 are coloured blue and correspond to the Knowledge Creation arrow. Blocks 5-6 are coloured pink and correspond to the Knowledge Action arrow. Block Labels: 1) Knowledge question. 2) Research planning & design. 3) Data collection & analysis. 4) Tailoring & telling others about results. 5) Changing practice. 6) Re-evaluate & sustain knowledge.

Figure 1. The Knowledge Translation Process 2

The Canadian Strategy for Patient Oriented Research defines patient engagement as the meaningful and active collaboration of patients in the governance, priority setting and conduct of research, as well as in summarizing, distributing, sharing and applying its resulting knowledge. 3,4 In this context, ‘patient’ refers to individuals with personal experience of a health issue and their informal caregivers, including family and friends. 4

Benefits of patient engagement include:

  • Alignment of limited research funds to patient priorities;
  • Improved quality of research results; and
  • Accelerated uptake of research findings and, in turn, improved patient outcomes.

The Canadian Retinoblastoma Patient Engagement Strategy:

Dr. Helen Dimaras and her research team developed the Canadian Retinoblastoma Patient Engagement Strategy in 2016. The aim of the strategy is to: i) include a large diverse group of people affected by retinoblastoma in research; ii) share research results with people affected by retinoblastoma; and iii) promote research that is created and led by people affected by retinoblastoma. The Canadian Retinoblastoma Research Advisory Board (CRRAB), a national multidisciplinary group, leads the strategy.

Establishing the Top 10 Retinoblastoma Research Priorities:

CRRAB collectively agreed that an early objective of the strategy was to identify the top 10 retinoblastoma research priorities in Canada.

Inspired by the James Lind Alliance Priority Setting Protocol methodology, a three-step project was initiated at The Hospital for Sick Children (SickKids, Toronto, Ontario), late 2017.

  • Step 1 (October 2017): Using an online survey, patients, clinicians and researchers were invited to share questions about retinoblastoma that they would like to see answered by research.
  • Step 2 (November 2017): A national steering committee conducted an interim priority setting activity to reach a list of 30 retinoblastoma research questions.
  • Step 3 (December 2017): An in-person workshop was conducted with a 1:1 ratio of patients and non-patients (clinicians and researchers). Following the Nominal Group Technique, participants narrow down the questions from step 2, reaching the “Top 10” retinoblastoma research priorities.
A group of people gather around a sign that says "Retinoblastoma Research Symposium, Saturday December 9 - Sunday December 10. Registration, 3rd floor lobby"

In-Person Priority Setting Workshop Participants

The final results from this study will be published in the coming months and available on Dr. Dimaras’ lab website. Highlights from the study include:

  • More than 150 retinoblastoma research questions were suggested.
  • The categories with the greatest number of questions were genetics and molecular, second cancer and psychosocial.
  • The most highly prioritized area of retinoblastoma research in Canada was early diagnosis.

Shaping the Canadian Retinoblastoma Research Agenda:

The impact of research priority setting exercise is dependent on endorsement by funders, clinicians, patients and researchers. To this end, Dr. Dimaras hired a Research Coordinator with lived retinoblastoma experience to:

  • Oversee a project that will create, and widely disseminate, educational materials about the top 10 retinoblastoma research priorities.
  • Coordinate a multidisciplinary team in the development of a research proposal that will address one of the top 10 research priorities.

We look forward to deploying these projects in partnership with all of our stakeholders – including patients and parents.

Follow the Canadian Retinoblastoma Research Advisory Board


About the Author

Kaitlyn Flegg, MSc is the Retinoblastoma Research Program Manager at SickKids. Providing support to Dr. Helen Dimaras (Scientist and Director of Global Eye Health Research) and Dr. Brenda Gallie (Head, Retinoblastoma Program). Kaitlyn is committed to meaningful patient engagement and passionate about improving outcomes for people affected by retinoblastoma.

Follow Kaitlyn on Twitter

Kaitlyn Flegg