Author: Ana Janic, MSc
In 2018, I was welcomed onto a diverse, interdisciplinary team of dedicated retinoblastoma researchers. At that time, I was just starting my graduate studies and, I must admit, knew little about retinoblastoma and even less about patient engagement. However, my supervisor, Dr. Helen Dimaras, quickly offered extensive insight into the retinoblastoma universe. Almost overnight I found myself immersed in a huge community of retinoblastoma researchers, healthcare professionals, and patients who strongly engage in research.
Over the past two years, Dr. Dimaras and I worked together on an incredible project that is rooted in patient advocacy and encourages patient-focused evaluation of disease. Patient-reported outcome measures (PROMs) are questionnaires, completed by patients, that provide subjective information on various health concepts, like a patient’s quality of life, evaluation of symptoms, and physical, mental, or social well-being. Within a PROM, questions are grouped into categories, referred to as domains, which reflect aspects of the chosen health concept(s). PROMs are developed in multiple phases, with robust patient involvement, and take years to complete; my project represents the initial steps in the development of a PROM to measure retinoblastoma treatment outcomes.
The primary goal of my project was to uncover which treatment outcomes are important and relevant to retinoblastoma survivors and parents of survivors. To do this, I held focus groups with adult survivors and parents of survivors. Further, we decided to interview pediatric survivors between the ages of 6 and 17 years old. Figuring that survivors in this age range could complete a retinoblastoma-specific PROM by themselves, we felt it imperative to identify the valued treatment outcomes in this demographic in order to develop a comprehensive and relevant questionnaire.
Focus group discussions with pediatric participants was a new method for us, therefore, we consulted several scientific resources to ensure our methods were sound. One of our greatest feats was developing an easy-to-understand interview guide for child survivors.
First, we started by mirroring the interview guide used in adult groups. The pediatric interview guide included the same question categories as the adult interview guide, however, question language was modified to adjust for comprehension. For instance, when discussing psychological and emotional outcomes, adult survivors and parents of survivors were asked:
“Does a good emotional state indicate successful treatment? What is a good emotional state?”
The question was modified for pediatric survivors to read:
“Some things make us happy, and other things make us uncomfortable and sad. Do you think that retinoblastoma, or retinoblastoma treatment, can make you happy or sad?”
After question modification, two pediatric interview guides emerged: one for child survivors aged 6 to 9, and another for adolescent survivors aged 10 to 17.
Still, even after the question language was modified, we recognized that some questions might be intimidating for survivors in the 6 to 9 age group to answer, possibly making them too shy or uncomfortable to answer. This forced us to get creative and think outside the box. After a lot of reading and brainstorming, we decided to incorporate role-play into the interview guide. Children were asked to play the role of a doctor treating a fictional retinoblastoma patient; in our case, Elli the Elephant. Elli, widely used as a retinoblastoma comfort toy, is a stuffed animal with one removable eye, developed by the German retinoblastoma patient advocacy group Kinder Augen Krebs Stiftung (KAKS, https://www.kinderaugenkrebsstiftung.de/en/homepage/elli/). Questions were modified to reference Elli and her treatment, in hopes to elicit the children’s own ideas and perspectives surrounding treatment outcomes. For example, when discussing the concept of independence and daily functioning, child survivors were asked:
“Elli is quite independent. She likes to read books, ride bicycles, and draw pictures, all on her own! What type of things do you like doing by yourself? Are there any activities you wish you could do on your own that you can’t yet? Is it important that you are able to do things on your own?”
With these modifications (and a box of 20 Timbits as a thank you to each participant), we successfully included young survivors in our research. This additional perspective led to incredibly meaningful findings which were not elicited through conversations with adults. I personally found the pediatric interviews very moving and engaging. In addition to being insightful beyond their years, the pediatric participants were wildly funny and smart. Every child and adolescent interviewed provided a rich description of their experiences living with and managing retinoblastoma. Survivors also clearly explained their personal understanding of what successful treatment means and identified important outcomes to include in a questionnaire. Further, younger survivors gave a detailed account of how they would treat Elli; not only medically, to cure the tumor, but providing comfort throughout the process to help ease her nerves.
I would, however, be remiss not to acknowledge the research contribution of the adult survivors and parents of survivors. Their participation in the focus groups was extremely important in understanding the effects of retinoblastoma treatment over the course of time. It is their engagement and participation in research that drives retinoblastoma research in Canada and contributes to the success of the Canadian Retinoblastoma Research Advisory Board.
Having wrapped up the study now, I can say it was quite a success. We used the information gleaned from participants to identify potential health concepts and possible domains to assess in a future PROM. We are currently in the process of submitting this research for peer-review and publication, after which we will be able to confidently share the results with the broader public. Stay tuned for the release of our results and publication updates!
Conducting this research has taught me more than I can put into one blog post. There are many people who contributed to this work and I am beyond grateful for each and every one of them. However, this research ultimately could not have happened without the generosity and patience of the participants. To all the parents who spent their weekends with me, sharing difficult personal and family experiences; to all the survivors that participated, little and big, I have been profoundly moved by, and am deeply grateful for your kindness in sharing a piece of your life with me and allowing me the opportunity to learn about your world. You made this project real, thank you.