Values of Retinoblastoma Survivors and Parents Regarding Treatment Outcomes: A Qualitative Study

Authors:Ana Janic, Ajoy Vincent, Jennifer Stinson, Helen Dimaras. 

Author Disclosure Block:A. Janic: Funded grants; The Hospital for Sick Children, Restracomp Scholarship, The Hospital for Sick Children, Ophthalmology Research Fund, University of Toronto Vision Science Research Program (VSRP) Scholarship. A. Vincent: Funded grants; Foundation Fighting Blindness, USA.  J. Stinson: None. H. Dimaras: Membership on advisory boards or speakers’ bureaus; Canadian Retinoblastoma Research Advisory Board. Description of relationship(s); Board Member. Funded grants; The Hospital for Sick Children, Ophthalmology Research Fund. Funded grants or clinical trials.

Abstract Body:

Purpose: The purpose of this study was to uncover which treatment outcomes retinoblastoma survivors and their parents’ value. Retinoblastoma is an aggressive pediatric eye cancer. Patient-reported outcome measures - instruments that measure any outcome related to health that are directly reported by patients themselves - reveal important insights on how patients perceive their own health. Currently, no widely used or validated measure for the assessment of retinoblastoma outcomes exists.
Study Design: This qualitative, cross-sectional study included Canadian retinoblastoma survivors aged 6 years and older, and parents of retinoblastoma survivors. Participants who did not demonstrate fluency in English were excluded.
Methods: Study subjects participated in semi-structured interviews or focus groups, either in person at The Hospital for Sick Children, Toronto, Canada, or through secure videoconference, between March 3, 2019, and January 25, 2020. Iterative rounds of opening coding, codebook development, and co-researcher analysis, were utilized to identify key emergent themes and subthemes.
Results: Seventeen adults (8 survivors, 9 parents) participated in 6 focus groups. Nine pediatric survivors participated in individual interviews, five children aged 6-9, and 4 adolescents, aged 11-16. Four common themes emerged from all participant groups: (1) Definition of treatment success, (2) Enucleation - acceptance and challenges, (3) Treatment outcomes to measure, and (4) Need for outcome reporting. An additional, unique theme was identified in all pediatric discussions: (1) Worries and coping mechanisms. Treatment outcomes deemed valuable were related to the following domains: psychosocial outcomes, daily functioning, functional vision, retinoblastoma education, cosmetic outcomes, and secondary eye conditions.
Conclusions: This study represents the first stage in the development of a retinoblastoma-specific patient-reported outcome measure. Further, this work represents the first study of its kind for the retinoblastoma population and is novel in its inclusion of pediatric survivors as young as 6 years of age. The findings reveal insight into what outcomes are valued by survivors after treatment and offers promise to improve outcomes assessment for retinoblastoma.