The ENGAGE study: a randomized trial optimizing uptake of germline cancer genetic services in childhood cancer survivors

Background: Identifying childhood cancer survivors who are already at high risk of subsequent neoplasms and may also have an inherited genetic susceptibility is essential for effective surveillance and prevention. This trial evaluated the effectiveness of remote, centralized telehealth genetic services in increasing service uptake. Methods: Childhood Cancer Survivor Study (CCSS) participants at the St. Jude Research Hospital, who were ≥18 years old and survivors of a CNS tumor, sarcoma, or more than one primary cancer, were recruited for the study. After completing a baseline survey, participants were randomly assigned to one of the two arms: remote telehealth genetic services (via phone or videoconference) or usual care. Uptake of genetic services was obtained through study records and the six-month Status Survey. This trial was registered with ClinicalTrials.gov (NCT04455698), and accrual has closed. Findings: Of the 391 participants recruited, 262 were assigned to remote telehealth services (via phone or videoconference) and 129 to usual care. At six months, 43% (113/262) of participants in remote telehealth services received genetic services compared to 15% (19/129) in the usual care group (OR = 4.4, 95% CI: 2.5-8.0, p < 0.0001). Uptake of genetic counseling (42% vs. 15%, p < 0.0001) and genetic testing (19% vs. 9%, p = 0.020) were higher in remote telehealth services. Factors associated with higher uptake included not having high-deductible health insurance (OR = 1.67, 95% CI: 1.00-2.91, p = 0.049) and lower perceived cost of testing (OR = 1.51, 95% CI: 1.17-1.96, p = 0.0014). Top barriers included experiencing higher levels of depression (OR = 0.91, 95% CI: 0.85-0.98, p = 0.0067) and anxiety (OR = 0.93, 95% CI: 0.87-1.00, p = 0.036). Interpretation: Remote telehealth genetic services improve genetic counseling and testing uptake in childhood cancer. Addressing remaining barriers could maximize their impact and ensure equitable access for childhood cancer survivors and their families. Funding: National Cancer Institute (R01-CA237369, U24-CA55727).