The ENGAGE Study: A Randomized Trial Optimizing Uptake of Germline Cancer Genetic Services in Childhood Cancer Survivors
Discuss this preprint
Start a discussion What are Sciety discussions?Listed in
This article is not in any list yet, why not save it to one of your lists.Abstract
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 three arms: remote telehealth genetic services via phone, videoconference, or usual care. Uptake of genetic services was obtained through study records and the six-month Status Survey.
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).
Clinical Trial Registration
NCT 04455698 at http://www.ClinicalTrials.gov
