The ENGAGE Study: A Randomized Trial Optimizing Uptake of Germline Cancer Genetic Services in Childhood Cancer Survivors

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.

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 Competing Interest Statement The authors have declared no competing interest. Clinical Trial Clinical Trial Registration: NCT 04455698 at http://www.ClinicalTrials.gov Funding Statement This study was funded by the National Cancer Institute (R01 CA237369; U24 CA55727; CA21765) and the American Lebanese Syrian Associated Charities. Author Declarations I confirm all relevant ethical guidelines have been followed, and any necessary IRB and/or ethics committee approvals have been obtained. Yes The details of the IRB/oversight body that provided approval or exemption for the research described are given below: The IRB of the University of Pennsylvania gave ethical approval for this work. I confirm that all necessary patient/participant consent has been obtained and the appropriate institutional forms have been archived, and that any patient/participant/sample identifiers included were not known to anyone (e.g., hospital staff, patients or participants themselves) outside the research group so cannot be used to identify individuals. Yes I understand that all clinical trials and any other prospective interventional studies must be registered with an ICMJE-approved registry, such as ClinicalTrials.gov. I confirm that any such study reported in the manuscript has been registered and the trial registration ID is provided (note: if posting a prospective study registered retrospectively, please provide a statement in the trial ID field explaining why the study was not registered in advance). Yes I have followed all appropriate research reporting guidelines, such as any relevant EQUATOR Network research reporting checklist(s) and other pertinent material, if applicable. Yes Footnotes Disclaimer: The manuscript is currently under consideration at The Lancet Regional Health - Americas and has been revised following the first round of peer review. The manuscript has not been accepted for publication. The content may be further revised based on additional editorial or peer review feedback. Data Availability All data produced in the present study are available upon reasonable request to the authors. https://viz.stjude.cloud/community/cancer-survivorship-community~4/publications.