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Randomized Controlled Trial
. 2024 Feb 28;24(1):253.
doi: 10.1186/s12913-024-10586-z.

The ENGAGE study: a 3-arm randomized hybrid type 1 effectiveness and implementation study of an in-home, collaborative PCP model of remote telegenetic services to increase uptake of cancer genetic services in childhood cancer survivors

Tara O Henderson  1 , Mary Ashley Allen  2 , Rajia Mim  3 , Brian Egleston  4 , Linda Fleisher  4 , Elena Elkin  5 , Kevin Oeffinger  6 , Kevin Krull  7 , Demetrios Ofidis  3 , Briana Mcleod  3 , Hannah Griffin  3 , Elizabeth Wood  3 , Cara Cacioppo  3 , Michelle Weinberg  3 , Sarah Brown  3 , Sarah Howe  3 , Aaron McDonald  7 , Chris Vukadinovich  7 , Shani Alston  7 , Dayton Rinehart  7 , Gregory T Armstrong  7 , Angela R Bradbury  3   8
Affiliations
Randomized Controlled Trial

The ENGAGE study: a 3-arm randomized hybrid type 1 effectiveness and implementation study of an in-home, collaborative PCP model of remote telegenetic services to increase uptake of cancer genetic services in childhood cancer survivors

Tara O Henderson et al. BMC Health Serv Res. .

Abstract

Background: Germline cancer genetic testing has become a standard evidence-based practice, with established risk reduction and screening guidelines for genetic carriers. Access to genetic services is limited in many places, which leaves many genetic carriers unidentified and at risk for late diagnosis of cancers and poor outcomes. This poses a problem for childhood cancer survivors, as this is a population with an increased risk for subsequent malignant neoplasms (SMN) due to cancer therapy or inherited cancer predisposition. The ENGaging and Activating cancer survivors in Genetic services (ENGAGE) study evaluates the effectiveness of an in-home, collaborative PCP model of remote telegenetic services to increase uptake of cancer genetic testing in childhood cancer survivors compared to usual care options for genetic testing.

Methods: The ENGAGE study is a 3-arm randomized hybrid type 1 effectiveness and implementation study within the Childhood Cancer Survivor Study population which tests a clinical intervention while gathering information on its delivery during the effectiveness trial and its potential for future implementation among 360 participants. Participants are randomized into three arms. Those randomized to Arm A receive genetic services via videoconferencing, those in Arm B receive these services by phone, and those randomized to Arm C will receive usual care services.

Discussion: With many barriers to accessing genetic services, innovative delivery models are needed to address this gap and increase uptake of genetic services. The ENGAGE study evaluates the effectiveness of an adapted model of remote delivery of genetic services to increase the uptake of recommended genetic testing in childhood cancer survivors. This study assesses the uptake in remote genetic services and identify barriers to uptake to inform future recommendations and a theoretically-informed process evaluation which can inform modifications to enhance dissemination beyond this study population and to realize the benefits of precision medicine.

Trial registration: This protocol was registered at clinicaltrials.gov (NCT04455698) on July 2, 2020.

Keywords: Childhood cancer survivors; Collaborative PCP model; Genetic services; In-home; Remote genetic services; Telegenetic services.

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Conflict of interest statement

The authors declare no competing interests.

Figures

Fig. 1
Fig. 1
Comparison of on-site to in-home model of genetic services
Fig. 2
Fig. 2
Randomized study schema. *6-Month Outcome Survey also given to participants on Arms A and B that have lost contact with Penn Telegenetics before receiving genetic services; participants in Arms A and B who have not had genetic services can still receive services according to their randomized arm
Fig. 3
Fig. 3
U.S. Distribution of CCSS Participants
Fig. 4
Fig. 4
Randomized arm information flyers on how to obtain genetic services
Fig. 5
Fig. 5
Conceptual model to evaluate outcomes of innovations to delivery of genetic services
See this image and copyright information in PMC

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