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

IF 9.1 Q1 HEALTH CARE SCIENCES & SERVICES

Lancet Regional Health-Americas Pub Date : 2026-03-01 Epub Date: 2026-02-13 DOI:10.1016/j.lana.2026.101375

Tara O. Henderson , Brian Egleston , Sarah Howe , Mary Ashley Allen , Rajia Mim , Linda G. Fleisher , Elena B. Elkin , Kevin C. Oeffinger , Kevin R. Krull , Demetrios Ofidis , Briana McLeod , Hannah Griffin , Elisabeth M. Wood , Cara N. Cacioppo , Sarah Brown , Melody Perpich , Gregory T. Armstrong , Angela R. Bradbury

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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 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).

查看原文本刊更多论文

ENGAGE研究：一项优化儿童癌症幸存者生殖系癌症遗传服务吸收的随机试验

背景：鉴别儿童癌症幸存者是否已经处于后续肿瘤的高风险中，并且可能具有遗传易感性，对于有效的监测和预防至关重要。本试验评估了远程集中远程医疗遗传服务在提高服务接受度方面的有效性。方法招募St. Jude研究医院的儿童癌症幸存者研究（CCSS）参与者，年龄≥18岁，患有中枢神经系统肿瘤、肉瘤或一种以上原发癌症的幸存者。在完成基线调查后，参与者被随机分配到两组中的一组：远程远程医疗遗传服务（通过电话或视频会议）或常规护理。通过研究记录和为期六个月的状况调查获得了遗传服务的吸收情况。该试验已在ClinicalTrials.gov注册（NCT04455698），项目已结束。在被招募的391名参与者中，262人被分配到远程医疗服务（通过电话或视频会议），129人被分配到常规护理。六个月时，43%（113/262）的远程医疗服务参与者接受了遗传服务，而常规护理组的这一比例为15% (19/129)（OR = 4.4, 95% CI: 2.5-8.0, p < 0.0001）。在远程医疗服务中，遗传咨询（42%对15%，p < 0.0001）和基因检测（19%对9%，p = 0.020）的使用率更高。与较高的吸收率相关的因素包括没有高免赔额医疗保险（OR = 1.67, 95% CI: 1.00-2.91, p = 0.049）和较低的感知检测成本（OR = 1.51, 95% CI: 1.17-1.96, p = 0.0014）。主要障碍包括经历较高程度的抑郁（OR = 0.91, 95% CI: 0.85-0.98, p = 0.0067）和焦虑（OR = 0.93, 95% CI: 0.87-1.00, p = 0.036）。远程远程医疗遗传服务改善了儿童癌症遗传咨询和检测的吸收。解决剩下的障碍可以最大限度地发挥其影响，并确保儿童癌症幸存者及其家人公平获得机会。国家癌症研究所（R01-CA237369, U24-CA55727）。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Lancet Regional Health-Americas Multiple-

CiteScore

8.00

自引率

0.00%

发文量

期刊介绍： The Lancet Regional Health – Americas, an open-access journal, contributes to The Lancet's global initiative by focusing on health-care quality and access in the Americas. It aims to advance clinical practice and health policy in the region, promoting better health outcomes. The journal publishes high-quality original research advocating change or shedding light on clinical practice and health policy. It welcomes submissions on various regional health topics, including infectious diseases, non-communicable diseases, child and adolescent health, maternal and reproductive health, emergency care, health policy, and health equity.