Huang Ying, Feng Ziding
Fred Hutchinson Cancer Research Center,Seattle, WA, 98109.
Ann Appl Stat. 2024 Jun;18(2):1543-1564. doi: 10.1214/23-aoas1848. Epub 2024 Apr 5.
Cancer screening facilitates the early detection of cancer, at a stage when treatment is often most effective. However, it also brings the risk of over-diagnosis, where a diagnosis made through screening would not have led to symptoms or death during the patient's lifetime. In this paper, we tackle a significant unresolved issue in the evaluation of screening efficacy: selecting primary endpoints and inferential procedures that efficiently consider potential overdiagnosis in screening trials. This is motivated by the necessity to design and analyze a phase IV Early Detection Initiative (EDI) trial for evaluating a pancreatic cancer screening strategy. We introduce two novel approaches for assessing screening efficacy, grounded on cancer stage-shift. These methods address potential overdiagnosis by: i) borrowing information about clinical diagnosis from the control arm that hasn't undergone screening (the BR approach), and ii) performing sensitivity analysis, contingent upon a conservative bound of the overdiagnosis magnitude (the SEN-T approach). Analytical methods and extensive simulation studies underscore the superiority of our proposed methods, demonstrating enhanced efficiency in estimating and testing screening efficacy compared to existing methods. The latter either overlook overdiagnosis or adhere to a valid, yet conservative, cumulative incidence endpoint. We illustrate the practical application of these approaches using ovarian cancer data from the Prostate, Lung, Colorectal, and Ovarian Cancer Screening Trial. The results affirm that our methods bolster an efficient and robust study design for cancer screening trials.
癌症筛查有助于在治疗往往最有效的阶段早期发现癌症。然而,它也带来了过度诊断的风险,即通过筛查做出的诊断在患者一生中不会导致出现症状或死亡。在本文中,我们解决了评估筛查效果中一个重大的未解决问题:选择能有效考虑筛查试验中潜在过度诊断的主要终点和推断程序。这是出于设计和分析一项用于评估胰腺癌筛查策略的IV期早期检测倡议(EDI)试验的必要性。我们引入了两种基于癌症阶段转移来评估筛查效果的新方法。这些方法通过以下方式解决潜在的过度诊断问题:i)从未接受筛查的对照组借用有关临床诊断的信息(BR方法),以及ii)根据过度诊断幅度的保守界限进行敏感性分析(SEN-T方法)。分析方法和广泛的模拟研究强调了我们提出的方法的优越性,表明与现有方法相比,在估计和检验筛查效果方面具有更高的效率。现有方法要么忽略过度诊断,要么坚持一个有效但保守的累积发病率终点。我们使用前列腺、肺、结肠和卵巢癌筛查试验中的卵巢癌数据说明了这些方法的实际应用。结果证实,我们的方法支持了一种用于癌症筛查试验的高效且稳健的研究设计。