背景：许多国家为未出生和新生婴儿提供筛查计划（产前和新生儿筛查），以确定有某些疾病风险的婴儿，以帮助早期诊断和治疗。技术进步刺激了筛查计划的发展，以包括更多的条件，随后改变所需的信息和潜在的利益-风险权衡推动参与。量化筛选方案的偏好可以为方案专员提供了解潜在需求的数据,这种需求的驱动因素，支持方案所需的信息提供以及人口偏好差异的程度。这项研究旨在确定已发表的研究，这些研究激发了对产前和新生儿筛查计划的偏好，并概述了关键方法和发现。
方法：对电子数据库进行系统搜索，以确定关键术语的合格研究（1990年至2018年10月发表的与产前/新生儿检测/筛查相关的离散选择实验(DCE)或最佳-最差缩放比例(BWS)研究）。系统地提取了数据，在叙述性审查中列出并总结。
结果：共有19项研究使用DCE或BWS来引发产前筛查（n=15；79%）和新生儿筛查（n=4；21%）方案。大多数研究是在欧洲进行的（n=12;63％），但也有一些来自北美（n=2;11％）和澳大利亚（n=2;11％）的例子。最常见的属性是筛选的准确性（n=15;79％）和筛选发生时（n=13;68％）。其他常见的属性包括信息内容（n=11;58％）和流产风险（n=10;53％）。孕妇（n=11;58％）和医疗保健专业人员（n=11;58％）是最常见的研究样本。10项研究（53%）比较了不同受访者的偏好。两项研究（11％）在国家之间进行了比较。最流行的分析模型是标准的条件logit模型（n=11；58％），一项研究通过潜在类别分析调查了偏好异质性。
结论：现有文献确定了产前和新生儿筛查的偏好，但结合更复杂的设计和分析方法来调查偏好异质性可能会扩大研究结果的相关性，以告知新的筛查计划的委托。
BACKGROUND: Many countries offer screening programmes to unborn and newborn babies (antenatal and newborn screening) to identify those at risk of certain conditions to aid earlier diagnosis and treatment. Technological advances have stimulated the development of screening programmes to include more conditions, subsequently changing the information required and potential benefit-risk trade-offs driving participation. Quantifying preferences for screening programmes can provide programme commissioners with data to understand potential demand, the drivers of this demand, information provision required to support the programmes and the extent to which preferences differ in a population. This study aimed to identify published studies eliciting preferences for antenatal and newborn screening programmes and provide an overview of key methods and findings.
METHODS: A systematic search of electronic databases for key terms identified eligible studies (discrete choice experiments (DCEs) or best-worst scaling (BWS) studies related to antenatal/newborn testing/screening published between 1990 and October 2018). Data were systematically extracted, tabulated and summarised in a narrative
review.
RESULTS: A total of 19 studies using a DCE or BWS to elicit preferences for antenatal (n = 15; 79%) and newborn screening (n = 4; 21%) programmes were identified. Most of the studies were conducted in Europe (n = 12; 63%) but there were some examples from North America (n = 2; 11%) and Australia (n = 2; 11%). Attributes most commonly included were accuracy of screening (n = 15; 79%) and when screening occurred (n = 13; 68%). Other commonly occurring attributes included information content (n = 11; 58%) and risk of miscarriage (n = 10; 53%). Pregnant women (n = 11; 58%) and healthcare professionals (n = 11; 58%) were the most common study samples. Ten studies (53%) compared preferences across different respondents. Two studies (11%) made comparisons between countries. The most popular analytical model was a standard conditional logit model (n = 11; 58%) and one study investigated preference heterogeneity with latent class analysis.
CONCLUSIONS: There is an existing literature identifying stated preferences for antenatal and newborn screening but the incorporation of more sophisticated design and analytical methods to investigate preference heterogeneity could extend the relevance of the findings to inform commissioning of new screening programmes.
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