卫生经济评估用于确定产前或新生儿筛查计划产生净收益所需的资源,在多重利益和危害的驱使下,是合理的。目前尚不清楚评估这些方案的经济评价采取了哪些利弊,以及它们是否忽略了对相关利益攸关方重要的利弊。
■(1)确定卫生经济评估在该领域采用的益处和危害,并评估它们是如何被衡量和评估的;(2)确定未来经济评估中应考虑的属性或与利益相关者的相关性;(3)就这些研究应考虑的利益和危害提出建议。
■将系统回顾和定性工作相结合的混合方法。
■我们使用所有主要的电子数据库搜索了2000年1月至2021年1月的已发布和灰色文献。一个或多个经济合作与发展组织国家的产前或新生儿筛查计划的经济评估被认为是合格的。使用综合卫生经济评估报告标准清单评估报告质量。我们使用综合描述性分析确定了利弊,并构建了主题框架。
■我们对新生儿筛查经验的现有文献进行了元人种学研究,对与产前或新生儿筛查或生活在筛查条件下相关的现有个人访谈的二次分析,以及与利益相关者一起收集的有关其筛查经验的主要数据的主题分析。
■文献检索确定了52,244篇文章和报告,并纳入336项独特研究。专题框架产生了七个主题:(1)诊断筛查条件,(2)生命年和健康状况调整,(3)治疗,(4)长期成本,(5)过度诊断,(6)妊娠损失和(7)对家庭成员的溢出效应。筛查条件的诊断(115,47.5%),生命年和健康状况调整(90,37.2%)和治疗(88,36.4%)占评估产前筛查的大部分益处和危害。相同的主题占了评估新生儿筛查的研究中的大部分益处和危害。长期成本,过度诊断和溢出效应往往被忽视。筛查的广泛家庭影响被认为对利益相关者很重要。我们观察到专题框架和定性证据之间有很好的重叠。
■由于纳入了大量研究,因此在系统文献综述中提取双重数据是不可行的。很难在利益相关者的面试中招募医疗保健专业人员。
■在该领域的卫生经济评估中使用的益处和危害的选择没有一致性,建议需要额外的方法指导。我们提出的主题框架可用于指导未来卫生经济评估的发展,以评估产前和新生儿筛查计划。
■本研究注册为PROSPEROCRD42020165236。
■该奖项由美国国家卫生与护理研究所(NIHR)卫生技术评估计划(NIHR奖参考:NIHR127489)资助,并在《卫生技术评估》中全文发表;卷。28号25.有关更多奖项信息,请参阅NIHR资助和奖励网站。
NHS每年都会为孕妇提供筛查测试,以评估她们或未出生的婴儿患有或发展为健康状况的机会。它还为新生婴儿提供筛查测试,以寻找一系列健康状况。筛查计划的实施以及对妇女和婴儿的护理需要NHS的许多资源和资金,因此,重要的是筛选程序代表物有所值。这意味着NHS在计划上花费的金额由该计划提供的收益来证明。我们想看看研究人员在计算物有所值时是否考虑了与孕妇和新生儿筛查相关的所有重要益处和危害。要做到这一点,我们搜索了发达国家的所有研究,以确定他们认为的益处和危害.我们还考虑了父母和医疗保健专业人员对为家庭和更广泛的社会创造的好处和危害筛查的意见。我们发现,筛查的益处和危害的识别是复杂的,因为筛查结果会影响一系列人群(母婴,父母,大家庭和更广泛的社会)。研究人员计算筛查项目的物有所值,到目前为止,集中在狭窄范围的益处和危害上,而忽略了许多对筛查结果影响的人很重要的因素。从我们与父母和医疗保健专业人员的讨论中,我们发现,对家庭的更广泛影响是一个重要的考虑因素。我们研究的只有一项研究考虑了对家庭的更广泛影响。我们的工作还发现父母的识别能力,吸收和应用新的信息,以了解他们的孩子的筛查结果或条件是重要的。参与筛查的医疗保健专业人员在支持患有某种疾病的儿童家庭时应考虑这一点。我们为研究人员创建了一份清单,以确定未来研究中重要的益处和危害。我们还确定了研究人员评估这些益处和危害的不同方式,所以他们以一种有意义的方式融入到他们的研究中。
UNASSIGNED: Health economic assessments are used to determine whether the resources needed to generate net benefit from an antenatal or newborn screening programme, driven by multiple benefits and harms, are justifiable. It is not known what benefits and harms have been adopted by economic evaluations assessing these programmes and whether they omit benefits and harms considered important to relevant stakeholders.
UNASSIGNED: (1) To identify the benefits and harms adopted by health economic assessments in this area, and to assess how they have been measured and valued; (2) to identify attributes or relevance to stakeholders that ought to be considered in future economic assessments; and (3) to make recommendations about the benefits and harms that should be considered by these studies.
UNASSIGNED: Mixed methods combining systematic
review and qualitative work.
UNASSIGNED: We searched the published and grey literature from January 2000 to January 2021 using all major electronic databases. Economic evaluations of an antenatal or newborn screening programme in one or more Organisation for Economic Co-operation and Development countries were considered eligible. Reporting quality was assessed using the Consolidated Health Economic Evaluation Reporting Standards checklist. We identified benefits and harms using an integrative descriptive analysis and constructed a thematic framework.
UNASSIGNED: We conducted a meta-ethnography of the existing literature on newborn screening experiences, a secondary analysis of existing individual interviews related to antenatal or newborn screening or living with screened-for conditions, and a thematic analysis of primary data collected with stakeholders about their experiences with screening.
UNASSIGNED: The literature searches identified 52,244 articles and reports, and 336 unique studies were included. Thematic framework resulted in seven themes: (1) diagnosis of screened for condition, (2) life-years and health status adjustments, (3) treatment, (4) long-term costs, (5) overdiagnosis, (6) pregnancy loss and (7) spillover effects on family members. Diagnosis of screened-for condition (115, 47.5%), life-years and health status adjustments (90, 37.2%) and treatment (88, 36.4%) accounted for most of the benefits and harms evaluating antenatal screening. The same themes accounted for most of the benefits and harms included in studies assessing newborn screening. Long-term costs, overdiagnosis and spillover effects tended to be ignored. The wide-reaching family implications of screening were considered important to stakeholders. We observed good overlap between the thematic framework and the qualitative evidence.
UNASSIGNED: Dual data extraction within the systematic literature
review was not feasible due to the large number of studies included. It was difficult to recruit healthcare professionals in the stakeholder\'s interviews.
UNASSIGNED: There is no consistency in the selection of benefits and harms used in health economic assessments in this area, suggesting that additional methods guidance is needed. Our proposed thematic framework can be used to guide the development of future health economic assessments evaluating antenatal and newborn screening programmes.
UNASSIGNED: This study is registered as PROSPERO CRD42020165236.
UNASSIGNED: This award was funded by the National Institute for Health and Care Research (NIHR) Health Technology Assessment programme (NIHR award ref: NIHR127489) and is published in full in Health Technology Assessment; Vol. 28, No. 25. See the NIHR Funding and Awards website for further award information.
Every year the NHS offers pregnant women screening tests to assess the chances of them or their unborn baby having or developing a health condition. It also offers screening tests for newborn babies to look for a range of health conditions. The implementation of screening programmes and the care for women and babies require many resources and funding for the NHS, so it is important that screening programmes represent good value for money. This means that the amount of money the NHS spends on a programme is justified by the amount of benefit that the programme gives. We wanted to see whether researchers consider all the important benefits and harms associated with screening of pregnant women and newborn babies when calculating value for money. To do this, we searched all studies available in developed countries to identify what benefits and harms they considered. We also considered the views of parents and healthcare professionals on the benefits and harms screening that creates for families and wider society. We found that the identification of benefits and harms of screening is complex because screening results affect a range of people (mother–baby, parents, extended family and wider society). Researchers calculating the value for money of screening programmes have, to date, concentrated on a narrow range of benefits and harms and ignored many factors that are important to people affected by screening results. From our discussions with parents and healthcare professionals, we found that wider impacts on families are an important consideration. Only one study we looked at considered wider impacts on families. Our work also found that parent’s ability to recognise, absorb and apply new information to understand their child’s screening results or condition is important. Healthcare professionals involve in screening should consider this when supporting families of children with a condition. We have created a list for researchers to identify the benefits and harms that are important to include in future studies. We have also identified different ways researchers can value these benefits and harms, so they are incorporated into their studies in a meaningful way.