Parental Attitudes Toward Standard Newborn Screening and Newborn Genomic Sequencing: Findings From the BabySeq Study.
Parental Attitudes Toward Standard Newborn Screening and Newborn Genomic Sequencing: Findings From the BabySeq Study.
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DOI：
10.3389/fgene.2022.867371
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发表时间：
2022
影响因子：
3.7
通讯作者：
中科院分区：
文献类型：
--
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简介：随着基因组测序的效用增加和成本降低，已经提出了用新生儿基因组测序（nGS）来增强标准新生儿筛查（NBS）程序。在将nGS纳入新生儿筛查之前，必须更好地了解父母的观点。 目的：本文使用对参加BabySeq项目的健康新生儿的父母进行的调查数据，该项目是一项nGS与NBS的随机临床试验，本文报告了父母对基于人群的NBS和nGS的态度，在结果披露后3个月进行评估。 研究方法：父母对是否所有新生儿都应该接受NBS和nGS以及是否需要知情同意的态度，以及是否应该强制执行nGS，使用从强烈反对（=1）到强烈同意（=5）的5点量表进行评估。父母对接收nGS结果类型的兴趣采用5分制进行评估，从完全不感兴趣（=1）到非常感兴趣（=5）。使用Fisher精确检验、配对t检验和重复测量ANOVA分析调查响应。 结果：在披露后3个月，174名健康新生儿的248名父母提交了调查。对每个接受标准NBS的新生儿的支持（平均4.67）高于每个接受nGS的新生儿（平均3.60; p < 0.001）。对NBS（平均3.44）要求知情同意的支持低于nGS（平均4.27，p < 0.001）。父母对NBS和NGS的态度与自我报告的政治取向没有显着相关。如果假设在BabySeq项目之外接受nGS，大多数父母报告说，他们非常有兴趣接受有关婴儿在儿童时期患上可以预防、治疗或治愈的疾病的风险（86.8%）以及他们在成年期患上可以预防、治疗或治愈的疾病的风险（84.6%）的信息。 讨论内容：父母的意见对于公共卫生方案的设计和实施至关重要，因为方案的成功取决于父母的信任和参与。为了在不影响目前国家统计局高参与率的情况下满足家长的偏好，除了国家统计局之外，还可以选择性地增加对国家一般服务的同意，这可能是一个可行的办法。试用注册ClinicalTrials.gov标识符：NCT 02422511。
Introduction: With increasing utility and decreasing cost of genomic sequencing, augmentation of standard newborn screening (NBS) programs with newborn genomic sequencing (nGS) has been proposed. Before nGS can be integrated into newborn screening, parents’ perspectives must be better understood. Objective: Using data from surveys administered to parents of healthy newborns who were enrolled in the BabySeq Project, a randomized clinical trial of nGS alongside NBS, this paper reports parents’ attitudes regarding population-based NBS and nGS assessed 3 months after results disclosure. Methods: Parental attitudes regarding whether all newborns should receive, and whether informed consent should be required for, NBS and nGS, as well as whether nGS should be mandated were assessed using 5-point scales from strongly disagree (=1) to strongly agree (=5). Parents’ interest in receiving types of results from nGS was assessed on a 5-point scale from not at all interested (=1) to very interested (=5). Survey responses were analyzed using Fisher’s exact tests, paired t-tests, and repeated measures ANOVA. Results: At 3 months post-disclosure, 248 parents of 174 healthy newborns submitted a survey. Support for every newborn receiving standard NBS (mean 4.67) was higher than that for every newborn receiving nGS (mean 3.60; p < 0.001). Support for required informed consent for NBS (mean 3.44) was lower than that for nGS (mean 4.27, p < 0.001). Parents’ attitudes toward NBS and nGS were not significantly associated with self-reported political orientation. If hypothetically receiving nGS outside of the BabySeq Project, most parents reported being very interested in receiving information on their baby’s risk of developing a disease in childhood that can be prevented, treated, or cured (86.8%) and their risk of developing a disease during adulthood that can be prevented, treated, or cured (84.6%). Discussion: Parents’ opinions are crucial to inform design and delivery of public health programs, as the success of the program hinges on parents’ trust and participation. To accommodate parents’ preferences without affecting the current high participation rates in NBS, an optional add-on consent to nGS in addition to NBS may be a feasible approach. Trial Registration ClinicalTrials.gov Identifier: NCT02422511.
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作者：
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DOI：
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发表时间：
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