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在英格兰传达通过新生儿镰状细胞病和囊性纤维化普遍筛查检测出的携带者状态结果:对当前实践和政策挑战的定性研究

Imparting carrier status results detected by universal newborn screening for sickle cell and cystic fibrosis in England: a qualitative study of current practice and policy challenges.

作者信息

Parker Hilda, Qureshi Nadeem, Ulph Fiona, Kai Joe

机构信息

Division of Primary Care, University of Nottingham, Graduate Medical School, Derby City General Hospital, Uttoxeter Road, Derby, DE22 3DT, UK.

出版信息

BMC Health Serv Res. 2007 Dec 13;7:203. doi: 10.1186/1472-6963-7-203.

Abstract

BACKGROUND

Universal newborn screening for early detection of children affected by sickle cell disorders and cystic fibrosis is currently being implemented across England. Parents of infants identified as carriers of these disorders must also be informed of their baby's result. However there is a lack of evidence for most effective practice internationally when doing so. This study describes current or proposed models for imparting this information in practice and explores associated challenges for policy.

METHODS

Thematic analysis of semi-structured interviews with Child Health Coordinators from all English Health Regions.

RESULTS

Diverse methods for imparting carrier results, both within and between regions, and within and between conditions, were being implemented or planned. Models ranged from result by letter to in-person communication during a home visit. Non-specialists were considered the best placed professionals to give results and a similar approach for both conditions was emphasised. While national guidance has influenced choice of models, other factors contributed such as existing service structures and lack of funding. Challenges included uncertainty about guidance specifying face to face notification; how best to balance allaying parental anxiety by using familiar non-specialist health professionals with concerns about practitioner competence; and extent of information parents should be given. Inadequate consideration of resource and service workload was seen as the main policy obstacle. Clarification of existing guidance; more specific protocols to ensure consistent countrywide practice; integration of the two programmes; and 'normalising' carrier status were suggested as improvements.

CONCLUSION

Differing models for communicating carrier results raise concerns about equity and clinical governance. However, this variation provides opportunity for evaluation. Timely and more detailed guidance on protocols with clarification of existing recommendations is needed.

摘要

背景

目前英格兰正在推行新生儿普遍筛查,以早期发现受镰状细胞病和囊性纤维化影响的儿童。被确定为这些疾病携带者的婴儿的父母也必须被告知其孩子的检测结果。然而,在国际上,缺乏关于这样做时最有效做法的证据。本研究描述了在实践中传递此信息的当前或提议模式,并探讨相关的政策挑战。

方法

对来自英格兰所有卫生区域的儿童健康协调员进行半结构化访谈的主题分析。

结果

正在实施或计划在区域内和区域间、疾病内和疾病间采用多种方式传递携带者检测结果。模式从通过信件告知结果到家访时进行面对面交流。非专科医生被认为是告知结果的最佳专业人员,并强调针对这两种疾病采用类似方法。虽然国家指南影响了模式的选择,但其他因素也有作用,如现有的服务结构和资金短缺。挑战包括指南中关于面对面通知的不确定性;如何最好地平衡通过使用熟悉的非专科卫生专业人员减轻父母焦虑与对从业者能力的担忧;以及应向父母提供的信息量。对资源和服务工作量考虑不足被视为主要的政策障碍。建议进行改进,包括澄清现有指南;制定更具体的方案以确保全国范围内做法一致;整合这两个项目;以及使携带者状态“正常化”。

结论

传递携带者检测结果的不同模式引发了对公平性和临床治理的担忧。然而,这种差异提供了评估的机会。需要及时提供更详细的方案指南,并澄清现有建议。

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