Conijn Thirsa, van Dijke Ivy, Haverman Lotte, Lakeman Phillis, Wijburg Frits A, Henneman Lidewij
Amsterdam UMC, Pediatric Metabolic Diseases, Emma Children's Hospital and Amsterdam Lysosome Center "Sphinx", University of Amsterdam, Amsterdam, the Netherlands.
Amsterdam UMC, Psychosocial Department, Emma Children's Hospital, University of Amsterdam, Amsterdam, the Netherlands.
J Community Genet. 2021 Jul;12(3):311-323. doi: 10.1007/s12687-021-00519-2. Epub 2021 Mar 22.
Preconception expanded carrier screening (ECS) enables prospective parents to assess their risk of having a child with an autosomal recessive disorder. Knowledge on motivations, feelings, and considerations people have towards the offer and use of ECS is limited. To enrich the public and professional discussion on ECS implementation, this study explored the perspectives towards various aspects of ECS in seven focus groups compromising first- and second-degree relatives of MPS III patients (N=9, N=4, N=5, N=5) and members of the general Dutch population (N=6, N=7, N=5). The focus groups were audio recorded and the transcripts were qualitatively analyzed to identify themes. Both relatives of MPS III patients and participants from the general population supported offering ECS, in particular for severe, childhood-onset disorders. Important barriers identified for ECS were a lack of genetic knowledge and a perceived lack of personal relevance and awareness, as well as out-of-pocket costs of testing. The majority of participants would prefer full disclosure of individual test results instead of couple-based test results. Moreover, offering people a choice for the way of reporting was proposed. All participants agreed that more efforts, for example by governmental campaigns, should be made to increase awareness on the availability, potentials, and limitations of ECS. Educating prospective parents about ECS is essential for increasing awareness and informed decision making. This study provides valuable insights that can be used by governments and public health authorities when considering implementation of preconception ECS.
孕前扩展携带者筛查(ECS)可使准父母评估生育患常染色体隐性疾病孩子的风险。人们对ECS的提供和使用的动机、感受及考虑因素的了解有限。为丰富关于ECS实施的公众和专业讨论,本研究在七个焦点小组中探讨了对ECS各方面的看法,这些焦点小组包括黏多糖贮积症III型(MPS III)患者的一级和二级亲属(分别为9人、4人、5人、5人)以及荷兰普通人群的成员(分别为6人、7人、5人)。焦点小组进行了录音,并对文字记录进行定性分析以确定主题。MPS III患者的亲属和普通人群的参与者都支持提供ECS,特别是针对严重的儿童期发病疾病。确定的ECS的重要障碍包括缺乏遗传知识、感觉缺乏个人相关性和认知,以及检测的自付费用。大多数参与者更喜欢完全披露个人检测结果而不是基于夫妻的检测结果。此外,建议为人们提供报告方式的选择。所有参与者都同意,应该做出更多努力,例如通过政府宣传活动,以提高对ECS的可获得性、潜力和局限性的认识。为准父母提供关于ECS的教育对于提高认识和做出明智决策至关重要。本研究提供了有价值的见解,政府和公共卫生当局在考虑实施孕前ECS时可以加以利用。