Fraser Harry G, Redmond Rebecca Z, Scotcher Diana F
Division of Evolution and Genomic Sciences, School of Biological Sciences, University of Manchester, Manchester, UK.
Manchester Centre for Genomic Medicine, St Mary's Hospital, Central Manchester University Hospitals NHS Foundation Trust Manchester Academic Health Sciences Centre, 6th Floor, Oxford Road, Manchester, M13 9WL, UK.
J Genet Couns. 2018 Dec;27(6):1349-1359. doi: 10.1007/s10897-018-0266-0. Epub 2018 Jul 4.
Duchenne muscular dystrophy (DMD) and Becker muscular dystrophy (BMD) are X-linked recessive degenerative muscular conditions. Carrier testing is available to at-risk females. Though carrier testing is often offered to adolescent females, it raises ethical issues related to autonomy. This study aimed to address the impact of DMD/BMD carrier testing during adolescence, to elucidate what motivates adolescents to seek testing, and to assess the carrier testing experience. Retrospective semi-structured telephone interviews were conducted with 12 women out of 28 initially contacted. Data were coded using thematic analysis. For most (8/12) participants, discovering their carrier status during adolescence appeared to have helped alleviate uncertainty. The majority (9/12) of participants felt that they had made an autonomous decision and most (10/12) seemed to have adjusted well to their test result. Reproductive factors were framed as having been a key motivator prior to testing. However, following testing, participants' views on prenatal diagnosis seemed more closely linked to their lived experience than to their test result. Just over half (7/12) the participants reported having not had the opportunity for genetic counseling prior to testing and after receiving their result, an issue that warrants further consideration.
杜兴氏肌营养不良症(DMD)和贝克氏肌营养不良症(BMD)是X连锁隐性退行性肌肉疾病。有针对高危女性的携带者检测。虽然携带者检测通常提供给青春期女性,但这引发了与自主权相关的伦理问题。本研究旨在探讨青春期进行DMD/BMD携带者检测的影响,阐明青少年寻求检测的动机,并评估携带者检测经历。对最初联系的28名女性中的12名进行了回顾性半结构化电话访谈。数据采用主题分析进行编码。对于大多数(8/12)参与者来说,在青春期发现自己的携带者状态似乎有助于减轻不确定性。大多数(9/12)参与者认为他们做出了自主决定,并且大多数(10/12)似乎对检测结果适应良好。生殖因素被认为是检测前的关键动机。然而,检测后,参与者对产前诊断的看法似乎与其生活经历的联系比与检测结果的联系更为紧密。略多于一半(7/12)的参与者报告在检测前和收到结果后没有接受遗传咨询的机会,这一问题值得进一步考虑。