Radiological Sciences, Mental Health and Division of Clinical Neuroscience, University of Nottingham, Nottingham, UK.
Department of Radiology, Nottingham University Hospitals NHS Trust, Nottingham, UK.
Health Expect. 2023 Jun;26(3):1358-1367. doi: 10.1111/hex.13756. Epub 2023 Mar 16.
BACKGROUND/OBJECTIVES: Ataxia-telangiectasia (A-T) is a complex inherited disease associated with an increased risk of malignancy. Surveillance guidelines have demonstrated significant health benefits in other cancer predisposition syndromes. However, evidence-based guidelines for cancer screening are not currently used in the United Kingdom for people affected by A-T. This study aims to understand how people with A-T and their parents feel about cancer surveillance using whole-body magnetic resonance imaging (MRI) to inform the future development of cancer surveillance guidelines.
DESIGN/METHODS: We conducted semistructured interviews with people affected by A-T. Data were analysed inductively using thematic analysis.
Nine parents of children with A-T and four adults with A-T were interviewed. Five main themes emerged from the data, including (1) cancer screening was considered invaluable with the perceived value of early detection highlighted; (2) the cancer fear can increase anxiety; (3) the perceived limitations around current practice, with the responsibility for monitoring falling too strongly on parents and patients; (4) the need for effective preparation for cancer screening, including clear communication and (5) the challenges associated with MRI screening, where specific recommendations were made for improving the child's experience.
This study suggests that stakeholders are positive about the perceived advantages of a cancer screening programme. Ongoing support and preparation techniques should be adopted to maximise adherence and minimise adverse psychosocial outcomes.
People with A-T and parents of people with A-T were actively involved in this study by giving their consent to be interviewed. An independent parent representative contributed to the study, supporting the research team in interpreting and commenting on the appropriateness of the language used in this report.
背景/目的：共济失调毛细血管扩张症（A-T）是一种复杂的遗传性疾病，与恶性肿瘤风险增加有关。监测指南在其他癌症易感性综合征中已显示出显著的健康益处。然而，目前在英国，针对受 A-T 影响的人群，并未使用基于证据的癌症筛查指南。本研究旨在了解 A-T 患者及其父母对使用全身磁共振成像（MRI）进行癌症监测的看法，为未来癌症监测指南的制定提供信息。
设计/方法：我们对 A-T 患者进行了半结构式访谈。使用主题分析对数据进行了归纳分析。
共访谈了 9 名 A-T 患儿的家长和 4 名成年 A-T 患者。数据中出现了 5 个主要主题，包括：（1）癌症筛查被认为具有极高的价值，早期检测的重要性得到了强调；（2）癌症恐惧会增加焦虑；（3）对当前实践存在认知局限性，监测责任主要落在父母和患者身上；（4）对癌症筛查进行有效准备的需求，包括清晰的沟通；（5）与 MRI 筛查相关的挑战，针对改善儿童体验提出了具体建议。
本研究表明，利益相关者对癌症筛查计划的潜在优势持积极态度。应采用持续的支持和准备技术，以最大限度地提高依从性并最小化不良心理社会后果。
A-T 患者及其父母通过同意接受访谈积极参与了本研究。一位独立的家长代表也参与了这项研究，支持研究团队对本报告中使用的语言的适当性进行解释和评论。
