Whole-body MRI for cancer surveillance in ataxia–telangiectasia: A qualitative study of the perspectives of people affected by A-T and their families

Neves, Renata; Perez, Blanca De Dios; Tindall, Tierney; Fernandez, Natasha Schneider; Panek, Rafal; Wilne, Sophie; Suri, Mohnish; Whitehouse, William; Jagani, Sumit; Dandapani, Madhumita; Dineen, Robert; Glazebrook, Cris

Abstract

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.

Results
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.

Conclusion
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.

Patient or Public Contribution
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.

Citation

Neves, R., Perez, B. D. D., Tindall, T., Fernandez, N. S., Panek, R., Wilne, S., …Glazebrook, C. (in press). Whole-body MRI for cancer surveillance in ataxia–telangiectasia: A qualitative study of the perspectives of people affected by A-T and their families. Health Expectations, Article hex.13756. https://doi.org/10.1111/hex.13756

• Journal Article Type: Article
• Acceptance Date: Mar 3, 2023
• Online Publication Date: Mar 16, 2023
• Deposit Date: Mar 31, 2023
• Publicly Available Date: Mar 31, 2023
• Journal: Health Expectations
• Print ISSN: 1369-6513
• Electronic ISSN: 1369-7625
• Peer Reviewed: Peer Reviewed
• Article Number: hex.13756
• DOI: https://doi.org/10.1111/hex.13756
• Keywords: Cancer predisposition; life-limiting disease; psychosocial impact; Public and Patient Involvement and Engagement; qualitative research
• Public URL: https://nottingham-repository.worktribe.com/output/9906952
• Publisher URL: https://onlinelibrary.wiley.com/doi/10.1111/hex.13756
• Additional Information: This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited. © 2023 The Authors. Health Expectations published by John Wiley & Sons Ltd.