Whole-Body MRI for cancer surveillance in Ataxia Telangiectasia: A qualitative study of the perspectives of people affected by A-T and their families

Abstract

Background/objectives: Ataxia Telangiectasia (A-T) is a complex inherited disease that is associated with an increased risk of malignancy. Surveillance guidelines have demonstrated significant health benefits in other cancer predisposition syndromes (CPS). Evidence-based guidelines for cancer screening are not currently used in the UK 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 MRI to inform the future development of cancer surveillance guidelines. Design/Methods: We conducted semi-structured interviews of 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. All participants viewed cancer screening for children with A-T as invaluable and emphasised the perceived value of early detection. The second theme acknowledged the anxiety associated with cancer and cancer surveillance and the third theme highlighted the perceived limitations around current practice, with the responsibility for monitoring falling too strongly on parents and patients. The fourth theme identified the need for effective preparation for cancer screening, including clear communication, and the challenges of MRI screening were recognised in the fifth theme with specific recommendations 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.

Feasibility of whole‐body MRI for cancer screening in children and young people with ataxia telangiectasia: A mixed methods cross‐sectional study

“…Parents reported an increase in anxiety in anticipation of the scan and receiving the results, but contextualised this against the fact that living with A‐T is already emotionally demanding and some felt they have developed the resilience needed to manage the additional short‐term anxiety 33,49 . ‘Scanxiety’ was reported in similar studies related to cancer surveillance and referrers to the temporary increase of anxiety observed around the screening time and during the waiting period to receive the results 44–46 .…”

“…22/YH/0053). The study was prospectively registered at http://clinicaltrials.gov (NCT05252819) and the protocol published in an open‐access online research repository prior to commencement of data analysis 34 …”

“…gov (NCT05252819) and the protocol published in an openaccess online research repository prior to commencement of data analysis. 34 2.2 | Procedures…”

“…26 A recent qualitative study with people with A-T and family members found that they want cancer screening because of the potential benefits of early cancer diagnosis, but identified concerns that screening could heighten anxiety related to the scan procedure and potential for an unexpected cancer diagnosis. 33 The primary aim of this prospective, mixed methods study was to establish the technical feasibility of WB-MRI for cancer screening in children and young people with A-T. Secondary aims were to establish the prevalence and spectrum of cancer and non-cancer abnormalities detected on WB-MRI in this population and to assess the views of, and psychological impact on, participants and families in response to participating in a feasibility study of WB-MRI for cancer screening.…”

“…As well as the physical and emotional impact of scanning for children, screening can increase anxiety for parents/carers due to the possibility of finding cancer or other pathologies 26 . A recent qualitative study with people with A‐T and family members found that they want cancer screening because of the potential benefits of early cancer diagnosis, but identified concerns that screening could heighten anxiety related to the scan procedure and potential for an unexpected cancer diagnosis 33 …”