Widening engagement and participation in cancer screening research in Ataxia Telangiectasia

Background Ataxia telangiectasia (A-T) is a complex multi-system disorder caused by inherited mutations in a single gene. A-T is characterised by cerebellar ataxia, immunodeficiency and increased risk of cancer in both children and adults. It is estimated in the UK around 37% of the A-T population are from non-white ethnic groups. These groups experience significant cultural and language barriers to access A-T services and participate in research. In contrast to other cancers, there are still no evidence-based cancer screening guidelines for A-T. Recent qualitative work with families, technical feasibility studies and international engagement has provided groundwork for the development of future trials for cancer screening in A-T using whole-body MRI combined with blood tests. To inform the inclusive design of future cancer screening trials and given the diversity within the UK A-T population it is important to delve into the barriers to accessing services and participating in research for patients, families and caregivers. Aims and objectives Aim: "To identify social, cultural, geographic and language barriers that limit uptake of cancer screening in the A-T population and cocreate approaches to improve representation in future trials and screening programmes." Objectives: To understand the views of people with A-T and parents/caregivers on screening in the A-T population, focusing on the views of people from diverse groups. To map the role of social, cultural, geographic and language factors in relation to screening for A-T, and how these may facilitate or hinder uptake. To co-develop and co-produce recommendations and approaches to facilitate uptake of screening among the diverse AT population. Methods A qualitative study comprising 3 work packages. WP1 – Multi-stakeholder focus groups to co-produce recruitment strategy for WP2. WP2 – Interview study with patients, families/caregivers to explore barriers and facilitators to screening for patients with A-T from diverse groups. WP3 – Multi-stakeholder focus groups to co-produce a toolkit of approaches and recommendations to promote inclusivity within research and service delivery among the diverse A-T population. Timelines for delivery The study will run for 24 months Study approvals: Months 0-3 WP1 recruitment of stakeholders to focus group, data collection and analysis, development of WP2 recruitment strategy: Months 3-6 WP2 patient, parent, caregiver recruitment, interviews and data analysis: Months 6-18 WP3 recruitment for consensus panels, data collection, consultation and agreement of panel outputs: Months 18-24 Final reporting and dissemination event: Months 21-24 Impact Engage with A-T patients and families from diverse groups. Identify barriers to engagement in A-T clinical pathway and research for patients and families from underrepresented groups. Co-develop solutions relevant to future A-T research, trials and service delivery. Dissemination A community engagement event with key stakeholders involved in A-T research, service delivery and advocacy, to disseminate a toolkit of recommendations and approaches for inclusive clinical research and clinical service design and delivery, to improve engagement of underrepresented groups affected by A-T. The target audience for the event are healthcare professionals delivering A-T services, researchers, industry partners, A-T society representatives, patients, families and caregivers. Open access, peer-reviewed journals and scientific conferences.