Co-producing healthcare guidance for children and young people with ataxia telangiectasia (A-T) including the exploration and design of a home-based complex intervention

This doctoral research aimed to co-produce allied healthcare guidance to improve therapeutic care, self-management, and quality of life for children and young people (CYP) living with ataxia telangiectasia (A-T), a rare and complex neurodegenerative condition. Addressing significant gaps in non-medical care provision, the project was structured into four interrelated work packages (WPs), each contributing original insights and practical tools for families and professionals. <br/>WP1 involved a comprehensive scoping review of published and grey literature to map the existing evidence on allied health and nursing management of A-T related symptoms in CYP. The review synthesises 50 sources of evidence and highlighted effective interventions such as therapeutic exercise and respiratory training, resulting in the first best-practice guidance for allied health and nursing care in A-T. <br/>WP2 focused on the co-production of My A-T Pack, a family-owned, person-centred healthcare resource designed to support self-management, symptom tracking, and care coordination. Insights from focus groups with CYP and parents informed the content and design, with the final resource made available in both print and digital formats.<br/>WP3 developed a novel, home-based complex intervention tailored to the needs of CYP with A-T. Through collaborative consultations with families, clinicians, and allied health professionals, a multi-component programme combining adapted yoga and a bespoke breathing exercise movie was co-produced to support functional ability and respiratory health.<br/>WP4 explored the feasibility, acceptability, and perceived benefits of the intervention in a feasibility randomised trial involving six children. Findings demonstrated the intervention was both feasible and acceptable, with preliminary evidence of improved coordination, respiratory health, and overall well-being. <br/>Together, these four WPs offer the first co-produced, evidence-informed allied healthcare framework for CYP with A-T. The project delivers practical resources, original data, and new insights to inform clinical practice and empower families, laying the groundwork for future research and scalable intervention development

Author dc:creator

• Khan, Munira

Contributors dc:contributor

• Lisa Bunn, Tracey Parkin, Amanda Wallace, Oliver Beer

Language dc:language

eng

Repository record dc:identifier

https://pearl.plymouth.ac.uk/hp-theses/28

OAI identifier oai:identifier

oai:pearl.plymouth.ac.uk:hp-theses-1030