Chest illness accounts for 51% of deaths in children and young people with cerebral palsy, and is the most common reason for this population to seek primary care consultation, emergency hospital care and intensive care. Chest illness accrues significant healthcare cost and affects quality of life for children and their families.
Outcome measures remain a significant barrier in improving chest health and preventing hospital admissions in this population. Relevant clinical trials are characterised by low quality methodology and inconsistent measures, limiting capacity for evidence synthesis to determine treatment effectiveness. In clinical practice, early detection of chest infections is vital to support timely treatment decisions and prevent hospital stays. Yet, widely used measures such as spirometry are challenging to replicate in CYP with CP. This has resulted in inadequate monitoring, late reactive treatment provision and life-threatening consequences. A proposed COS in chest health for CYP with CP is timely as interest and efforts to increase research capacity in this topic are growing, whilst health and research initiatives are driving preventative approaches to care.
There is an existing overarching COS for children with cerebral palsy by Vargus-Adams et al. (2009), which sets out broad outcomes domains e.g. fine motor skills. However, many of these domains are not sensitive to the specific outcomes for neuro-respiratory impairments, whilst the recommended instruments are challenging to replicate in CYP with more complex CP, associated with chest morbidity.
Aim:
To develop a Core Outcome Set (COS) and their linked measurement instrument to assess, monitor and evaluate interventions aiming to improve chest health in children and young people with cerebral palsy, informed by key international stakeholders (children and young people, caregivers and professionals) from diverse communities.
Mrs Rachel Knight Lozano (Principal Investigator), UNiversity of Plymouth
Professor Jos Latour (Supervisor), University of Plymouth
Dr Harriet Shannon (Supervisor), UCL Great Ormond Street Institute of Child Health
Professor Chris Morris (Supervisor), University of Exeter
Professor Jon Marsden (Supervisor), UNiversity of Plymouth
Mrs Rachel Rapson (Contributor), University of Plymouth / Child and Family Health Devon
Disease Category: Child health
Disease Name: Cerebral palsy, Respiratory diseases
Age Range: 2 - 18
Sex: Either
Nature of Intervention: Other
- Charities
- Clinical experts
- Families
- Governmental agencies
- Patient/ support group representatives
- Researchers
- Service providers
- Service users
- COS for clinical trials or clinical research
- COS for practice
- Consensus meeting
- Delphi process
- Interview
- Literature review
- Nominal group technique (NGT)
- Systematic review
A 3-phase COS research strategy is proposed. The scope focuses on assessment and evaluation of any community-based intervention aimed at improving chest health in in children and young people across the spectrum of cerebral palsy.
Phase 1: Potential outcome domains will be identified through literature review and qualitative interviews with key stakeholders (children and young people, caregivers and professionals.
Phase 2: Proposed outcome domains will be scored in an international e-Delphi survey, using a 9-point Likert scale. The most important outcome domains for a core outcome set (COS) will be agreed and refined in a final consensus meeting with key stakeholders.
Phase 3: Measurement instruments will be identified for each core domain, and evaluated to determine reliability, validity, responsiveness, interpretability, appropriateness, precision, acceptability and feasibility. The most appropriate measurement instruments for each core outcome domain will be agreed and refined in a final consensus meeting with key stakeholders.