Background: Although ichthyosis refers to a group of rare, genetic, incurable skin disorders which is documented to have one of the most harmful impacts on a patient's quality of life, little is known about the psychosocial impact on pediatric caregivers. An absence of research exists around the development of a core domain set (CDS), the what to measure, making it difficult to inform clinical practice and formal service delivery. Following the COMET guidelines, we focused on developing a core outcome set (COS).
Method: We conducted a systematic review to identify outcomes reported in literature. An international multi-method qualitative study identified caregiver-reported outcomes which were classified into domains. Items were refined using (i) an international e-Delphi survey process whereby an international multi-disciplinary expert (n=15) and caregiver (n=30) group across 8 countries repeatedly rated items until pre-defined consensus was reached and (ii) qualitative feedback from both groups and (iii) exploratory factor analysis (EFA).
Results: Lack of appropriate outcome or assessment measures contributes to the mismatch between the recognised impact of ichthyosis caregiving and the failure of effective management by clinicians, increasing caregiver hypervigilance and reducing social participation and psychosocial wellbeing of all family members. EFA revealed 3 factors which explained 74% of common variance and reflected six core domains.
Conclusion: We have developed a CDS for ichthyoses. Its application in reporting ichthyoses care and research will facilitate identifying and comparing levels of caregiver need. In order to develop an accessible, purposeful, solution-focused model of reporting outcomes, we will now use a test-retest study approach to psychometrically evaluate each of the identified domains.
Walsh, C., Leavey, G., McLaughlin, M.
Disease Category: Skin
Disease Name: Ichthyosis
Age Range: 0 - 100
Sex: Either
Nature of Intervention: Physical, Psychological & behavioural
- Charities
- Clinical experts
- Consumers (caregivers)
- Consumers (patients)
- Epidemiologists
- Families
- Methodologists
- Patient/ support group representatives
- Policy makers
- Researchers
- Service providers
- Service users
- Statisticians
- COS for practice
- COS for clinical trials or clinical research
- Consensus meeting
- Delphi process
- Focus group(s)
- Interview
- Literature review
- Semi structured discussion
- Systematic review
We conducted a systematic review to identify outcomes reported in literature. An international multi-method qualitative study identified caregiver-reported outcomes which were classified into domains. Items were refined using (i) an international e-Delphi survey process whereby an international multi-disciplinary expert (n=15) and caregiver (n=30) group across 8 countries repeatedly rated items until pre-defined consensus was reached and (ii) qualitative feedback from both groups and (iii) exploratory factor analysis (EFA).