Informing the development of an international core information set for ichthyoses towards a core outcome set to inform clinical practice and service delivery

Background: Although information-giving in healthcare services plays a considerable role in attenuating caregiver burden and increasing satisfaction with services, standards of information are lacking and practice varies for caregivers of children affected by ichthyosis. This is the first study focusing on service-provided information needs from the perspective of caregivers. The development and use of a core information set may improve healthcare understanding of the importance of information needs assessment and provides a minimum set of information to use in all consultations. This is particularly significant for caregivers of children with rare and chronic dermatological disease who are increasingly responsible for providing medical, psychological and social support within the home environment.

Methods: Information was identified from the literature and from a qualitative study using transnational focus groups and individual interviews. We used Framework Analysis to identify key outcomes relating to the provision of information around caregiver knowledge, skills and coping. These outcomes will be integrated into an online e-Delphi survey for item refinement and to agree the core set.

Results: Thirty-nine caregivers caring for 46 children with various forms of ichthyosis participated. Caregivers represented ten countries across four continents. A theoretical framework that underscores five key themes is proposed, relating to the type and source of service-provided information, discussed at three points on the care continuum: (i) screening (ii) active caregiving and (iii) survivorship. Key themes identified include genetic diagnosis and counselling, service-provided information at hospital level, service-provided information at community level, online medical support groups and structured follow-up practical information support. Key recommendations for optimising service-provided information support will be included. An international multi-disciplinary expert group (n=15) and caregiver group (n=30) will participate in an e-Delphi survey where they will be asked to rate the importance of information from ‘extremely important’ to not important’ to determine which items reach pre-defined consensus levels.

Conclusion: Information support is crucial to help caregivers for children with rare dermatological disorders cope with such stressful and resource-intensive conditions. This study will establish a core information set for ichthyosis caregivers and may inform the effective targeting of valuable and limited healthcare resources in hospital and community healthcare settings. As information support is a modifiable factor, it is imperative that improved healthcare education around these themes become an urgent public health matter to help reduce caregiver physical and emotional burnout.

Contributors

Walsh, C. (Bamford Centre for Mental Health and Wellbeing, Ulster University, Northern Ireland)
Leavey, G. (Bamford Centre for Mental Health and Wellbeing, Ulster University, Northern Ireland)
McLaughlin, M. (Ulster University)

Further Study Information

Current Stage: Ongoing
Date: January 2021 - December 2021
Funding source(s): None


Health Area

Disease Category: Skin

Disease Name: Ichthyoses

Target Population

Age Range: 0 - 100

Sex: Either

Nature of Intervention: Physical, Psychological & behavioural

Stakeholders Involved

- Clinical experts
- Consumers (caregivers)
- Consumers (patients)
- Epidemiologists
- Families
- Methodologists
- Patient/ support group representatives
- Policy makers
- Researchers
- Service providers
- Service users

Study Type

- Patient perspectives

Method(s)

- Consensus meeting
- Delphi process
- Focus group(s)
- Interview
- Literature review
- Systematic review

Information was identified from the literature and from a qualitative study using transnational focus groups and individual interviews. We used Framework Analysis to identify key outcomes relating to the provision of information around caregiver knowledge, skills and coping. These outcomes will be integrated into an online e-Delphi survey for item refinement and to agree the core set.