Information provision to caregivers of children with rare dermatological disorders: an international multimethod qualitative study.

OBJECTIVE: To identify service-provided information needs among dermatological caregivers of patients living with ichthyosis. DESIGN: This is the first online international qualitative study to explore caregiver-reported service-provided information needs, using transnational focus groups (n=6), individual interviews (n=7) and in-depth emails (n=5). NVivo facilitated the coding process and Framework Analysis was applied. SETTING: Caregivers were recruited through two online ichthyosis support groups and resided across 10 countries and 5 continents (USA, Greece, Netherlands, Ireland, UK, Canada, India, Philippines, Switzerland and Australia). PARTICIPANTS: A purposive sample of 8 male and 31 female caregivers participated (mean age range 35-44 years). Participants were aged 18 years old or older and fluent in English. Participants cared for a total of 46 children (1:1 ratio for child gender and clinical classification of disease severity). Participants represented all stages along the care continuum, including neonatal intensive care unit and bereavement. RESULTS: This study advances understanding of how to optimise information-sharing across hospital, community and online settings at three points along the care continuum (screening, active caregiving and survivorship). Timely, personalised and appropriate service-provided information support was considered key in influencing the self-efficacy, coping ability and psychosocial well-being of both the caregiver and their child. The modification of information support, through feedback loops, can result in a different bidirectional psychosocial impact for the caregiver and the affected child. CONCLUSION: Our findings provide a novel insight into how existing gaps between caregiver expectations and needs, in terms of information support, can be addressed. As information support is a modifiable factor, improved healthcare education around these themes should become an urgent public health matter to inform future educational and psychosocial interventions.

Novel mixed-method, inclusive protocol involving global key stakeholders, including carers as experts, to co-develop relevant Caregiver-Reported Outcome Domains (CRODs) in skin disease.

INTRODUCTION: Ichthyoses comprise a heterogenous group of rare genetic skin disorders that involves the entire skin surface, often with additional syndromic features, and pose many clinical challenges. Without curative intervention, the mainstay of life-long symptom management is supportive in nature and can remain the responsibility of the caregiver. Although impact on the wider family is considered an important outcome of policies and services, there is a lack of caregiver consensus on what outcome domains to measure to fully assess the impact of ichthyosis on the patient and the caregiver. This project aims to identify a set of core outcome domains towards a core outcome set for ichthyosis that can measure all relevant concepts of ichthyosis in clinical practice, service delivery and research. METHODS AND ANALYSIS: Following the COMET (Core Outcome Measures in Effectiveness Trials) initiative, this project will employ a mixed-method study design which was developed using public and patient involvement and an international multidisciplinary expert group (clinical experts, patients and their representatives, policymakers, researchers and service providers). Experts by experience, or caregivers, will be recruited through online ichthyosis support groups. Phase one will focus on item generation and involve: (1) a systematic literature review, (2) a multimethods international qualitative study with ichthyosis caregivers and (3) co-development of items for an e-survey. Phase two, item refinement, will employ a novel four-pronged consensus approach: (1) an e-Delphi survey, (2) statistical analysis of e-Delphi survey results, (3) online qualitative feedback and (4) an online consensus discussion. All methodological considerations will be clearly linked with each Core Outcome Set-STAndards for Developing recommendation. ETHICS AND DISSEMINATION: Research Ethics Committee approval obtained from the School of Psychology, Ulster University (UK)(Ref:REC/20/0004). Results will be presented in published international peer-reviewed journals, at scientific meetings and support groups. REGISTRATION: COMET database (January 2019).

Systematic review of psychosocial needs assessment tools for caregivers of paediatric patients with dermatological conditions.

OBJECTIVE: To identify validated dermatology-specific and disease-specific psychosocial needs assessment tools for caregivers of paediatric patients with dermatological conditions. A secondary objective was to assess the adequacy of their measurement properties. DESIGN: Systematic review. DATA SOURCES: EMBASE, PsycINFO, MEDLINE (in Ovid SP), Cochrane, Cumulative Index to Nursing and Allied Health EBSCO, U Search and Web of Science were searched (2000-5 October 2021). Grey literature, bibliographies, online databases of QoL tools and several trial registers were searched (2000-5 Oct 2021). ELIGIBILITY CRITERIA: Eligible studies involved adult caregivers caring for a child (no age limit) with any form of any skin condition. Predetermined exclusion criteria, as per protocol, were applied to the search results. DATA ABSTRACTION AND SYNTHESIS: Title, abstract, full-text screening and data abstraction (standardised forms) were done independently in duplicate. Both's predefined methodological criteria assessed risk of bias. Narrative synthesis was used to present the findings. RESULTS: 187 full-text articles were examined from a total of 8979 records. Most tools were generic QoL tools, relevant to spouse/partner or based on their child's perception of the disease or assessed patients' quality of life. Following quality appraisal, 26 articles were identified, and 11 tools (1 dermatology-specific and 10 disease-specific) were included. Information outcome domains were provided for each tool (study specific, questionnaire specific, adequacy of measurement properties and risk of bias). No literature was found pertaining to the use of these tools within healthcare settings and/or as e-tools. DISCUSSION: With limited evidence supporting the quality of their methodological and measurement properties, this review will inform future dermatological Core Outcome Set development and improve evidence-based clinical decisions. Increasing demand on limited healthcare resources justifies the codevelopment of an accessible solution-focused psychosocial needs assessment e-tool to promote caregiver health outcomes. PROSPERO REGISTRATION NUMBER: PROSPERO (CRD42019159956).