Fatigue and activity management education for individuals with systemic sclerosis: Adaptation and feasibility study of an intervention for a rare disease

Abstract

Purpose: Fatigue is an overlooked symptom for musculoskeletal diseases, including rare conditions like systemic sclerosis (SSc). The purpose of this study were the following: (1) to adapt the content and delivery method of an existing fatigue intervention, and (2) to determine the feasibility of Fatigue and Activity Management Education in Systemic Sclerosis (FAME-iSS).

Methods: In Phase 1 adaptations were recorded using the Framework for Modification and Adaptations. In Phase 2, participants completed the 6-week FAME-iSS intervention and baseline, post-intervention, and 3-month follow-up assessments measuring fatigue levels, fatigue impact, mental health, self-efficacy, and use of energy conservation strategies. Qualitative data included a post-intervention focus group and 3-month follow-up interviews.

Results: Three main changes were made to the original intervention: (1) the content was adapted from management of Lupus-related fatigue to management of SSc-related fatigue, (2) context for delivery from Ireland to United States, and (3) in-person to online format. Participants (n = 4) were 51.8 ± 12.1 years old, had been diagnosed with SSc for 12.0 ± 8.0 years, were receiving disability support, and had college degrees. All participants had 100% attendance and completed all study activities. Participants had positive comments about the programme format, content, and implementation. Post-intervention, improvements were observed in most measures. Moderate effect sizes were noted in fatigue and self-efficacy scores.

Conclusion: Participants' positive responses supported the need for FAME-iSS for people with SSc. Even with the small sample, FAME-iSS led to improvements in fatigue and use of energy conservation strategies. The online format allowed for participation and sharing of ideas, especially during the global pandemic.

Trial registration: ClinicalTrials.gov NCT04401943.

Keywords: activity participation; fatigue; non-pharmacological intervention; occupational therapy; rheumatology; scleroderma; self-management; systemic sclerosis.