Multicenter Qualitative Study Exploring the Patient Experience of Digital Ulcers in Systemic Sclerosis

Abstract

Objective. Digital ulcers (DUs) are a major cause of disease-related morbidity and are a difficult-to-treat vascular complication of systemic sclerosis (SSc). Demonstrating treatment efficacy has traditionally focused on clinician assessment of DUs alone. No existing patient-reported outcome (PRO) instrument captures the multifaceted impact of SSc-DU. We report the findings of a multicenter qualitative research study exploring the patient experience of SSc-DU.Methods. Patient focus groups were conducted across 3 scleroderma units, following a topic guide devised by SSc patients, experts, and experienced qualitative researchers. A purposive sampling framework ensured that the experiences of a diverse group of patients were captured. Focus groups were audio recorded, and information was transcribed, anonymized, and analyzed using inductive thematic analysis. We continued focus groups until thematic saturation was achieved.Results. Twenty-nine SSc patients with a history of DU disease participated in 4 focus groups across the UK (Bath, Manchester, and London). Five major interrelated themes (and subthemes) were identified that encompass the patient experience of SSc-DU: disabling pain and hypersensitivity; deep and broad-ranging emotional impact; impairment of physical and social activity; factors aggravating occurrence, duration, and impact; and mitigating, managing, and adapting.Conclusion. The patient experience of SSc-DU is multifaceted and comprises a complex interplay of experiences associated with significant pain and morbidity. Patient experiences of SSc-DU are not captured using existing SSc-DU outcomes. Our findings will inform the development of a novel PRO instrument to assess the severity and impact of SSc-DU for use in future SSc-DU clinical trials.

What narrative devices do people with systemic sclerosis use to describe the experience of pain from digital ulcers: a multicentre focus group study at UK scleroderma centres

“…FGs were held in informal settings away from the clinical areas and were all facilitated by MH (a consultant rheumatologist with special interest in SSc), sometimes in conjunction with another consultant rheumatologist and SSc specialist (JP) and a qualitative researcher (AJM) from the project team. 3 FGs continued until thematic saturation had been reached, such that no new codes or themes were developing by the fourth FG. 38 We used the Standards for Reporting Qualitative Data (SRQR) checklist when writing our report.…”

“…41 Previously reported findings from this study included the range and nature of the impact of SSC-DUs on hand functionality, social functioning, psychological impacts and pain. 3 As pain was a major element, we felt that a more in-depth exploration was warranted. This analysis explores the theme of pain in greater depth, for the purposes of developing a future patient-reported outcome measure (PROM), and to inform discussions about pain between healthcare practitioners and people diagnosed with SSc experiencing DUs.…”

“…Studies of the lived experience of people with systemic sclerosis (SSc) indicate that pain is one of the most challenging symptoms of this rare autoimmune connective tissue disease. [1][2][3] SSc is characterised by widespread vasculopathy, tissue fibrosis and immune system activation. 4 Around half of people with SSc experience digital ulcers (DUs) at some point in their disease course.…”

“…3 10 11 A recent study by Hughes and colleagues found five themes that described patient experience of those with SSC-DUs; 'Disabling pain and hypersensitivity', 'Deep and broad-ranging emotional impact', 'Impairment of physical and social activity', 'Factors aggravating occurrence, duration and impact' and 'Mitigating, managing and adapting'. 3 Some people with DUs experience such difficult symptoms that they express a desire to have their fingers amputated. 1 3 People with SSc-DUs report higher levels of pain than those with SSc without DUs.…”

“…37 The aim of the present study was to explore the nature of pain related to SSc-DUs using data obtained from a multicentre qualitative research study examining the patient experience of SSc-DUs, which had identified pain as a key and central focus defining the patient experience. 3 The specific objectives of this analysis were to first capture 'pain expression' and the pain narrative of people with SSc-DUs and second, to understand how the language and expressions relating to DUs could inform the narrative of clinical consultations.…”