Michael Hughes1, John D Pauling2, Jennifer Jones3, Christopher P Denton4, Robyn T Domsic5, Tracy M Frech6, Ariane L Herrick7, Dinesh Khanna8, Marco Matucci-Cerinic9, Lorraine McKenzie10, Lesley Ann Saketkoo11, Rachael Gooberman-Hill12, Andrew Moore3. 1. University of Manchester, Manchester, Royal Hallamshire Hospital, and Sheffield Teaching Hospitals NHS Foundation Trust, Sheffield, UK. 2. Royal National Hospital for Rheumatic Diseases and University of Bath, Bath, UK. 3. Bristol Medical School, Bristol, UK. 4. Royal Free Hospital, University College London, London, UK. 5. University of Pittsburgh Medical Center, Pittsburgh, PA. 6. University of Utah and Salt Lake Veterans Affairs Medical Center, Salt Lake City. 7. University of Manchester, Salford Royal NHS Foundation Trust, and Manchester Academic Health Science Centre, Manchester, UK. 8. University of Michigan, Ann Arbor. 9. University of Florence, Florence, Italy. 10. Patient representative (contact via Dr. Herrick, University of Manchester, Manchester, UK). 11. Tulane University School of Medicine, New Orleans Scleroderma and Sarcoidosis Patient Care and Research Center, and University Medical Center Comprehensive Pulmonary Hypertension Center, New Orleans, Louisiana. 12. Bristol Medical School, NIHR Bristol Biomedical Research Centre, and University Hospitals Bristol NHS Foundation Trust, Bristol, UK.
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.
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.
Authors: Alain Lescoat; Susan L Murphy; Yen T Chen; Nadia Vann; Francesco Del Galdo; David Cella; Maya H Buch; Dinesh Khanna Journal: Semin Arthritis Rheum Date: 2021-11-07 Impact factor: 5.431