Alain Lescoat1, Susan L Murphy2, Yen T Chen3, Nadia Vann4, Francesco Del Galdo5, David Cella6, Maya H Buch7, Dinesh Khanna8. 1. Department of Internal Medicine and Clinical Immunology, Rennes University Hospital, Rennes, France; Univ Rennes, CHU Rennes, Inserm, EHESP, Irset (Institut de Recherche en Santé, Environnement et Travail) - UMR_S 1085, Rennes, France; Michigan Scleroderma Program, University of Michigan, Ann Arbor, MI, USA. 2. Michigan Scleroderma Program, University of Michigan, Ann Arbor, MI, USA; Department of Physical Medicine & Rehabilitation, University of Michigan, USA; VA Ann Arbor Healthcare System, Geriatric Research Education and Clinical Center, GRECC, Ann Arbor, MI USA. Electronic address: sumurphy@med.umich.edu. 3. Michigan Scleroderma Program, University of Michigan, Ann Arbor, MI, USA; Department of Physical Medicine & Rehabilitation, University of Michigan, USA. 4. Michigan Scleroderma Program, University of Michigan, Ann Arbor, MI, USA. 5. Leeds Institute of Rheumatic and Musculoskeletal Medicine, University of Leeds, Leeds, UK; Leeds Biomedical Research Centre, National Institute for Health Research, Leeds, UK. 6. Department of Medical Social Sciences, Northwestern University Feinberg School of Medicine, 625 Michigan Ave, 21st Floor, Chicago, IL, 60611, USA. 7. Centre for Musculoskeletal Research, School of Biological Sciences, Faculty of Biology, Medicine & Health, University of Manchester, Manchester, UK.; NIHR Manchester Biomedical Research Centre, Manchester Academic Health Science Centre, Manchester University Foundation Trust, Manchester, UK. 8. Michigan Scleroderma Program, University of Michigan, Ann Arbor, MI, USA; Division of Rheumatology, University of Michigan, Ann Arbor, Michigan, USA.
Abstract
OBJECTIVES: Limited cutaneous systemic sclerosis (lcSSc) is the largest subgroup of people with SSc, but little is known about symptom experience from the patients' perspective. This study aimed to comprehensively identify domains and symptoms reported as bothersome by patients with lcSSc and to analyze themes and sub-themes capturing symptom experience in this population. METHODS: Focus groups of patients with lcSSc were conducted using a structured guide. Patients were recruited based on an a priori purposive framework to include men and women with SSc. Focus groups were recorded, transcribed, anonymized, and analyzed using the RADaR technique (rigorous and accelerated data reduction) combined with iterative, deductive and inductive approaches. RESULTS: Four 2-hour Focus groups comprising participants with lcSSc were conducted (N = 26). Ninety-four symptoms were identified with 22 domains. Symptoms from the following domains were mentioned in all Focus groups: skin, musculoskeletal (MSK), cardiac, pulmonary, gastro-intestinal (GI) manifestations, fatigue, sleep, Raynaud's phenomenon (RP), pain, and digital ulcers. The three most cited domains in a pre-meeting survey were GI, RP and MSK. Seven themes defining symptom experience were identified: difficulty attributing symptoms to this complex systemic disease, influence of symptom temporality, impairment of emotional well-being, limitations in functioning (including several sub-themes of functioning), survival, symptoms management and uncertainty. CONCLUSION: This study comprehensively identified domains and symptoms considered bothersome from the perspective of patients with lcSSc and summarized patient experience of lcSSc-related symptoms. This study is the first step in the design of a future combined response index dedicated to lcSSc that could foster specific research on this subpopulation of patients.
OBJECTIVES: Limited cutaneous systemic sclerosis (lcSSc) is the largest subgroup of people with SSc, but little is known about symptom experience from the patients' perspective. This study aimed to comprehensively identify domains and symptoms reported as bothersome by patients with lcSSc and to analyze themes and sub-themes capturing symptom experience in this population. METHODS: Focus groups of patients with lcSSc were conducted using a structured guide. Patients were recruited based on an a priori purposive framework to include men and women with SSc. Focus groups were recorded, transcribed, anonymized, and analyzed using the RADaR technique (rigorous and accelerated data reduction) combined with iterative, deductive and inductive approaches. RESULTS: Four 2-hour Focus groups comprising participants with lcSSc were conducted (N = 26). Ninety-four symptoms were identified with 22 domains. Symptoms from the following domains were mentioned in all Focus groups: skin, musculoskeletal (MSK), cardiac, pulmonary, gastro-intestinal (GI) manifestations, fatigue, sleep, Raynaud's phenomenon (RP), pain, and digital ulcers. The three most cited domains in a pre-meeting survey were GI, RP and MSK. Seven themes defining symptom experience were identified: difficulty attributing symptoms to this complex systemic disease, influence of symptom temporality, impairment of emotional well-being, limitations in functioning (including several sub-themes of functioning), survival, symptoms management and uncertainty. CONCLUSION: This study comprehensively identified domains and symptoms considered bothersome from the perspective of patients with lcSSc and summarized patient experience of lcSSc-related symptoms. This study is the first step in the design of a future combined response index dedicated to lcSSc that could foster specific research on this subpopulation of patients.
Authors: Daniel Sumpton; Adrien Bigot; Benedicte Sautenet; Jonathan C Craig; Geraldine Hassett; Vivek Thakkar; Peter Tugwell; Allison Tong Journal: Arthritis Care Res (Hoboken) Date: 2019-07-15 Impact factor: 4.794
Authors: Michael Hughes; John D Pauling; Jennifer Jones; Christopher P Denton; Robyn T Domsic; Tracy M Frech; Ariane L Herrick; Dinesh Khanna; Marco Matucci-Cerinic; Lorraine McKenzie; Lesley Ann Saketkoo; Rachael Gooberman-Hill; Andrew Moore Journal: Arthritis Care Res (Hoboken) Date: 2020-05 Impact factor: 4.794
Authors: Tanja A Stamm; Malin Mattsson; Carina Mihai; Juliane Stöcker; Alexa Binder; Bettina Bauernfeind; Georg Stummvoll; Gunvor Gard; Roger Hesselstrand; Gunnel Sandqvist; Oana Draghicescu; Ana Maria Gherghe; Malina Voicu; Klaus P Machold; Oliver Distler; Josef S Smolen; Carina Boström Journal: Ann Rheum Dis Date: 2011-06 Impact factor: 19.103