John D Pauling1, Robyn T Domsic2, Lesley A Saketkoo3, Celia Almeida4, Jane Withey5, Hilary Jay5, Tracy M Frech6, Francesca Ingegnoli7, Emma Dures4, Joanna Robson4, Neil J McHugh1, Ariane L Herrick8, Marco Matucci-Cerinic9, Dinesh Khanna10, Sarah Hewlett4. 1. Royal National Hospital for Rheumatic Diseases (at Royal United Hospitals), and University of Bath, Bath, UK. 2. University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania. 3. Tulane University School of Medicine, New Orleans, Louisiana. 4. University of the West of England, Bristol, UK. 5. Royal National Hospital for Rheumatic Diseases (at Royal United Hospitals), Bath, UK. 6. University of Utah and Salt Lake Regional Veterans Affairs Medical Center, Salt Lake City. 7. University of Milano, Milano, Italy. 8. University of Manchester, Salford Royal Hospital NHS Foundation Trust, and Manchester Academic Health Science Centre, Manchester, UK. 9. University of Florence, Florence, Italy. 10. University of Michigan, Ann Arbor.
Abstract
OBJECTIVE: Raynaud's phenomenon (RP) is the most common manifestation of systemic sclerosis (SSc). RP is an episodic phenomenon, not easily assessed in the clinic, leading to reliance on self-report. A thorough understanding of the patient experience of SSc-RP is essential to ensuring that patient-reported outcome (PRO) instruments capture domains important to the target patient population. We report the findings of an international qualitative research study investigating the patient experience of SSc-RP. METHODS: Focus groups of SSc patients were conducted across 3 scleroderma centers in the US and UK, using a topic guide and a priori purposive sampling framework devised by qualitative researchers, SSc patients, and SSc experts. Focus groups were audio recorded, transcribed, anonymized, and analyzed using inductive thematic analysis. Focus groups were conducted until thematic saturation was achieved. RESULTS: Forty SSc patients participated in 6 focus groups conducted in Bath (UK), New Orleans (Louisiana), and Pittsburgh (Pennsylvania). Seven major themes were identified that encapsulate the patient experience of SSc-RP: physical symptoms, emotional impact, triggers and exacerbating factors, constant vigilance and self-management, impact on daily life, uncertainty, and adaptation. The interrelationship of the 7 constituent themes can be arranged within a conceptual map of SSc-RP. CONCLUSION: We have explored the patient experience of SSc-RP in a diverse and representative SSc cohort and identified a complex interplay of experiences that result in significant impact. Work to develop a novel PRO instrument for assessing the severity and impact of SSc-RP, comprising domains/items grounded in the patient experiences of SSc-RP identified in this study, is underway.
OBJECTIVE:Raynaud's phenomenon (RP) is the most common manifestation of systemic sclerosis (SSc). RP is an episodic phenomenon, not easily assessed in the clinic, leading to reliance on self-report. A thorough understanding of the patient experience of SSc-RP is essential to ensuring that patient-reported outcome (PRO) instruments capture domains important to the target patient population. We report the findings of an international qualitative research study investigating the patient experience of SSc-RP. METHODS: Focus groups of SSc patients were conducted across 3 scleroderma centers in the US and UK, using a topic guide and a priori purposive sampling framework devised by qualitative researchers, SSc patients, and SSc experts. Focus groups were audio recorded, transcribed, anonymized, and analyzed using inductive thematic analysis. Focus groups were conducted until thematic saturation was achieved. RESULTS: Forty SSc patients participated in 6 focus groups conducted in Bath (UK), New Orleans (Louisiana), and Pittsburgh (Pennsylvania). Seven major themes were identified that encapsulate the patient experience of SSc-RP: physical symptoms, emotional impact, triggers and exacerbating factors, constant vigilance and self-management, impact on daily life, uncertainty, and adaptation. The interrelationship of the 7 constituent themes can be arranged within a conceptual map of SSc-RP. CONCLUSION: We have explored the patient experience of SSc-RP in a diverse and representative SSc cohort and identified a complex interplay of experiences that result in significant impact. Work to develop a novel PRO instrument for assessing the severity and impact of SSc-RP, comprising domains/items grounded in the patient experiences of SSc-RP identified in this study, is underway.
Authors: B Coleiro; S E Marshall; C P Denton; K Howell; A Blann; K I Welsh; C M Black Journal: Rheumatology (Oxford) Date: 2001-09 Impact factor: 7.580
Authors: A L Herrick; S Hollis; D Schofield; F Rieley; A Blann; K Griffin; T Moore; J M Braganza; M I Jayson Journal: Clin Exp Rheumatol Date: 2000 May-Jun Impact factor: 4.473
Authors: John R Kirwan; Susan J Bartlett; Dorcas E Beaton; Maarten Boers; Ailsa Bosworth; Peter M Brooks; Ernest Choy; Maarten de Wit; Francis Guillemin; Sarah Hewlett; Tore K Kvien; Robert B Landewé; Amye L Leong; Anne Lyddiatt; Lyn March; James May; Pamela Lesley Montie; Enkeleida Nikaï; Pam Richards; Marieke M J H Voshaar; Wilma Smeets; Vibeke Strand; Peter Tugwell; Laure Gossec Journal: J Rheumatol Date: 2014-03-01 Impact factor: 4.666
Authors: John D Pauling; Joana Caetano; Corrado Campochiaro; Giacomo De Luca; Ana Maria Gheorghiu; Maria Grazia Lazzaroni; Dinesh Khanna Journal: J Scleroderma Relat Disord Date: 2019-11-25
Authors: Lesley Ann Saketkoo; Tracy Frech; Cecília Varjú; Robyn Domsic; Jessica Farrell; Jessica K Gordon; Carina Mihai; Nora Sandorfi; Lee Shapiro; Janet Poole; Elizabeth R Volkmann; Monika Lammi; Kendra McAnally; Helene Alexanderson; Henrik Pettersson; Faye Hant; Masataka Kuwana; Ami A Shah; Vanessa Smith; Vivien Hsu; Otylia Kowal-Bielecka; Shervin Assassi; Maurizio Cutolo; Cristiane Kayser; Victoria K Shanmugam; Madelon C Vonk; Kim Fligelstone; Nancy Baldwin; Kerri Connolly; Anneliese Ronnow; Beata Toth; Maureen Suave; Sue Farrington; Elana J Bernstein; Leslie J Crofford; László Czirják; Kelly Jensen; Monique Hinchclif; Marie Hudson; Matthew R Lammi; Jennifer Mansour; Nadia D Morgan; Fabian Mendoza; Mandana Nikpour; John Pauling; Gabriela Riemekasten; Anne-Marie Russell; Mary Beth Scholand; Elise Seigart; Tatiana Sofia Rodriguez-Reyna; Laura Hummers; Ulrich Walker; Virginia Steen Journal: Best Pract Res Clin Rheumatol Date: 2021-09-15 Impact factor: 4.991
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
Authors: Murray Baron; Bashar Kahaleh; Elana J Bernstein; Lorinda Chung; Philip J Clements; Christopher Denton; Robyn T Domsic; Nava Ferdowsi; Ivan Foeldvari; Tracy Frech; Jessica K Gordon; Marie Hudson; Sindhu R Johnson; Dinesh Khanna; Zsuzsannah McMahan; Peter A Merkel; Sonali Narain; Mandana Nikpour; John D Pauling; Laura Ross; Antonia Maria Valenzuela Vergara; Alessandra Vacca Journal: J Scleroderma Relat Disord Date: 2018-07-18