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The Scleroderma Patient-centered Intervention Network (SPIN) Cohort: protocol for a cohort multiple randomised controlled trial (cmRCT) design to support trials of psychosocial and rehabilitation interventions in a rare disease context
  1. Linda Kwakkenbos1,2,
  2. Lisa R Jewett1,3,
  3. Murray Baron1,4,
  4. Susan J Bartlett4,
  5. Dan Furst5,
  6. Karen Gottesman6,
  7. Dinesh Khanna7,
  8. Vanessa L Malcarne8,9,
  9. Maureen D Mayes10,
  10. Luc Mouthon11,12,
  11. Serge Poiraudeau11,13,14,
  12. Maureen Sauve15,
  13. Warren R Nielson16,
  14. Janet L Poole17,
  15. Shervin Assassi10,
  16. Isabelle Boutron18–20,
  17. Carolyn Ells21,
  18. Cornelia HM van den Ende22,
  19. Marie Hudson1,4,
  20. Ann Impens23,
  21. Annett Körner1,3,24,
  22. Catarina Leite25,26,
  23. Angela Costa Maia25,
  24. Cindy Mendelson27,
  25. Janet Pope28,
  26. Russell J Steele1,29,
  27. Maria E Suarez-Almazor30,
  28. Sara Ahmed31,32,
  29. Stephanie Coronado-Montoya1,
  30. Vanessa C Delisle1,3,
  31. Shadi Gholizadeh9,
  32. Yeona Jang33,
  33. Brooke Levis1,34,
  34. Katherine Milette1,3,
  35. Sarah D Mills9,
  36. Ilya Razykov1,2,
  37. Rina S Fox9,
  38. Brett D Thombs1–534–36
  1. 1Lady Davis Institute for Medical Research, Jewish General Hospital, Montréal, Québec, Canada
  2. 2Department of Psychiatry, Montréal, Québec, Canada
  3. 3Department of Educational and Counselling Psychology, Montréal, Québec, Canada
  4. 4Department of Medicine, McGill University, Montréal, Québec, Canada
  5. 5Division of Rheumatology, Geffen School of Medicine at the University of California, Los Angeles, California, USA
  6. 6Southern California Chapter, Scleroderma Foundation, Los Angeles, California, USA
  7. 7University of Michigan Scleroderma Program, Ann Arbor, Michigan, USA
  8. 8Department of Psychology, San Diego State University, San Diego, California, USA
  9. 9San Diego State University/University of California, San Diego Joint Doctoral Program in Clinical Psychology, San Diego, California, USA
  10. 10University of Texas Health Science Center Houston, Houston, Texas, USA
  11. 11Université Paris Descartes, Assistance Publique-Hôpitaux de Paris (AP-HP), Paris, France
  12. 12Pôle de Médecine Interne, Hôpital Cochin, Paris, France
  13. 13Pôle Ostéo-articulaire, Hôpital Cochin, Paris, France
  14. 14IFR Handicap INSERM, Paris, France
  15. 15Scleroderma Societies of Canada and Ontario, Hamilton, Ontario, Canada
  16. 16Beryl & Richard Ivey Rheumatology Day Programs, St Joseph's Health Care, London, Ontario, Canada
  17. 17Occupational Therapy Graduate Program, University of New Mexico, Albuquerque, New Mexico, USA
  18. 18INSERM U738, Paris, France
  19. 19University Paris Descartes, Paris, France
  20. 20Assistance Publique-Hôpitaux de Paris, Hôpital Hôtel-Dieu, Centre d'Epidémiologie Clinique, Paris, France
  21. 21Biomedical Ethics Unit, McGill University, Montréal, Québec, Canada
  22. 22Department of Rheumatology, Sint Maartenskliniek, Ubbergen, Gelderland, The Netherlands
  23. 23Internal Medicine, Midwestern University, Downers Grove, Illinois, USA
  24. 24Department of Oncology, McGill University, Montréal, Québec, Canada
  25. 25School of Psychology, University of Minho, Braga, Portugal
  26. 26Federation of European Scleroderma Associations, Froyennes, Belgium
  27. 27College of Nursing, University of New Mexico, Albuquerque, New Mexico, USA
  28. 28Medicine, Division of Rheumatology, University of Western Ontario, St Joseph's Health Care, London, Ontario, Canada
  29. 29Department of Mathematics and Statistics, McGill University, Montréal, Québec, Canada
  30. 30University of Texas MD Anderson Cancer Center, Houston, Texas, USA
  31. 31School of Physical and Occupational Therapy, McGill University, Montréal, Québec, Canada
  32. 32McGill University Health Center, Clinical Epidemiology Montréal, Montréal, Québec, Canada
  33. 33Desautels Faculty of Management, McGill University, Montréal, Québec, Canada
  34. 34Department of Epidemiology, Biostatistics, and Occupational Health, McGill University, Montréal, Québec, Canada
  35. 35Department of Psychology, McGill University, Montréal, Québec, Canada
  36. 36School of Nursing, McGill University, Montréal, Québec, Canada
  1. Correspondence to Dr Brett D Thombs; brett.thombs{at}mcgill.ca

Abstract

Introduction Psychosocial and rehabilitation interventions are increasingly used to attenuate disability and improve health-related quality of life (HRQL) in chronic diseases, but are typically not available for patients with rare diseases. Conducting rigorous, adequately powered trials of these interventions for patients with rare diseases is difficult. The Scleroderma Patient-centered Intervention Network (SPIN) is an international collaboration of patient organisations, clinicians and researchers. The aim of SPIN is to develop a research infrastructure to test accessible, low-cost self-guided online interventions to reduce disability and improve HRQL for people living with the rare disease systemic sclerosis (SSc or scleroderma). Once tested, effective interventions will be made accessible through patient organisations partnering with SPIN.

Methods and analysis SPIN will employ the cohort multiple randomised controlled trial (cmRCT) design, in which patients consent to participate in a cohort for ongoing data collection. The aim is to recruit 1500–2000 patients from centres across the world within a period of 5 years (2013–2018). Eligible participants are persons ≥18 years of age with a diagnosis of SSc. In addition to baseline medical data, participants will complete patient-reported outcome measures every 3 months. Upon enrolment in the cohort, patients will consent to be contacted in the future to participate in intervention research and to allow their data to be used for comparison purposes for interventions tested with other cohort participants. Once interventions are developed, patients from the cohort will be randomly selected and offered interventions as part of pragmatic RCTs. Outcomes from patients offered interventions will be compared with outcomes from trial-eligible patients who are not offered the interventions.

Ethics and dissemination The use of the cmRCT design, the development of self-guided online interventions and partnerships with patient organisations will allow SPIN to develop, rigourously test and effectively disseminate psychosocial and rehabilitation interventions for people with SSc.

  • Rheumatology
  • Statistics & Research Methods
  • Rehabilitation Medicine
  • Mental Health

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