Scleroderma is a rare chronic autoimmune connective tissue disease characterized by abnormal fibrotic processes and excessive collagen production. Common problems faced by people with scleroderma include limitations in hand function and mobility, pain, fatigue, constant itch, depression, and body image distress from disfiguring aspects of the disease.
SPIN was created to bring together people living with scleroderma, healthcare professionals who care for them, and scleroderma researchers from around the world to develop and test accessible patient support tools for scleroderma patients, including self-management, rehabilitation, psychological, and educational tools. SPIN’s mission is to help scleroderma patients live better lives with their disease and to serve as a model of a way forward for other rare diseases.
SPIN was conceived and founded by Dr. Thombs, who is the Director. Dr. Linda Kwakkenbos is the co-Director, and Marie-Eve Carrier is SPIN’s Coordinator. SPIN leadership also includes a Steering Committee, comprised of experts and patient organization leaders from participating countries around the world, and a Patient Advisory Board with 10 patient representatives from 6 countries.
SPIN has been able to develop and conduct robust tests of patient-oriented tools in a rare-disease context by (1) leveraging the combined efforts of hundreds of members of the scleroderma community from across the globe; (2) adapting well-tested patient support strategies from other chronic diseases to meet the needs of people with scleroderma and making them accessible by delivering them via the internet; and (3) utilizing novel research design and management methods to reduce costs dramatically in a resource-starved, but expensive, rare disease research and patient-care environment.
SPIN was originally launched in 2011 with a donation of US $30,000 from the Scleroderma Society of Ontario and Scleroderma Canada. Since then, SPIN has leveraged this seed funding into research grants totaling approximately $3.5 million, primarily from the Canadian Institutes of Health Research. To achieve its goals, SPIN counts on a core team of over 50 investigators, its active Patient Advisory Board, and clinical and investigative staff from 46 treatment centres in 7 countries (Canada, United States, United Kingdom, France, Spain, Mexico, Australia). Soon, SPIN will expand to the Netherlands also. SPIN maintains an ongoing cohort of over 1,800 active participants, who complete assessments every 3 months that help the SPIN team to understand their needs and who are available to test interventions.
SPIN’s mission is to work with people with scleroderma and other stakeholders to identify their needs and prioritize research in areas most important to people living with the disease in order to develop, test and disseminate accessible interventions that improve quality of life and empower people living with scleroderma and their loved ones.
SPIN advances science by developing and demonstrating a novel, reproducible model for large-scale research in rare diseases and by providing patients and researchers with training to effectively address challenges and opportunities in rare disease research.
(Names of members of research team staff and students in bold)
Delisle VC, Gumuchian ST, El-Baalbaki G, Körner A, Malcarne VL, Peláez S, Carrier M-E, Pépin M, Thombs BD, Scleroderma Support Group Project Advisory Team. Training and support needs of scleroderma support group facilitators: the North American Scleroderma Support Group Facilitators Survey. Disabil Rehabil. In press.
Gumuchian ST, Delisle VC, Kwakkenbos L, Pépin M, Carrier M-E, Malcarne CL, Peláez S, El-Baalbaki G, Thombs BD, Scleroderma Support Group Project Advisory Team. Reasons for attending support groups and organizational preferences: the European Scleroderma Support Group Members Survey. Disabil Rehabil. In press.
Kwakkenbos L, Sanchez T, Turner KA, Mouthon L, Carrier ME, Hudson M, van den Ende CHM, Schouffoer AA, Welling J, Sauvé M, Thombs BD, and the SPIN Investigators. The association of sociodemographic and disease variables with hand function: A Scleroderma Patient-centered Intervention Network Cohort Study. Clin Exp Rheum. 2018;36(Suppl. 113):S88-S94.
Carrier M-E, Kwakkenbos L, Boutron I, Welling J, Sauvé M, van den Ende C, Schouffoer AA, Hudson M, Thombs BD, Mouthon L, SPIN Investigators. Randomized feasibility trial of the Scleroderma Patient-centered Intervention Network hand exercise program (SPIN-HAND): study protocol. J Scleroderma Relat Disord. 2018;3(1):91-97.
Rice DB, Cañedo Ayala M, Turner KA, Gumuchian ST, Malcarne VL, Hagedoorn M, Thombs BD, Scleroderma Caregiver Advisory Committee. Use of the nominal group technique to identify stakeholder priorities and inform survey development: an example with informal caregivers of people with scleroderma. BMJ Open. 2018;8:e019726.
Kwakkenbos L, Thombs BD, Khanna D, Carrier M-E, Baron M, Furst D, Gottesman K, van den Hoogen F, Malcarne VL, Mayes MD, Mouthon L, Nielson WR, Poiraudeau S, Riggs R, Sauvé M, Wigley F, Hudson M, Bartlett S, SPIN Investigators. Performance of the Patient-Reported Outcomes Measurement Information System-29 (PROMIS-29) in scleroderma and associations with clinical characteristics: A Scleroderma Patient-centered Intervention Network (SPIN) Cohort study. Rheumatology. 2017;56(8):1302-1311.
Levis AW, Harel D, Kwakkenbos L, Carrier M-E, Mouthon L, Poiraudeau S, Bartlett SJ, Khanna D, Malcarne VL, Sauve M, Poole JL, Schouffoer AA, Steele RJ, van den Ende CHM, Welling J, Thombs BD, SPIN Investigators. Development and Validation of the Cochin Hand Function Scale – Short Form (CHFS-6) Using Optimal Test Assembly Methods: A Scleroderma Patient-centered Intervention Network (SPIN) Cohort Study. Arthritis Care Res. 2016;68(11):1704-1713.
Kwakkenbos L, Jewett LR, Baron M, Bartlett SJ, Furst D, Gottesman K, Khanna D, Malcarne VL, Mayes MD, Mouthon L, Poiraudeau S, Sauve M, Nielson WR, Poole JL, Assassi S, Boutron I, Ells C, van den Ende CHM, Hudson M, Impens A, Körner A, Leite C, Costa Maia A, Mendelson C, Pope J, Steele RJ, Suarez-Almazor ME, Ahmed S, Coronado-Montoya S, Delisle VC, Gholizadeh S, Jang Y, Levis B, Milette K, Mills SD, Razykov I, Sobel RM, Thombs BD. The Scleroderma Patient-centered Intervention Network (SPIN) Cohort: protocol for a cohort multiple randomized controlled trial (cmRCT) design to support trials of psychosocial and rehabilitation intervention trials in a rare disease context. BMJ Open. 2013;3:e003563.