{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,8]],"date-time":"2026-02-08T13:28:12Z","timestamp":1770557292911,"version":"3.49.0"},"reference-count":54,"publisher":"BMJ","issue":"8","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["BMJ Open"],"accepted":{"date-parts":[[2013,7,10]]},"published-print":{"date-parts":[[2013,8]]},"abstract":"<jats:sec><jats:title>Introduction<\/jats:title><jats:p>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.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods and analysis<\/jats:title><jats:p>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\u20132000 patients from centres across the world within a period of 5\u2005years (2013\u20132018). Eligible participants are persons \u226518\u2005years of age with a diagnosis of SSc. In addition to baseline medical data, participants will complete patient-reported outcome measures every 3\u2005months. 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.<\/jats:p><\/jats:sec><jats:sec><jats:title>Ethics and dissemination<\/jats:title><jats:p>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.<\/jats:p><\/jats:sec>","DOI":"10.1136\/bmjopen-2013-003563","type":"journal-article","created":{"date-parts":[[2013,8,9]],"date-time":"2013-08-09T00:58:25Z","timestamp":1376009905000},"page":"e003563","source":"Crossref","is-referenced-by-count":113,"title":["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"],"prefix":"10.1136","volume":"3","author":[{"given":"Linda","family":"Kwakkenbos","sequence":"first","affiliation":[]},{"given":"Lisa R","family":"Jewett","sequence":"additional","affiliation":[]},{"given":"Murray","family":"Baron","sequence":"additional","affiliation":[]},{"given":"Susan J","family":"Bartlett","sequence":"additional","affiliation":[]},{"given":"Dan","family":"Furst","sequence":"additional","affiliation":[]},{"given":"Karen","family":"Gottesman","sequence":"additional","affiliation":[]},{"given":"Dinesh","family":"Khanna","sequence":"additional","affiliation":[]},{"given":"Vanessa L","family":"Malcarne","sequence":"additional","affiliation":[]},{"given":"Maureen D","family":"Mayes","sequence":"additional","affiliation":[]},{"given":"Luc","family":"Mouthon","sequence":"additional","affiliation":[]},{"given":"Serge","family":"Poiraudeau","sequence":"additional","affiliation":[]},{"given":"Maureen","family":"Sauve","sequence":"additional","affiliation":[]},{"given":"Warren R","family":"Nielson","sequence":"additional","affiliation":[]},{"given":"Janet L","family":"Poole","sequence":"additional","affiliation":[]},{"given":"Shervin","family":"Assassi","sequence":"additional","affiliation":[]},{"given":"Isabelle","family":"Boutron","sequence":"additional","affiliation":[]},{"given":"Carolyn","family":"Ells","sequence":"additional","affiliation":[]},{"given":"Cornelia HM","family":"van den Ende","sequence":"additional","affiliation":[]},{"given":"Marie","family":"Hudson","sequence":"additional","affiliation":[]},{"given":"Ann","family":"Impens","sequence":"additional","affiliation":[]},{"given":"Annett","family":"K\u00f6rner","sequence":"additional","affiliation":[]},{"given":"Catarina","family":"Leite","sequence":"additional","affiliation":[]},{"given":"Angela","family":"Costa Maia","sequence":"additional","affiliation":[]},{"given":"Cindy","family":"Mendelson","sequence":"additional","affiliation":[]},{"given":"Janet","family":"Pope","sequence":"additional","affiliation":[]},{"given":"Russell J","family":"Steele","sequence":"additional","affiliation":[]},{"given":"Maria E","family":"Suarez-Almazor","sequence":"additional","affiliation":[]},{"given":"Sara","family":"Ahmed","sequence":"additional","affiliation":[]},{"given":"Stephanie","family":"Coronado-Montoya","sequence":"additional","affiliation":[]},{"given":"Vanessa C","family":"Delisle","sequence":"additional","affiliation":[]},{"given":"Shadi","family":"Gholizadeh","sequence":"additional","affiliation":[]},{"given":"Yeona","family":"Jang","sequence":"additional","affiliation":[]},{"given":"Brooke","family":"Levis","sequence":"additional","affiliation":[]},{"given":"Katherine","family":"Milette","sequence":"additional","affiliation":[]},{"given":"Sarah D","family":"Mills","sequence":"additional","affiliation":[]},{"given":"Ilya","family":"Razykov","sequence":"additional","affiliation":[]},{"given":"Rina S","family":"Fox","sequence":"additional","affiliation":[]},{"given":"Brett D","family":"Thombs","sequence":"additional","affiliation":[]}],"member":"239","published-online":{"date-parts":[[2013,8,7]]},"reference":[{"key":"2024051511062904000_3.8.e003563.1","unstructured":"Canadian Medical Association. Health care transformation in Canada . 2010. http:\/\/www.cma.ca\/multimedia\/CMA\/Content_Images\/Inside_cma\/Advocacy\/HCT\/HCT-2010report_en.pdf"},{"key":"2024051511062904000_3.8.e003563.2","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.322.7284.444"},{"key":"2024051511062904000_3.8.e003563.3","unstructured":"Lewis S . Patient-centered care: an introduction to what it is and how to achieve it. A discussion paper for the Saskatchewan Ministry of Health . 2009. http:\/\/www.health.gov.sk.ca\/Default.aspx?DN=49372758-a388-4e95-8fe2-043c18362458"},{"key":"2024051511062904000_3.8.e003563.4","doi-asserted-by":"crossref","first-page":"79","DOI":"10.3138\/ijfab.4.2.79","article-title":"Relational autonomy as an essential component of patient-centered care","volume":"4","author":"Ells","year":"2011","journal-title":"Int J Fem Approaches Bioeth"},{"key":"2024051511062904000_3.8.e003563.5","unstructured":"Multidisciplinary care for patients with rheumatic and musculoskeletal diseases: The Rheumatology Health Care Team. http:\/\/www.rheumatology.org\/practice\/clinical\/position\/multidisciplinaryCare.pdf ."},{"key":"2024051511062904000_3.8.e003563.6","doi-asserted-by":"crossref","first-page":"1484","DOI":"10.1007\/s00103-007-0382-8","article-title":"[Patient-centred care in rare diseases. A patient organisations\u2019 perspective]","volume":"50","author":"Reimann","year":"2007","journal-title":"Bundesgesundheitsblatt Gesundheitsforschung Gesundheitsschutz"},{"key":"2024051511062904000_3.8.e003563.7","unstructured":"National Institutes of Health Office of Rare Diseases Research http:\/\/rarediseases.info.nih.gov\/RareDiseaseList.aspx?PageID=1 ."},{"key":"2024051511062904000_3.8.e003563.8","doi-asserted-by":"publisher","DOI":"10.1097\/00006199-200609000-00002"},{"key":"2024051511062904000_3.8.e003563.9","doi-asserted-by":"publisher","DOI":"10.1037\/a0017633"},{"key":"2024051511062904000_3.8.e003563.10","doi-asserted-by":"publisher","DOI":"10.1037\/1082-989X.3.1.23"},{"key":"2024051511062904000_3.8.e003563.11","doi-asserted-by":"publisher","DOI":"10.1001\/archpsyc.63.5.484"},{"key":"2024051511062904000_3.8.e003563.12","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2288-10-1"},{"key":"2024051511062904000_3.8.e003563.13","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2648.2001.01757.x"},{"key":"2024051511062904000_3.8.e003563.14","doi-asserted-by":"publisher","DOI":"10.4104\/pcrj.2010.00062"},{"key":"2024051511062904000_3.8.e003563.15","doi-asserted-by":"crossref","first-page":"82","DOI":"10.1097\/NNR.0b013e3181ffbcf2","article-title":"Computer intervention impact on psychosocial adaptation of rural women with chronic conditions","volume":"60","author":"Weinert","year":"2011","journal-title":"Nurs Res"},{"key":"2024051511062904000_3.8.e003563.16","doi-asserted-by":"crossref","first-page":"1181","DOI":"10.1002\/acr.20187","article-title":"Psychological health and well-being in systemic sclerosis: State of the science and consensus research agenda","volume":"62","author":"Thombs","year":"2010","journal-title":"Arthritis Care Res (Hoboken)"},{"key":"2024051511062904000_3.8.e003563.17","doi-asserted-by":"publisher","DOI":"10.1136\/ard.2008.096677"},{"key":"2024051511062904000_3.8.e003563.18","unstructured":"Seibold J . Scleroderma. In: Harris ED Budd RC Firestein GS . eds. Kelley's textbook of rheumatology. \u00a0Philadelphia: Elsevier, 2005:1279\u2013308."},{"key":"2024051511062904000_3.8.e003563.19","unstructured":"Wigley FM Hummers LK . Clinical features of systemic sclerosis. In: Hochberg MC Silman AJ Smolen JS . eds Rheumatology. 3rd edn. Philadelphia: Mosby, 2003:1463\u201380."},{"key":"2024051511062904000_3.8.e003563.20","doi-asserted-by":"publisher","DOI":"10.1002\/art.10486"},{"key":"2024051511062904000_3.8.e003563.21","doi-asserted-by":"publisher","DOI":"10.1016\/j.semarthrit.2006.08.002"},{"key":"2024051511062904000_3.8.e003563.22","doi-asserted-by":"publisher","DOI":"10.1016\/j.semarthrit.2007.05.003"},{"key":"2024051511062904000_3.8.e003563.23","doi-asserted-by":"publisher","DOI":"10.1002\/art.24339"},{"key":"2024051511062904000_3.8.e003563.24","doi-asserted-by":"publisher","DOI":"10.1016\/S0889-857X(03)00020-6"},{"key":"2024051511062904000_3.8.e003563.25","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/keq310"},{"key":"2024051511062904000_3.8.e003563.26","doi-asserted-by":"crossref","unstructured":"Jewett LR Haythornthwaite JA Thombs BD . Psychosocial issues and care for patients with systemic sclerosis. In: Varga J Denton CP Wigley FM . eds Scleroderma: frompathogenesis to comprehensive management. New York: Springer, 2012:641\u20138.","DOI":"10.1007\/978-1-4419-5774-0_53"},{"key":"2024051511062904000_3.8.e003563.27","doi-asserted-by":"crossref","unstructured":"Jewett LR Hudson M Thombs BD . A 38-year-old woman with elevated muscle enzymes, Raynaud's phenomenon and positive anti-topoisomerase I antibody: is she depressed? In: Silver RM Denton CP . eds Case studies in systemic sclerosis. New York: Springer, 2011:273\u201380.","DOI":"10.1007\/978-0-85729-641-2_24"},{"key":"2024051511062904000_3.8.e003563.28","first-page":"23","article-title":"New directions for patient-centered care in scleroderma: the Scleroderma Patient-centered Intervention Network","volume":"30","author":"Thombs","year":"2012","journal-title":"Clin Exp Rheum"},{"key":"2024051511062904000_3.8.e003563.29","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0021274"},{"key":"2024051511062904000_3.8.e003563.30","doi-asserted-by":"crossref","first-page":"S4","DOI":"10.5694\/j.1326-5377.2010.tb03685.x","article-title":"The efficacy of internet interventions for depression and anxiety disorders: a review of randomised controlled trials","volume":"192","author":"Griffiths","year":"2010","journal-title":"Med J Aust"},{"key":"2024051511062904000_3.8.e003563.31","doi-asserted-by":"publisher","DOI":"10.1186\/1741-7015-7-79"},{"key":"2024051511062904000_3.8.e003563.32","doi-asserted-by":"crossref","first-page":"594","DOI":"10.1002\/acr.21875","article-title":"Needs for online information and support of patients with systemic sclerosis","volume":"65","author":"Van der Vaart","year":"2013","journal-title":"Arthritis Care Res (Hoboken)"},{"key":"2024051511062904000_3.8.e003563.33","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.c1066"},{"key":"2024051511062904000_3.8.e003563.34","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.a2390"},{"key":"2024051511062904000_3.8.e003563.35","doi-asserted-by":"publisher","DOI":"10.1016\/S0895-4356(99)00141-9"},{"key":"2024051511062904000_3.8.e003563.36","doi-asserted-by":"crossref","unstructured":"Torgerson DJ Torgerson CJ . Designing randomised trials in health, education, and the social sciences: an introduction. Palgrave Macmillan, 2008.","DOI":"10.1057\/9780230583993"},{"key":"2024051511062904000_3.8.e003563.37","doi-asserted-by":"publisher","DOI":"10.1002\/acr.20591"},{"key":"2024051511062904000_3.8.e003563.38","doi-asserted-by":"publisher","DOI":"10.1002\/art.1780401110"},{"key":"2024051511062904000_3.8.e003563.39","doi-asserted-by":"publisher","DOI":"10.1016\/j.jad.2008.06.026"},{"key":"2024051511062904000_3.8.e003563.40","doi-asserted-by":"publisher","DOI":"10.1016\/j.genhosppsych.2010.03.006"},{"key":"2024051511062904000_3.8.e003563.41","doi-asserted-by":"crossref","first-page":"753","DOI":"10.1002\/acr.21894","article-title":"The utility of the PHQ-9 to assess suicide risk in patients with systemic sclerosis","volume":"65","author":"Razykov","year":"2013","journal-title":"Arthritis Care Res (Hoboken)"},{"key":"2024051511062904000_3.8.e003563.42","doi-asserted-by":"publisher","DOI":"10.1037\/\/1040-3590.10.1.64"},{"key":"2024051511062904000_3.8.e003563.43","doi-asserted-by":"crossref","first-page":"1779","DOI":"10.1002\/acr.20307","article-title":"Development and validation of the brief-satisfaction with appearance scale for systemic sclerosis","volume":"62","author":"Jewett","year":"2010","journal-title":"Arthritis Care Res (Hoboken)"},{"key":"2024051511062904000_3.8.e003563.44","doi-asserted-by":"publisher","DOI":"10.1016\/j.bodyim.2006.11.002"},{"key":"2024051511062904000_3.8.e003563.45","first-page":"1167","article-title":"Development and validation of a rheumatoid hand functional disability scale that assesses functional handicap","volume":"23","author":"Duruoz","year":"1996","journal-title":"J Rheumatol"},{"key":"2024051511062904000_3.8.e003563.46","first-page":"256","article-title":"Effect of a self-management program on patients with chronic disease","volume":"4","author":"Lorig","year":"2001","journal-title":"Eff Clin Pract"},{"key":"2024051511062904000_3.8.e003563.47","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.325.7367.766"},{"key":"2024051511062904000_3.8.e003563.48","doi-asserted-by":"publisher","DOI":"10.1503\/cmaj.051625"},{"key":"2024051511062904000_3.8.e003563.49","doi-asserted-by":"publisher","DOI":"10.1191\/0962280205sm403oa"},{"key":"2024051511062904000_3.8.e003563.50","unstructured":"Clinical Trials Registry. http:\/\/www.clinicaltrials.gov ."},{"key":"2024051511062904000_3.8.e003563.51","doi-asserted-by":"publisher","DOI":"10.7326\/0003-4819-148-4-200802190-00008"},{"key":"2024051511062904000_3.8.e003563.52","doi-asserted-by":"crossref","first-page":"W60","DOI":"10.7326\/0003-4819-148-4-200802190-00008-w1","article-title":"Methods and processes of the CONSORT Group: example of an extension for trials assessing nonpharmacologic treatments","volume":"148","author":"Boutron","year":"2008","journal-title":"Ann Intern Med"},{"key":"2024051511062904000_3.8.e003563.53","unstructured":"Draft policy on access to CIHR-funded research output. http:\/\/www.cihr-irsc.gc.ca\/e\/32326.html ."},{"key":"2024051511062904000_3.8.e003563.54","unstructured":"Taillefer SS Bernstein J Schieir O . Canadian scleroderma patient survey of health concerns and research priorities: an initiative of the Scleroderma Society of Canada and the Canadian Scleroderma Research Group. Report. Montr\u00e9al, 2010."}],"container-title":["BMJ Open"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bmjopen-2013-003563","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,5,15]],"date-time":"2024-05-15T18:06:48Z","timestamp":1715796408000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmjopen.bmj.com\/lookup\/doi\/10.1136\/bmjopen-2013-003563"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2013,8]]},"references-count":54,"journal-issue":{"issue":"8","published-print":{"date-parts":[[2013,8]]}},"alternative-id":["10.1136\/bmjopen-2013-003563"],"URL":"https:\/\/doi.org\/10.1136\/bmjopen-2013-003563","relation":{},"ISSN":["2044-6055","2044-6055"],"issn-type":[{"value":"2044-6055","type":"print"},{"value":"2044-6055","type":"electronic"}],"subject":[],"published":{"date-parts":[[2013,8]]}}}