Randomized Controlled Trial of the Scleroderma Patient-centered Intervention Network Hand Exercise Program

For individuals with SSc, skin and tendon involvement of the hands with significant functional impairment is nearly universal. Hand function limitations impact disability more than any other aspect of the disease. However, given the challenges of conducting high-quality, adequately powered clinical trials and disseminating disease management tools in a rare disease context, proven rehabilitation interventions, including those for hands, are typically not available to rare disease patients. To address this gap, the Scleroderma Patient-centered Intervention Network (SPIN) was founded to (1) assemble a large, multinational cohort of SSc patients as a framework for large-scale rehabilitation and self-management intervention trials; and (2) to develop, test, and disseminate effective disease management tools to patients with SSc.

The SPIN Cohort currently includes over 2000 English- and French-speaking patients from over 40 sites in Canada, the USA, France, and the United Kingdom. SPIN Cohort patients complete outcome measures via the internet upon enrolment and subsequently every 3 months. SPIN utilizes the cohort multiple RCT (cmRCT) design as a framework for conducting trials of interventions. SPIN Cohort patients consent to allow their data to be used for observational research, to assess intervention trial eligibility and, if eligible, be randomized. They also consent that if eligible and randomized to usual care, their data can be used to evaluate intervention effectiveness without them being notified that they have been randomized to the usual care group and not offered the intervention. Thus, in SPIN trials, trial status is masked for patients in the control arm, but not the intervention arm.

The SPIN-HAND trial will evaluate the effect on hand function and HRQL of offering SPIN's online hand exercise program, in addition to usual care, to Cohort patients who have at least mild hand function limitations. The SPIN hand exercise program was designed by SPIN experts in rehabilitation, behavioural therapies, and e-health interventions, as well as patient representatives in SPIN. The program core consists of 4 modules that address specific aspects of hand function, including (1) Thumb Flexibility and Strength (3 exercises); (2) Finger Bending (3 exercises); (3) Finger Extension (3 exercises); and (4) Wrist Flexibility and Strength (2 exercises). The program also integrates tools to support key components of successful self-management programs, including goal-setting and feedback, social modeling, and mastery experiences. 586 SPIN Cohort participants will be enrolled in SPIN-HAND. Eligible patients will be able to use the online intervention in English or French, have at least mild hand function limitations (Cochin Hand Function Scale (CHFS) ≥ 3) and have indicated high interest in using an online hand exercise intervention (≥6 on 0-10 scale).

Randomization will occur at the time of patients' regular SPIN Cohort assessments. Eligible patients, based on questionnaire responses, will be randomized automatically with a 3:2 ratio, using a feature in the SPIN Cohort platform, which provides immediate randomization and complete allocation sequence concealment. A 3:2 ratio is being used in order to increase the number of patients who receive the intervention and to support secondary analyses that examine compliers versus non-compliers.

Trial outcomes will be assessed immediately pre-randomization and at 3 months, 6 months, 12 months, and 24 months post-randomization via the SPIN Cohort for both patients randomized to be offered the intervention and patients who receive usual care only. The primary outcome analysis will compare CHFS scores between eligible patients offered the intervention and those not offered the intervention at 3 months post-randomization. Secondary outcomes will include patient-reported health status, usage logs, user satisfaction, and HRQL.