The Scleroderma Patient-centered Intervention Network - Scleroderma Support Group Leader EDucation Program Trial (SPIN-SSLED)
Phase: NA
Conditions: Scleroderma, Systemic
Sponsor: Lady Davis Institute

THOMBS, BRETT – Ethics Proposal: The Scleroderma Support group Leader Education (SPIN-SSLED) Program 
 
SPIN-SSLED Protocol, Version 2 
July 18, 2019 
 
 
 
 
PROTOCOL TITLE: 
The Scleroderma Patient-centered Intervention Network - Scleroderma Support group Leader 
EDucation (SPIN-SSLED) Program Trial 
 
NCT03965780 
July 18, 2019 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 

THOMBS, BRETT – Ethics Proposal: The Scleroderma Support group Leader Education (SPIN-SSLED) Program 
 
SPIN-SSLED Protocol, Version 2 
July 18, 2019 
 
 
 
 
PROTOCOL TITLE: 
The Scleroderma Patient-centered Intervention Network - Scleroderma Support group Leader 
EDucation (SPIN-SSLED) Program Trial 
 
 
1. THE NEED FOR A TRIAL 
1.1 What is the problem to be addressed? 
People with rare diseases face the same 
challenges as those with more common diseases 
plus unique challenges, including limited 
disease education and lack of specialized 
support options.1-12 Professionally organized 
support services for common diseases are often 
available through the healthcare system,13,14 but 
are not typically available in rare diseases.10,15 
As a result, many people with rare diseases look 
to peer-led support groups for disease-specific 
education and support.16-20 
Support groups provide important benefits to 
people with burdensome medical conditions, based on the principle that people who face similar 
challenges can empower one another through emotional and practical support.14,21 Support groups may 
be held face-to-face or online, led by professionals or peers, and have a structured or an unstructured 
format. Activities typically involve an educational or information-shar ing component and the 
exchange of emotional and practical support.14,18-22 
Systemic sclerosis (SSc), or scleroderma, is a rare, chronic, autoimmune connective tissue disease 
characterized by abnormal fibrotic processes and excessive collagen production.23-25 Peer-led support 
groups play an important role for many people with SSc.17,26 Currently, there are approximately 275 
leaders and co-leaders affiliated with our partners: Scleroderma Canada and Canadian provincial 
organizations, including Sclérodermie Québec; the Scleroderma Foundation (United States); 
Scleroderma & Raynaud’s UK; Scleroderma Australia and Australian state organizations; and 
Scleroderma New Zealand; almost all are led by people with SSc.27-30 Many people with SSc, however, 
cannot access support groups, and many initiated support groups are not sustained due to challenges 
that could be addressed via leader training.18,19 Our partner organizations are committed to improving 
support group quality and access by providing training to existing support group leaders and to new 
leaders so that they can start groups in underserved areas and via the Internet. Our Scleroderma Patient-
centered Intervention Network (SPIN) team has partnered with patient organization leaders and a team 
of scleroderma support group leaders to develop the Scleroderma Support group Leader EDucation 
(SPIN-SSLED) Program. The program is a 3-month group videoconference training program, designed 
to improve skills and self-efficacy, reduce burden, and reduce emotional distress among support group 
leaders. Our organizational partners have worked with us to develop the program, and they plan to 
provide the program to support group leaders post-trial. 
Table 1: Abbreviations 
CONSORT 
Consolidated Standard of 
Reporting Trials 
ICC 
Intra-class Correlation Coefficient 
PHQ-8 
Patient Health Questionnaire-8 
OLBI 
Oldenburg Burnout Inventory 
PN-RCT  
Partially Nested Randomized 
Controlled Trial 
RCT 
Randomized Controlled Trial 
SSc 
Systemic Sclerosis 
SPIN-SSLED 
Program 
SPIN Scleroderma Support group 
Leader EDucation Program 
SPIN 
Scleroderma Patient-centered 
Intervention Network 
SSGLSS 
 
VSI 
Scleroderma Support Group 
Leader Self-efficacy Scale 
Volunteer Satisfaction Index 

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We recently completed a feasibility trial of the SPIN-SSLED Program,31 we are requesting ethical 
approval to conduct a full-scale trial of the SPIN-SSLED Program. 
1.2 What are the principal research questions to be addressed? We will evaluate the effect of the 
SPIN-SSLED Program on support group leaders’ self-efficacy (defined as their perceived ability to 
carry out actions needed to be successful in support group leadership),32 burnout, satisfaction with 
leading a support group, and emotional distress. 
1.2.1. Primary objective: To evaluate the effect of the SPIN-SSLED Program on support group 
leaders’ self-efficacy, measured by the Scleroderma Support Group Leader Self-efficacy Scale 
(SSGLSS)33 post-intervention. 
1.2.2. Secondary objectives: To evaluate the program’s effects on (1) the SSGLSS33 at 3 months post-
intervention; (2) burnout, measured by the Oldenburg Burnout Inventory (OLBI)34,35 post-intervention 
and at 3 months post-intervention; (3) leader satisfaction that leading a group is helping others, 
measured by the participation efficacy subscale of the Volunteer Satisfaction Index (VSI)36 post-
intervention and at 3 months post-intervention; and (4) emotional distress, measured by the Patient 
Health Questionnaire-8 (PHQ-8)37,38 post-intervention. In addition, we will evaluate participant 
satisfaction among those randomized to the program via the Client Satisfaction Questionnaire-8 (CSQ-
8) post-intervention.39,40 
1.3 Why is a trial needed now? Peer support interventions increase positive health behaviours, self-
efficacy for disease management, and mental health.41-44 In common diseases, peer support services are 
often organized through the healthcare system,13,14 but these services are less accessible to people with 
rare diseases.15 One reason is that there are major obstacles to evaluating and delivering organized 
support (e.g., support groups, peer support) for people with rare diseases. We searched PubMed using 
the names of the approximately 7,000 rare diseases listed in Orphanet’s Orphadata45 but did not find a 
single trial of an organized support program for patients with any rare disease.46 
Between 15,000 and 20,000 Canadians are affected by SSc,47 a rare, chronic, autoimmune connective 
tissue disease.23-25 Onset typically occurs between the ages of 30 and 50 years, and approximately 80% 
of people with SSc are women. Abnormal fibrotic processes that occur in SSc can affect multiple organ 
systems, including the skin, lungs, gastrointestinal tract, and heart and can cause immune dysfunction 
and vascular injury.23-25 Common manifestations include Raynaud’s phenomenon,48 esophageal disease 
and gastrointestinal symptoms, 49,50 and pulmonary disease.25 People with SSc commonly experience 
hand function and mobility limitations, pain, fatigue, sleep problems, pruritus, depression, and body 
image distress from disfigurement (e.g., skin tightening, pigment changes, hand contractures, 
telangiectasias).51-62 Presentation is extremely heterogeneous, and course is highly unpredictable.23-25 
While many people with SSc rely on support groups in order to learn how to better manage physical 
and emotional aspects of living with the disease, the majority of SSc patients are not able to access 
these groups.17-20,27-30 Currently, there are only a handful of support groups delivered via teleconference 
or videoconference.27-30 As such, people with SSc must live close enough to a local group and be able 
to travel to participate.18,19 When local groups do exist, they are sometimes disbanded due to the 
leader’s health worsening or to issues related to untrained peer leaders. Some patients prefer not to 
attend SSc support groups because the group in their area is poorly organized or is overly negative.18,19 
Our research in SSc and research in other diseases, including cancer, has established that leading a 
support group poses significant challenges and a high level of burden for patient leaders, often resulting 
in burnout. Peer leaders of illness-based support groups report challenges that include practical 

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difficulties, such as a lack of resources or poor coordination with medical professionals; difficulties 
with group leadership tasks, such as managing complex group dynamics or dealing with the worsening 
health or death of group members; and personal challenges, such as balancing personal and group 
demands, preventing burnout and stress, and managing one’s own health condition while supporting 
others.18-20,63-67 These challenges are magnified for peer leaders of rare disease support groups, who 
also face logistical problems related to small numbers of potential group members, even in urban 
settings, and limited support from healthcare and patient organizations, which are not as well-resourced 
as organizations for people with more common diseases, such as cancer, heart disease, or arthritis.20,46 
By providing key knowledge and skills, the SPIN-SSLED Program could improve the ability of SSc 
peer support group leaders to lead sustainable, effective support groups; reduce emotional and physical 
toll on leaders; and encourage new leaders to set up support groups where none exist, locally or via the 
Internet. The program will be delivered by videoconference because in rare diseases, including SSc, 
support group leaders are widely dispersed geographically. Videoconferencing has been used 
successfully to train educators, therapists, other health service providers, and parents of children with 
behavioural difficulties, for instance.68-74 Systematic reviews have found that training healthcare 
service providers via videoconferencing achieves similar learning outcomes as traditional face-to-face 
models.73,74 Most SSc patients use the Internet. As an example, a 2013 study75 found that 85% of Dutch 
SSc patients used the Internet to search for information about SSc. In Canada, SPIN15 uses Internet-
based data collection methods to support an international SSc patient cohort (N > 2000) and is 
currently testing patient self-management tools delivered via the Internet.76,776 
1.3.1 Evidence from systematic reviews: We conducted a systematic review of trials that evaluated the 
effects of training programs for patient leaders of illness-based support groups on the competency, self-
efficacy, burden, and emotional well-being of group leaders.78 Only one RCT met inclusion criteria. 
That study evaluated confidence and self-efficacy of cancer support group leaders randomized to either 
4-month long high-resource (N = 29; website, discussion forum, 2-day face-to-face training) or low-
resource (N = 23; website, discussion forum) interventions. The RCT did not find evidence that the 
high-resource program was more effective. However, the trial was substantially underpowered, not 
enough information was provided to determine intervention content or how it was delivered, and the 
risk of bias was high due to methodological limitations. We updated the systematic review through 
June 4, 2018, but did not identify any additional trials.79 
1.3.2. Patient interviews and scleroderma support group survey: Prior to developing the SPIN-SSLED 
Program, to assess need and inform program development, we sought information on (1) reasons why 
people with SSc attend or do not attend support groups, (2) the perceived benefits and limitations of 
participating in support groups, and (3) the training and support needs of SSc support group leaders. To 
do this, our team, which includes researchers, leaders from our partner patient organizations, and a 
Support Group Leader Advisory Team of 10 SSc support group leaders, conducted a series of 
studies.17-20 
We first conducted one-on-one interviews with 30 SSc support group leaders, group members, and 
non-attenders. Then, we developed and disseminated the Scleroderma Support Group Survey. We 
generated an initial item pool from our interviews and from surveys done with support groups in more 
common diseases. We worked with our Advisory Team to edit individual items, delete repetitive items 
or items less relevant for SSc, and generate new items to reflect content important to SSc that were not 
reflected in the initial item pool. We disseminated the survey to SSc support group leaders, members, 
and non-attenders from North America and internationally via (1) postings on SSc organization 

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websites and other social media venues (e.g., Facebook, Twitter); (2) announcements in SSc patient 
newsletters; (3) emails to support group leaders and members; and (4) postings in SSc-related chat 
rooms. Approximately 600 North American patients and 700 SSc patients from other parts of the world 
completed the survey (approximately 45% non-attenders, 40% group members, and 15% leaders).17-20 
From the interviews and survey,17-20 we learned that important reasons for attending SSc support 
groups include giving and receiving emotional and practical support, learning how to manage SSc-
related challenges, feeling supported by others with a rare disease, and learning about SSc and SSc 
research from group members and guest speakers. We learned that many people with SSc do not attend 
support groups due to factors that include not having access to a local support group, being too ill or 
disabled to travel, and negative perceptions about support groups (e.g., they are too negative, would not 
provide useful information). Support group leaders consistently reported difficulty performing tasks 
necessary to successfully initiate and sustain the groups (e.g., publicizing the group), coping with the 
emotional demands of leading the group, and managing complex group dynamics. Among support 
group members, 88% indicated that it was important that groups have trained leaders. 
Thus, our research made it clear that there is a need for a well-designed and executed trial of a training 
program for peer leaders of SSc support groups. We recently completed a feasibility trial31 that 
demonstrated that delivering the SPIN-SSLED Program is highly feasible and that it meets the training 
needs of support group leaders. It also provided preliminary evidence that the program has the potential 
to improve self-efficacy for carrying out leadership tasks, reduce burden on leaders, and improve 
emotional distress levels in leaders (see section 2.19 for details of SPIN-SSLED Feasibility Trial). 
1.4 How will the results of this trial be used? The preliminary research we have conducted, including 
our feasibility trial, and the SPIN-SSLED Trial, comprise an integrated knowledge translation process, 
in which researchers and stakeholders work together to define research questions, determine the most 
appropriate methodology, collect data and interpret findings, and disseminate results (www.cihr-
irsc.gc.ca/e/45321.html#a3). Our main patient organization partners, Scleroderma Canada and the 
Scleroderma Foundation, with whom we have a long history of collaboration, are working with our 
team to ensure that as many people with SSc as possible have consistent access to effective support 
group services. We have worked closely with them and with our Support Group Leader Advisory Team 
to design each stage of preliminary research, the SPIN-SSLED Program, and the SPIN-SSLED 
feasibility and full-scale trials. Recently, patient organizations from the United Kingdom, Australia, 
and New Zealand joined our team. 
Once tested, SPIN-SSLED will be the only peer support group leader training program that has been 
evaluated in a well-conducted RCT in any disease. If effective, the SPIN-SSLED Program will be 
implemented immediately by Scleroderma Canada and all other patient organization partners to train 
and certify peer support group leaders. These organizations have committed to support ongoing training 
and to provide logistical and other support to groups with certified leaders. Beyond SSc, the SPIN-
SSLED Program will be easily adapted for use in other diseases, and our team is committed to working 
with organizations outside of SSc to develop capacity to provide training for support group leaders. 
1.5 Are there any risks or benefits for participants involved in the trial? The proposed trial 
intervention is of minimal risk. We do not anticipate any safety concerns with the use of the SPIN-
SSLED Program. Participation in the SPIN-SSLED Trial will involve weekly online training sessions, 
completion of online measures, and participation in a post-program interview. Although it is 
hypothesized that the SPIN-SSLED Program will improve leaders’ self-efficacy for performing leader 
tasks, reduce burnout, and reduce emotional distress, it cannot be guaranteed that leaders will receive 

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any benefits from this study. However, information learned from this research may lead to more 
effective SSc support group leader training, which may in turn benefit those living with SSc in the 
future or people with other diseases. There will be no financial compensation for leaders who are 
participating in the SPIN-SSLED Trial. 
1.6 Will participants receive a compensation for their participation in the trial? No compensation 
is offered to take part in the SPIN-SSLED Trial.  
2. THE PROPOSED TRIAL 
2.1 What is the proposed trial design? The proposed study will be a pragmatic RCT that tests 
whether the SPIN-SSLED Program improves support group leader outcomes compared to leaders 
assigned to a waitlist control. Pragmatic RCTs differ from explanatory or mechanistic trials in that they 
are intended to test the effectiveness of adding an intervention to routine practice in order to inform 
practice and policy decisions rather than explain intervention mechanisms.80-82 
Support group leaders assigned to the SPIN-SSLED Program will be individually randomized and then 
clustered into training groups. Members of each training group will interact during videoconference 
training modules. Support group leaders assigned to the waitlist control will not be clustered. They will 
be randomized individually and will only complete trial measures. A standard cluster RCT design is 
used when interventions are delivered to groups, rather than individuals, in order to account for 
dependence between individuals within clusters.83-85 The SPIN-SSLED Trial will need to account for 
clustering in the intervention arm, but not the control arm. Thus, we will use a partially nested RCT 
trial design (PN-RCT).86-88 The PN-RCT design is a hybrid between a conventional RCT with 
individual participant randomization and a cluster RCT, in which pre-existing clusters (e.g., primary 
care practices) are randomized to intervention or control arms. In the PN-RCT design, analyses account 
for dependence within intervention arm clusters, but treat participants assigned to the control arm 
individually, as in a conventional RCT.86-90 Although less common in medical research, PN-RCTs are 
used extensively in educational and behavioural research.86 
The reason that we will use a waitlist control group that will receive the program post-trial is that our 
patient organization partners are invested in providing the training program, regardless of trial 
outcomes, for reasons of organizational liability and in order to support their support group leader 
community, the members of which have expressed a strong desire to receive training. 
The trial will be registered prior to patient enrollment (clinicaltrials.gov) and will be reported in 
accordance with standards articulated in the Consolidated Standard of Reporting Trials (CONSORT) 
statement91,92 and CONSORT extensions for nonpharmacologic trials,93 cluster trials,83 pragmatic 
trials,80 and e-health trials.94 
2.2 What are the planned trial interventions? The SPIN-SSLED Program uses a problem-based 
learning approach, which is a learner-centered approach that integrates theory and practice by 
providing necessary knowledge and skills, presenting complex, real-world problems, and working to 
identify approaches to solving problems.95-98 Each module, or learning session, will introduce a topic 
and provide an overview of key information. In modules that involve managing group or individual 
interactions, videos that we recorded with members of our Support Group Leader Advisory Team will 
show SSc support group leaders faced with a problem or situation similar to those that training group 
participants may encounter in their role as a support group leader. Then, there will be a guided 
discussion among training group participants about possible approaches and solutions. 

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The SPIN-SSLED Program includes 13 modules that are delivered via videoconference over the course 
of the 3-month program in weekly 60- to 90-minute sessions. Module topics include (1) The Leader’s 
Role; (2) Starting a Support Group; (3) Structuring a Support Group Meeting; (4) Scleroderma 101; (5) 
Successful Support Group Culture; (6) Managing Support Group Dynamics; (7) Loss and Grief: The 
Support Group Leader; (8) Loss and Grief: Supporting Group Members; (9) Advertising and 
Recruitment for the Support Group; (10) The Continuity of the Group; (11) Supporting Yourself as a 
Leader; (12) Remote Support Groups; and (13) Transitions in Support Groups. See Appendix 1 for an 
overview of module content, Appendix 2 for the training manual, Appendix 3 for the participant 
manual, and Appendices 4-6 for example module PowerPoint slides. 
All English-language SPIN-SSLED training groups will be facilitated by one instructor, and the 
French-language training groups will be facilitated by a second instructor. In addition to the live 
modules, SPIN-SSLED Program participants will receive a manual that summarizes didactic material 
from sessions. Based on our previous experience and consistent with previous trials of videoconference 
training, 6 support group leaders will be assigned to each training group to maximize effective 
interaction and participation.68-71 Training sessions will be delivered using the GoToMeeting 
videoconferencing platform, a high-performance platform that has been used successfully in similar 
applications72,99,100 and that was used successfully in the SPIN-SSLED Feasibility Trial31 (see section 
2.19). Participants will receive an instruction guide relating to the use of the videoconferencing 
platform to ensure that everyone can log in successfully (see Appendix 7 for the instruction guide). In 
addition to the videoconference sessions, participants will have access to the secure, monitored SPIN-
SSLED online forum to interact with other participants about program content. (See Appendices 8-10 
for forum guidelines and the instruction guide on how to use the chatroom and access session 
recordings). Participants will also have access to an online resource center containing videos and 
educational activities that leaders can use during their support group meetings (see Appendix 11 for 
screenshots of the website). 
All SPIN-SSLED sessions will be video-recorded and audited for fidelity to the program manual by 
two members of the research team. We will use standard methods for evaluating intervention 
fidelity,101 including observation of entire sessions for a randomly selected sample of 25% of sessions. 
Raters will evaluate adherence to each session’s goals and content. Consistent with best-practice 
recommendations for assessing treatment fidelity,101 this will be done using a checklist based on a 
standardized format adapted for the specific components of the SPIN-SSLED Program manual. 
Participants allocated to the waitlist control will be informed that the SPIN-SSLED Program will be 
provided to those on the waitlist at the end of the trial. During the trial, they will complete trial 
measures but will not receive any active intervention as part of the trial. 
2.3 What are the proposed practical arrangements for recruitment, consent, and allocation of 
participants to trial groups? At the initiation of the trial, our partners from Scleroderma Canada and 
Canadian provincial organizations, including Sclérodermie Québec; the Scleroderma Foundation in the 
United States; Scleroderma & Raynaud’s UK; Scleroderma Australia and Australian state 
organizations, and Scleroderma New Zealand will contact group leaders to describe the SPIN-SSLED 
Program and will provide the SPIN team with a list of eligible support group leaders. The description 
will also contain a link to the public SPIN-SSLED page on SPIN’s website 
(www.spinsclero.com/projects/spin-ssled). This webpage describes the SPIN-SSLED program and 
presents both written and video testimonials from participants that have participated in the feasibility 

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trial. Additionally, there is also a video introducing and explaining how the program came to be. (See 
Appendix 12 for a screenshot of the webpage and links to the webpage videos) 
SPIN-SSLED personnel will then send an email invitation with a link to a Qualtrics survey containing 
the consent form, a demographic questionnaire with information about participants’ support group 
experience (e.g., years of experience or new leader), and questions regarding the days and times when 
the interested group leaders could attend training sessions. In addition to describing the study, the 
consent form will explain (1) that some group leaders who enroll in the study will be randomly selected 
every 3 months to participate in the SPIN-SSLED Program and that some others will be allocated to a 
waitlist; (2) that participants randomized to participate in the program plus those allocated to the 
waitlist will complete measures online at the time of randomization post-intervention, and 3 months 
post-intervention; (3) that, depending on the number of leaders who enroll, it is possible that some 
group leaders who enroll will not be selected to receive the training nor be asked to complete trial 
measures as part of the control group; and (4) that, enrolled participants who do not receive the training 
as part of the trial, either because they are selected for the waitlist or because they are not selected for 
the training group or the waitlist, will be offered the training post-trial per our agreement with our 
partner patient organizations. For existing support groups with more than one leader, one co-leader will 
be specified as the primary participant and any others as secondary; only one leader per support group 
will be eligible for inclusion in the trial, and the secondary leaders will only be enrolled if the primary 
leader day and time availabilities do not match those that are able to be provided in the trial. Interested 
leaders will be provided contact information for SPIN-SSLED trial personnel, who will answer any 
questions they may have during the consent process and over the course of the trial. At the initiation of 
the trial, we will send up to 3 emails, one per week, to leaders who do not respond to the initial email or 
enroll in the trial. 
There will be 15 total training groups offered. We will deliver the program to 3 training groups 
simultaneously. Thus, the intervention will be delivered in 5 “waves” with 3 training groups of 6 
participants each per wave, plus 18 participants randomized to the waitlist control per wave. Prior to 
starting a new wave, we will email participants who have not yet been selected for participation in a 
prior wave to allow them to update their available days and times. We will then determine 
characteristics (language, day, time) of the training groups that are needed for the new wave.  
Interested leaders who provide consent for participation will be entered into different pools based on 
their availabilities. For each wave, the random selection of leaders to be allocated to the intervention 
and waitlist trial arms will be conducted by a third-party centralized randomization service, the Griffith 
Randomisation Service (https://www151.griffith.edu.au/). External centralized randomization will 
ensure that the allocation sequence is concealed and not able to be influenced by study investigators.101 
For each of the 3 new training groups within each wave, SPIN-SSLED personnel will provide the 
Griffith Randomisation Service with an anonymized list of participants (only ID numbers will be 
provided) who could participate in the training group based on their day and time availabilities. For 
each of the 3 groups, the service will randomly select 12 participants from the pool of enrolled group 
leaders available during the designated day and time for the group and will randomly allocate the 6 to 
the training group and 6 to the waitlist group using block randomization. To maximize sharing of 
experiences in groups, we will limit the number of new group leaders without prior experience to 1-2 
per training group, depending on the number of new leaders who enroll (to be determined). Thus, the 
maximum number of new leaders per 12 selected will be either 2 or 4, and randomization will be 
stratified by new and existing leaders. 

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All 12 leaders (training group = 6, waitlist= 6) will receive an email invitation including a clickable 
link to the online survey platform Qualtrics, where they will be asked to complete the study baseline 
measures. This email will also communicate participants’ assignment to the training program or waitlist 
control. A second email will be sent to leaders allocated to the training group with the date and time of 
their first training session, the topic of the first session, the program manual, and information on how to 
login to the SPIN-SSLED videoconferencing system and online chatroom. 
2.4 What are the proposed methods for protecting against sources of bias? A potential concern is 
that participants will not be blinded to intervention status. In most pragmatic trials of training, 
education, or behavioural interventions, participants cannot be blinded. This is understood as part of the 
response to being offered a treatment, similar to what occurs in clinical practice.80-82 A second concern 
relates to the potential for contamination if participants randomized to the SPIN-SSLED Program share 
learning material with participants in the waitlist control. It is not likely that material would be shared 
between leaders from different support groups. Nonetheless, to attempt to minimize the influence of 
possible contamination, we will explain this concern to participants in the training arm of the trial and 
ask them not to share their material or discuss the training sessions with other group leaders. 
2.5 What are the planned inclusion/exclusion criteria? There are currently approximately 275 
leaders or co-leaders affiliated with our partner patient organizations, not counting potential new group 
leaders. To be eligible, support group leaders must be on one of our partner organizations’ lists of 
current or potential leaders, must be able to use the Internet to access training sessions, and must be 
able to complete study questionnaires online. In addition to these requirements, we will only enroll one 
support group leader per support group. In the case where there are multiple leaders for a single group, 
the co-leaders must come to a decision together on who they would like to be the primary leader and 
prioritized for enrolment. The leader(s) designated as secondary will only be enrolled if the primary 
leader in the group does not have any day and time availabilities that match what is offered as part of 
the trial. Those that are designated as secondary and who do not undergo training will be placed in a 
separate waitlist to undergo training post-trial. 
2.6 What is the proposed duration of treatment period? The program consists of 13 modules that 
will be delivered weekly over a 3-month period. 
2.7 What is the proposed frequency and duration of follow up? Trial outcomes will be assessed at 
the time of randomization, 3 months later (post-intervention), and 6 months later (3 months post-
intervention).  
2.8 What are the proposed primary and secondary outcome measures? A demographics 
questionnaire will be administered to all participants (intervention group and waitlist group) before the 
trial. The demographics questionnaire designed for this study includes basic demographic information, 
such as gender, age and employment status. The questionnaire also includes disease-related variables, 
such as years since scleroderma diagnosis and questions relating to participant’s leadership role, such 
as years in role as support group leader or status as a new leader. 
2.8.1. Primary outcome: The primary outcome analysis will compare SSGLSS33 scores between group 
leaders allocated to the SPIN-SSLED Program versus the waitlist control post-intervention. The 
SSGLSS is a 32-item scale designed to assess SSc support group leader confidence to successfully 
perform leader tasks (e.g., organizational skills), manage group and interpersonal interactions, and 
balance group leadership and self-care needs. The measure reflects the core educational content of the 

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SPIN-SSLED Program. It utilizes a 6-point Likert scale ranging from 1 (strongly disagree) to 6 
(strongly agree), with higher total scores indicating greater self-efficacy. 
We developed the SSGLSS with our Support Group Leader Advisory Team and validated it via our 
patient surveys (see section 1.3.2). Prior to the development of the SSGLSS, there were no existing 
measures of support group leader self-efficacy. We validated the SSGLSS33 in two samples of SSc 
support group leaders (N = 102, N = 55) and found that it had good internal consistency (Cronbach’s 
alpha 0.96 and 0.95) and hypothesis-consistent convergent validity with a burnout measure, the 
OLBI.33,34 In our feasibility trial,31 SSGLSS pre-post difference was large among participants 
(standardized mean difference = 1.70; 1.1 point difference per item), suggesting sensitivity to change 
and further supporting its validity (see section 2.19).  
2.8.2. Secondary outcomes: Secondary outcomes include the SSGLSS 3 months post-intervention and 
other outcome measures post-intervention and 3 months post-intervention. Leader burnout will be 
measured by the OLBI, which has been validated in diverse populations (16 items, 4-point scale from 1 
= strongly disagree to 4 = strongly agree).34,35 Leader satisfaction (participation efficacy) will be 
measured using a modified version of the participation efficacy subscale of the VSI.36 The original 
version of the VSI was validated using a sample of volunteers (N = 327) and was found to be reliable 
and constructually valid.36 The participation efficacy subscale asks respondents to indicate their level of 
satisfaction on 7 items using a 7-point Likert scale from 1 (very dissatisfied) to 7 (very satisfied). 
Emotional distress will be assessed with the PHQ-8.37,38 The PHQ-8 items measure depressive 
symptoms over the last 2 weeks on a 4-point scale, ranging from 0 (not at all) to 3 (nearly every day) 
with higher scores indicating more depressive symptoms. The PHQ-8 performs equivalently to the 
PHQ-9,37 which is a valid measure of depressive symptoms in patients with SSc.38 Participant 
Satisfaction with the SPIN-SSLED Program will be evaluated with the CSQ-8,39,40 a standardized 
measure that is used to assess satisfaction with health services. Items are scored on a Likert scale from 
1 (low satisfaction) to 4 (high satisfaction) with total scores ranging from 8 to 32. The CSQ-8 has been 
widely validated across a range of client populations.39  
2.9 How will the outcome measures be measured at follow up? All outcome measures are self-
report. At baseline, post-intervention, and 3 months post-intervention, SPIN-SSLED personnel will 
email participants with a link to a Qualtrics survey that includes outcome measures. If measures are not 
completed within a week, a reminder email will be sent, followed by a phone call from trial personnel 
10 days from the original email. This is the same method that was used in the SPIN-SSLED Feasibility 
Trial. 
2.10 What is the proposed sample size and what is the justification for the assumptions 
underlying the power calculations? We identified several meta-analyses that have evaluated self-
efficacy in terms of knowledge acquisition and confidence in implementing skills acquired in training 
programs. A 2016 Cochrane review reported a standardized mean difference effect size of 0.87 for 4 
educational interventions designed to change knowledge of sickle cell disease among patients and 
caregivers (standardized mean difference = 1.12 with an outlier study removed).103 Several other meta-
analyses have reported effect sizes of between 0.58 and 0.94.104-107 In the SPIN-SSLED feasibility trial, 
which only included 10 participants, the pre-post change in self-efficacy for carrying out leadership 
tasks was 1.70.31 For an assumed effect size of 0.70, a two-tailed test with  = 0.05, and an intra-class 
correlation coefficient (ICC) of 0.05, N = 75 would provide ≥ 80% power for self-efficacy for carrying 
out leader tasks. There was no loss to follow-up in our feasibility trial. Assuming 20% loss to follow-up 
in the proposed trial, we would need to randomize 94 support group leaders. We believe that this is a 

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conservative power and sample size estimate. First, based on existing systematic reviews and on results 
of our feasibility trial, we believe that the true effect size is likely larger than 0.70. Second, in cluster 
RCTs, ICC values for individual patient outcomes are typically lower than our 0.05 estimate, even 
when different interveners are involved, and we will use the same trainer across groups in each 
language. If the true ICC is lower than our 0.05 estimate, this will result in greater power than 
estimated.108-110 Third, there was no loss to follow-up in our feasibility trial, and in our previous 
completed studies in SSc that required follow-up, loss to follow-up has been 10% or less.58,111 
For the secondary outcomes, burnout and emotional distress, based on published meta-analyses, a 
standardized mean difference effect size of 0.50 represents a clinically meaningful effect size for 
improvement that has been achieved in training programs for managers, caregivers of chronically ill 
patients, and parents of children with difficult behaviour.112-115 This is also considered a clinically 
meaningful effect size for patient-reported health outcomes, including depressive symptoms.116-118 For 
effect size = 0.50, a two-tailed test with  = 0.05, and an ICC of 0.05, N = 146 would provide ≥ 80% 
power for both self-efficacy and patient-reported health outcomes. Assuming 20% loss to follow-up in 
the proposed trial, we would need to randomize 182 support group leaders. 
Members of our Support Group Leader Advisory Team and our patient organization partners have 
emphasized the importance of evaluating the trial’s planned secondary outcomes. Thus, we will attempt 
to enroll 180 participants total (15 training groups of 6 participants; 90 participants in waitlist control) 
in order to have sufficient power to adequately evaluate secondary outcomes. 
2.11 Will health service research issues be addressed? Because SSc support group leaders, who are 
mostly people with SSc, have reported in interviews that leading support groups is burdensome and 
impacts quality of life, we will measure leader burnout with the OLBI, and emotional distress with the 
PHQ-8. We will not perform a formal economic evaluation, but we will collect detailed resource 
utilization data and report this descriptively.  
2.12 What is the planned recruitment rate? How will the recruitment be organized? Over what 
time period will recruitment take place? What evidence is there that the planned recruitment 
rate is achievable? We will recruit 180 eligible leaders who are currently affiliated with our 
organizational partners (approximately 250, excluding co-leaders; 65%) or who are potential leaders 
not currently leading a group, which will add to the pool of eligible participants. Our patient 
organizational partners will contact potential participants. We will identify and recruit potential 
participants starting one to two months prior to initiation of training groups and will continue 
throughout the trial. We believe that this is feasible. To obtain 10 participants in the feasibility trial (see 
section 2.19), our partner organizations provided names of 12 possible participants from their lists, and 
all 12 agreed to participate (although one person withdrew prior to initiation for medical reasons, and 
we invited one of two from the waitlist to replace that participant).31 There appears to be a high-level of 
enthusiasm in the international SSc community about the program. For instance, we have received 10-
20 emails since we launched the feasibility trial from other leaders seeking to participate, even though 
the program was not advertised. Our patient organization partners will advertise the program to leaders, 
will emphasize their support, and will explain that the program will enable leaders to be certified, 
which our organizational partners plan to require for organizational affiliation. 
2.13 Are there likely to be any problems with compliance? On what evidence are the compliance 
figures based? Of the 10 participants in the feasibility trial (see section 2.19), 2 participants missed 2 
of 13 sessions (hospitalization, vacation), and 3 participants missed 1 session each. Overall, session 

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attendance was 95% (123 of 130 sessions). All 10 participants completed all baseline and post-trial 
measures.31 Thus, we believe that compliance concerns will be minimal. 
2.14 What is the likely rate of loss to follow up? On what evidence is the loss to follow-up rate 
based? We believe that the loss to follow-up will be low based on our feasibility trial results (no loss to 
follow-up) and previous studies in SSc. In a study of depression, for instance, where patients were 
recruited from multiple centers around Canada and completed burdensome telephone interviews, 
follow-up one-month post-baseline was completed by 309 of 345 participants (90%).58,111 We have 
conservatively estimated a 20% loss to follow-up. 
2.15 How many centers will be involved? We will recruit participants from lists provided by 
Scleroderma Canada and Canadian provincial organizations, including Sclérodermie Québec, the 
Scleroderma Foundation (United States), Scleroderma & Raynaud’s UK, Scleroderma Australia and 
Australian state organizations, and Scleroderma New Zealand. 
2.16 What is the proposed type of analyses? Analyses will be conducted by a statistician blind to trial 
arm allocation. For the primary outcome analysis (SSGLSS post-intervention), we will use an intent-to-
treat analysis that compares all patients randomly allocated to the SPIN-SSLED Program to all patients 
allocated to the waitlist control. Intervention effect will be estimated using a generalized linear random 
effects model, adjusted for baseline SSGLSS scores. The model will include a random effect to account 
for clustering of participants in the training groups, but not for participants in the waitlist control 
arm.89,91 We will investigate the effects of missing data using multiple imputation analysis. As a 
secondary analysis, we will examine SSGLSS scores post-intervention adjusted for baseline participant 
SSGLSS scores, age, sex, whether or not the leader has SSc, and new versus experienced leader status. 
Analysis of the SSGLSS at 3 months post-intervention will similarly be done with two analyses. 
Analyses of leader burnout and emotional distress outcomes will only include experienced leaders 
because new leader would not yet experience burnout or emotional distress due to the burden of leading 
a group. Analyses of these outcomes will similarly be done (1) controlling for baseline scores only and 
(2) controlling for baseline scores, age, sex, whether or not the leader has SSc, and new versus 
experienced leader status. 
Statistical significance for all analyses will be determined based on two-sided α = 0.05. 
2.17 What is the proposed frequency of analyses? The analyses described above will be performed 
once, at the end of the trial. No interim analyses are planned. 
2.18 Are there any planned subgroup analyses? We will evaluate all outcomes after removing any 
participants who do not have SSc and, for the SSGLSS, after removing participants who are not current 
group leaders. We anticipate that well over 90% will have SSc and well over 90% will be current group 
leaders based on information from our organizational partners. In our feasibility trial, 9 of 10 
participants were people with SSc and 10 of 10 were group leaders, including one who recently 
started.31 
2.19 Has any pilot study been carried out using this design? We conducted a feasibility trial of the 
SPIN-SSLED Program (NCT03508661) that involved delivery of the SPIN-SSLED Program to two 
training groups of 5 participants each.31 Scleroderma Canada and the Scleroderma Foundation each 
provided our team with names of 6 potential participants. All 12 agreed to participate in the program. 
We enrolled 10 initially, but one was hospitalized before the trial began; therefore, we thus added one 
participant who had been wait-listed. 

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Participant attendance was high (95%; 123 of 130 sessions). All 10 participants completed all baseline 
and post-trial measures, including an individual interview guided by the Patient Education Materials 
Assessment Tool for Audiovisual Materials (PEMAT). The interview questions addressed topics 
related to usability, understandability, organization and clarity of the SPIN-SSLED program. We 
produced user-friendly instructions for GoToMeeting, the online user-forum, and the SPIN data 
management platform. No sessions were missed or delayed due to technological difficulties, and time 
for technological support from our team was between 1-2 hours for the entire program.31 
Pre-training, the mean (standard deviation [SD]) SSGLSS score, the primary outcome for the planned 
trial, was 124.4 (22.0), which was similar to the scores of our two international samples from the SSGLSS 
validation study (N = 102, mean [SD] SSGLSS = 122.9 [21.7]; N = 55, mean SSGLSS = 123.9 [19.4]). 
Post-training, the mean total score increased to 159.2 (17.1). The standardized mean difference effect 
size was 1.70, which is considered a large effect size. Items are scored on a 1-6 basis, and the average 
item score increase pre-post training was 1.1 points. For burnout (OLBI) and emotional distress (PHQ-
8), the effect size of post-trial score improvement was between 0.38 and 0.45, which is considered a 
moderate effect size.31 
Participant satisfaction was very high. Mean post-training score on the CSQ-8 was 30.6 (2.2). On a per 
item basis, the mean item score was 3.83 (item range 1-4). On the PEMAT interviews, there were 
relatively minor suggestions for improving the program, and feedback was extremely positive. The 
overall mean grade given by participants for the SPIN-SSLED Program was 9.4/10, and all 10 
participants indicated they would recommend the program to other support group leaders.31 
3. TRIAL  
3.1 What are the arrangements for day to day management of the trial? E.g. randomization, data 
handling, confidentiality, storage and who will be responsible for coordination. The SPIN team, 
led by Dr. Thombs and located at the Jewish General Hospital, has the primary responsibility for study 
design and data analysis, maintaining the protocol and implementation of standard operating 
procedures, conducting recruitment and providing technical support to participants, overseeing trial 
progress, coordinating study logistics with organizational partners, preparing and distributing regular 
progress reports, and monitoring progress. 
Outcome measures will be completed using the online surveying tool Qualtrics. Once the online survey 
data is collected, data will be exported to the statistics software program, IBM SPSS. Cleaning of the 
data will occur within SPSS by members of the study team. All information obtained about the 
participants during this study will be treated confidentially within the limits of the law. Only the 
researchers involved in this project will have access to the survey data. To protect the participants’ 
privacy, upon inclusion in the SPIN-SSLED Trial, a unique participant identification number will 
automatically be assigned to each participant. An encrypted database will be created for the SPIN-
SSLED Program, which includes the patient identification number. Only requests authorized by the 
principal investigator (Dr. Brett Thombs) will be granted access to this encrypted information. The 
survey is run through Qualtrics, a company whose computer servers are located in the USA. Data 
security measures in place at Qualtrics are described in the Qualtrics security statement 
(http://www.qualtrics.com/security-statement/). Information obtained from the survey and video 
recordings of the training sessions used to evaluate fidelity to program will be kept for 10 years on 
encrypted hard drives by the researchers responsible for this study. Access to the data will be limited to 
the investigators of the study at the Jewish General Hospital. 

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3.2 What will be the role of each investigator and co-investigator? As principal investigator, Dr. 
Thombs will oversee trial conduct, data analysis, and knowledge translation activities, and coordinate 
collaboration between the research team and partners. Dr. Thombs is the Founder and Director of 
SPIN, which has a long history of successful collaboration with our partners. His expertise in clinical 
trials and pragmatic trial designs is reflected in his leadership of an international team that has been 
funded by CIHR to develop a CONSORT extension for trials conducted in cohorts and existing data 
sources.119,120 Ms. Maureen Sauvé (Knowledge User) has SSc, is Past-President of Scleroderma 
Canada and the Scleroderma Society of Ontario, and leads the patient support work of these 
organizations. Ms. Kerri Connolly (Knowledge User) is Programs and Services Director for the 
Scleroderma Foundation and responsible for the foundation’s patient support programs. Dr. Linda 
Kwakkenbos, Assistant Professor, Radboud University, is Co-director of SPIN and will work with Dr. 
Thombs on day-to-day trial management. Dr. Linda Horwood is a SPIN Postdoctoral Fellow who will 
work with Dr. Thombs and Dr. Kwakkenbos as coordinator of the trial. Dr. Robert Platt is a 
biostatistician, Professor, and Albert Boehringer I Chair in Pharmacoepidemiology, McGill University. 
He has extensive experience in the design and analyses of of RCTs, including cluster trials. He 
designed and will conduct trial analyses. Dr. Vanessa Malcarne is Professor of Psychology, San 
Diego State University. She has researched psychosocial adjustment and support mechanisms in SSc 
for over 20 years. Dr. Ghassan El-Baalbaki is Associate Professor of Psychology, Université du 
Québec à Montréal. He has expertise in peer support in chronic diseases, including SSc, and in training 
support group leaders. Dr. Sandra Peláez is an investigator with the Lady Davis Institute for Medical 
Research, Jewish General Hospital. She studies coping and social support in chronic diseases, including 
SSc. Drs. Malcarne, El-Baalbaki, and Peláez will oversee training groups and monitor training program 
delivery fidelity. Dr. Marie Hudson is a rheumatologist and Associate Professor, McGill University, 
who is expert in SSc. She contributed to design of educational materials, and who will oversee their use 
in the training program. The SPIN-SSLED Support Group Leader Advisory Team consists of SSc 
patient support group leaders from Canada (Ms. Catherine Fortune, Ms. Geneviève Guillot, Ms. 
Michelle Richard, Mr. Ken Rozee, Ms. Maureen Sauvé) and the United States (Mr. Stephen Elrod, Ms. 
Jacqueline Gerena, Ms. Amy Gietzen, Ms. Karen Gottesman, Ms. Nancy Stephens, Ms. Laura Dyas). 
Members of the Advisory Team were involved in preliminary research and intervention development 
and preliminary testing and will be involved in patient outreach and collaboration. 
3.3 Describe the trial steering committee and if relevant the data safety and monitoring 
committee. The trial will be overseen by the SPIN Steering Committee 
(https://www.spinsclero.com/en/Team?teamID=0d6dd6a6-8bee-62ed-b515-ff0000ce1efe) along with 
the trial investigators. The Steering Committee will provide scientific direction for the RCT and will 
meet periodically to assess its progress. It will be responsible for RCT protocol execution, routine 
monitoring of data quality, and will meet semi-annually to discuss recruitment and retention and to 
assess that the trial is meeting key milestones consistent with the timeline. 
 
 

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