The document summarizes the Scleroderma Patient-centered Intervention Network (SPIN), which aims to develop and evaluate accessible and low-cost psychosocial and rehabilitation interventions for people living with scleroderma. SPIN is a collaboration between patients, clinicians, and researchers from over 30 sites in 8 countries. It involves developing online, self-guided interventions for issues like fatigue, hand function, and emotional coping. SPIN also conducts a large international cohort study and engages patients in its research. The goal is to improve quality of life for people with scleroderma.

1. Brett Thombs, PhD
brett.thombs@mcgill.ca
McGill University and Jewish General Hospital
Montreal, Quebec, Canada
The Scleroderma Patient-centered
Intervention Network:
SPIN

2. Patient-centered Care
• Patient empowerment
• Shared decision-making
• Care plans taking into account patient
preferences and values
• Care that meets the needs of individual patients
• Evidence-based
• In chronic disease:
comprehensive care to reduce disability and
improve health-related quality of life
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3. What about rare diseases?
• Typically no access to psychosocial and
rehabilitation interventions that are:
• Specific to needs of people with the disease
• Adequately tested to determine if effective
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4. Scleroderma
3
• The word scleroderma comes from the greek "scleros"
meaning hard and "derma" meaning skin
• SSc results in autoimmune changes, fibrosis (scarring)
and severe vascular problems (reduced bloodflow to
organs and extremities)

5. Challenges
• To develop supportive interventions
(psychosocial, rehabilitation) that are:
• Accessible to people with scleroderma
• Low cost for feasible implementation
• Can be delivered on an ongoing basis
• To conduct high-quality trials to confidentially
assess impact interventions (including at least
200-300 people)
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6. SPIN: The Scleroderma Patient-centered
Intervention Network
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7. SPIN
• A collaboration of people living with scleroderma,
clinicians and researchers
• More than 50 investigators
• More than 20 people with scleroderma
• More than 30 recruiting sites active or pending from 8
countries
• Aim: To develop and evaluate psychosocial and
rehabilitation interventions that are accessible, low-
cost, and can be delivered on an ongoing basis to
people living with scleroderma
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8. The SPIN Cohort
Large international study with people with
scleroderma (Target N=2,000):
• Online questionnaires every 3 months
• Insight in problems important to patients
• How best to measure these outcomes
• Natural history of scleroderma and treatment as
usual
• Currently enrolled > 600 since 2014
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9. SPIN Interventions
• Specific to living with scleroderma
• Self-guided (no therapist involved)
• Virtual, online
• Engaging (video, animations)
• Support patients in coping with their disease (e.g.,
emotional distress, body image)
• Reduce limitations in daily activities (e.g., exercise,
hand function)
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10. SPIN Interventions
• Fatigue
• Exercises to improve hand function
• Sleep
• Emotional coping
• Social anxiety from visible difference due to disease
• Pain management
• Exercise and fitness
• Nutrition and diet
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11. Partnering with Scleroderma Patient
Organizations
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12. Patient Engagement in Research: CIHR
• Patient engagement occurs when patients
meaningfully and actively collaborate in the
governance, priority setting, and conduct of
research, as well as in summarizing, distributing,
sharing, and applying its resulting knowledge
(i.e., the process referred to as "knowledge
translation").
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13. Patient Engagement in Research
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14. Engaging Patients in Research
• Typically done at beginning of a particular project
• Purposes:
• Increasing enrollment
• Securing funding
• Designing study protocols
• Choosing relevant outcomes
• Challenges:
• Logistical challenges (time, funding, coordination)
• Concern of tokenistic engagement
13
Domecq et al., BMC Health Services Research, 2014

15. Partnering with Scleroderma Patient
Organizations
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16. Drs Baron, Hudson,
Mathieu, Ligier & Grodzicky
Montreal, QC;
Dr. Masetto,
Sherbrooke, QC
Dr. Docherty
Moncton, NB
Dr. Sutton
Halifax, NS
Canadian Scleroderma Research Group
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Dr. Robinson
Winnipeg, MB
Dr. Pope, London &
Drs. Khalidi & Kaminska,
Hamilton, ON
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Dr. Markland
Saskatoon, SK
Dr. Jones
Edmonton, AB
Dr. LeClercq
Calgary, AB
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Dr. Thorne
Newmarket, ON

17. Montreal, QC
Basic Scientists: Anie Philip,
John Mort,
Anneliese Recklies Peter
Roughley; Marc Servant, Mark
Trifiro
Statistician: Russell
Steele; Dentist: Mervyn Gornitsky
Epidemiologist: Marie Hudson ,
Sasha Bernatsky
Psychologist: Brett Thombs
Calgary, AB
ADL Director:
Marvin Fritzler,
London, ON
Basic Scientist: Andrew Leask
Psychologist: Warren Nielson
Vancouver, BC
Basic Scientist:
Stephan van Eeden
Co-investigators
Patient Representative
Collaborators
Bob Buzza, SSC
Grant Dustin, SSC
Marion Pacy, SSC
Normand Ricard, SSC
Antigonish, NS
Phil Hughes
Other CSRG Members Around the Country
Toronto, ON
Maureen Sauvé, SSC
Halifax, NS
Mary Beth Clark

18. Psychological Health and Well-being: A
Consensus Research Agenda
• Fatigue
• Pain
• Depressive symptoms
• Pruritus (Itch)
• Body Image
• Sexual function
• Other areas
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19. The Canadian Scleroderma Patient Survey of
Health Concerns and Healthcare Needs
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• Collaboration between Scleroderma Society of
Canada and CSRG
• Based on:
• Existing surveys from other patient groups
• Patient input
• Health care professional input
• September 2008 to August 2009
• Over 600 people with scleroderma completed
this online or using paper forms distributed by
patient support groups across Canada

20. SPIN Background
• 2010: Consortium for clinical trials of behavioural,
psychological and educational interventions
• 2011: SPIN planning meeting
• Seed Funding from SSO/SSC $37,000
Canadian Institutes for Health Research Team Grant:
• $1.5 million (2012 – 2017) for operating costs
• Additional $300,000 in partner funding
• For SPIN in English and French
• Other languages: national funding (e.g. the Netherlands)
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21. SPIN
Emotional Distress
Hand Function
Body Image
Self-Management
Steering
Committee
Advisory
Board
Cores:
1. Information Technology
2. Data Management
3. Research Methods
and Biostatistics
4. Measurement
5. Health Economics
6. Bioethics
7. Knowledge Translation

22. Scleroderma Support Group Projects
• Started with a conversation over coffee (officially, but
unofficially a beer) in 2009
• Seed funding of $30,000 + investigator funds
• Partnership with SSO/SSC and Scleroderma
Foundation of the US
• Current grant applications submitted, work underway
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23. Researcher-Community Engagement in
Rare Diseases
• Ongoing – not project specific
• Pre-project needs identification and generation
• Engaging researchers
• Meetings
• Funding
• Micro and mid-level
• Recognition
• Focus on early career researchers
• Hands on role in some kinds of research
• Typical roles in Patient Engagement
• Research participation
• Advisory Boards
• Dissemination
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24. Scleroderma Support Group Projects
• Started with a conversation over coffee (officially, but
unofficially a beer) with Maureen Sauve in 2009
• Seed funding of $30,000 from SSO + investigator
funds
• Partnership with SSO/SSC and Scleroderma
Foundation of the US
• Current grant applications submitted, work underway
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25. Acknowledgements
• Patient Organizations:
Scleroderma Society Ontario, Sclérodermie Québec,
Scleroderma Association of British Columbia,
FESCA, Scleroderma Society of Canada,
Scleroderma Foundation
• Linda Kwakkenbos, SPIN coordinator
• Lisa Jewett, SPIN coordinator
• Marie-Eve Carrier, SPIN coordinator
• SPIN researchers (40-50!)
• Canadian Institute of Health Research
• Fonds de la Recherche en Santé Quebec