. 2021 Nov 27;22(1):856.

doi: 10.1186/s13063-021-05827-z.

The Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program: protocol for a two-arm parallel partially nested randomized controlled feasibility trial with progression to full-scale trial

Julia Nordlund^#¹, Richard S Henry^#^{1

2}, Linda Kwakkenbos^{3

4}, Marie-Eve Carrier¹, Brooke Levis⁵, Warren R Nielson⁶, Susan J Bartlett^{7

8}, Laura Dyas⁹, Lydia Tao¹, Claire Fedoruk¹, Karen Nielsen¹⁰, Marie Hudson^{1

7}, Janet Pope¹¹, Tracy Frech¹², Shadi Gholizadeh¹³, Sindhu R Johnson^{14

15}, Pamela Piotrowski¹⁶, Lisa R Jewett¹⁷, Jessica Gordon¹⁸, Lorinda Chung^{19

20}, Dan Bilsker^{21

22}, Alexander W Levis²³, Kimberly A Turner¹, Julie Cumin¹, Joep Welling²⁴, Catherine Fortuné²⁵, Catarina Leite²⁶, Karen Gottesman²⁷, Maureen Sauve^{10

28}, Tatiana Sofía Rodríguez-Reyna²⁹, Maggie Larche³⁰, Ward van Breda³¹, Maria E Suarez-Almazor³², Amanda Wurz³³, Nicole Culos-Reed^{34

35

36}, Vanessa L Malcarne^{37

38}, Maureen D Mayes³⁹, Isabelle Boutron^{40

41}, Luc Mouthon^{42

43}, Andrea Benedetti^{7

44

45}, Brett D Thombs^{46

47

48

49

50

51

52

53}; SPIN Investigators

Collaborators, Affiliations

Collaborators

SPIN Investigators:
Robert Riggs, Shervin Assassi, Ghassan El-Baalbaki, Carolyn Ells, Cornelia van den Ende, Kim Fligelstone, Amy Gietzen, Geneviève Guillot, Daphna Harel, Monique Hinchcliff, Christelle Nguyen, François Rannou, Michelle Richard, Anne A Schouffoer, Christian Agard, Nassim Ait Abdallah, Alexandra Albert, Marc André, Elana J Bernstein, Sabine Berthier, Lyne Bissonnette, Alessandra Bruns, Patricia Carreira, Marion Casadevall, Benjamin Chaigne, Chase Correia, Benjamin Crichi, Christopher Denton, Robyn Domsic, James V Dunne, Bertrand Dunogue, Regina Fare, Dominique Farge-Bancel, Paul R Fortin, Brigitte Granel-Rey, Genevieve Gyger, Eric Hachulla, Ariane L Herrick, Sabrina Hoa, Alena Ikic, Niall Jones, Suzanne Kafaja, Nader Khalidi, Marc Lambert, David Launay, Hélène Maillard, Nancy Maltez, Joanne Manning, Isabelle Marie, Maria Martin, Thierry Martin, Ariel Masetto, François Maurier, Arsene Mekinian, Sheila Melchor, Mandana Nikpour, Louis Olagne, Vincent Poindron, Susanna Proudman, Alexis Régent, Sébastien Rivière, David Robinson, Esther Rodriguez, Sophie Roux, Perrine Smets, Vincent Sobanski, Robert Spiera, Virginia Steen, Evelyn Sutton, Carter Thorne, John Varga, Pearce Wilcox, Angelica Bourgeault, Mara Cañedo Ayala, Andrea Carboni Jiménez, Marie-Nicole Discepola, Maria Gagarine, Nora Østbø

Affiliations

¹ Lady Davis Institute of the Jewish General Hospital, 3755 Côte-Sainte-Catherine Road, Montreal, Quebec, H3T 1E2, Canada.
² Department of Psychiatry, McGill University, Montreal, Quebec, Canada.
³ Department of Clinical Psychology, Radboud University, Nijmegen, The Netherlands.
⁴ Department of Medical Psychology, Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, The Netherlands.
⁵ Centre for Prognosis Research, School of Medicine, Keele University, Keele, Staffordshire, UK.
⁶ St. Joseph's Health Care, London, Ontario, Canada.
⁷ Department of Medicine, McGill University, Montreal, Quebec, Canada.
⁸ Centre for Outcomes Research and Evaluation, Research Institute of the McGill University Health Centre, Montreal, Quebec, Canada.
⁹ Scleroderma Foundation, Michigan Chapter, Southfield, MI, USA.
¹⁰ Scleroderma Society of Ontario, Hamilton, Ontario, Canada.
¹¹ Department of Medicine, University of Western Ontario, London, Ontario, Canada.
¹² Department of Internal Medicine, University of Utah, Salt Lake City, UT, USA.
¹³ California School of Professional Psychology/Alliant, Los Angeles, CA, USA.
¹⁴ Toronto Scleroderma Program, Mount Sinai Hospital & Toronto Western Hospital, Toronto, Ontario, Canada.
¹⁵ Institute of Health Policy, Management, and Evaluation, University of Toronto, Toronto, Ontario, Canada.
¹⁶ Private practice - Nutrition, Milton, Ontario, Canada.
¹⁷ Department of Psychology, Jewish General Hospital, Montreal, Quebec, Canada.
¹⁸ Department of Medicine, Hospital for Special Surgery, New York City, NY, USA.
¹⁹ Department of Medicine, Stanford University, Palo Alto, CA, USA.
²⁰ Department of Medicine, Palo Alto VA Health Care System, Palo Alto, CA, USA.
²¹ Faculty of Health Sciences, Simon Fraser University, Burnaby, British Columbia, Canada.
²² Department of Psychiatry, University of British Columbia, Vancouver, British Columbia, Canada.
²³ Department of Biostatistics, Harvard T. H. Chan School of Public Health, Boston, MA, USA.
²⁴ NVLE Dutch patient organization for systemic autoimmune diseases, Utrecht, The Netherlands.
²⁵ Ottawa Scleroderma Support Group, Ottawa, Ontario, Canada.
²⁶ University of Minho, Braga, Portugal.
²⁷ Scleroderma Foundation, Los Angeles, CA, USA.
²⁸ Scleroderma Canada, Hamilton, Ontario, Canada.
²⁹ Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, Mexico City, Mexico.
³⁰ Department of Health Research Methods, Evidence and Impact, McMaster University, Hamilton, Ontario, Canada.
³¹ Faculty of Behavioural and Movement Sciences, VU University, Amsterdam, The Netherlands.
³² Department of General Internal Medicine, University of Texas MD Anderson Cancer Center, Houston, TX, USA.
³³ School of Kinesiology, University of the Fraser Valley, Chilliwack, British Columbia, Canada.
³⁴ Faculty of Kinesiology, University of Calgary, Calgary, Alberta, Canada.
³⁵ Department of Oncology, Cumming School of Medicine, Calgary, Canada.
³⁶ Department of Psychosocial Resources, Tom Baker Cancer Centre, Alberta Health Services, Calgary, Alberta, Canada.
³⁷ Department of Psychology, San Diego State University, San Diego, CA, USA.
³⁸ Joint Doctoral Program in Clinical Psychology, San Diego State University/University of California San Diego, San Diego, CA, USA.
³⁹ Department of Internal Medicine, University of Texas McGovern School of Medicine, Houston, TX, USA.
⁴⁰ Université de Paris, Centre of Research Epidemiology and Statistics (CRESS), Inserm, INRA, Paris, France.
⁴¹ Centre d'Épidémiologie Clinique, Assistance Publique-Hôpitaux de Paris (AP-HP), Hôpital Hôtel Dieu, Paris, France.
⁴² Service de Médecine Interne, Centre de Référence Maladies Autoimmunes Systémiques Rares d'Ile de France, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (AP-HP), Paris, France.
⁴³ APHP-CUP, Hôpital Cochin, Université de Paris, F-75014, Paris, France.
⁴⁴ Department of Epidemiology, Biostatistics, and Occupational Health, McGill University, Montreal, Quebec, Canada.
⁴⁵ Respiratory Epidemiology and Clinical Research Unit, McGill University Health Centre, Montreal, Quebec, Canada.
⁴⁶ Lady Davis Institute of the Jewish General Hospital, 3755 Côte-Sainte-Catherine Road, Montreal, Quebec, H3T 1E2, Canada. brett.thombs@mcgill.ca.
⁴⁷ Department of Psychiatry, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁴⁸ Department of Medicine, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁴⁹ Department of Epidemiology, Biostatistics, and Occupational Health, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁵⁰ Respiratory Epidemiology and Clinical Research Unit, McGill University Health Centre, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁵¹ Department of Psychology, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁵² Department of Educational and Counselling Psychology, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁵³ Biomedical Ethics Unit, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.

^# Contributed equally.

PMID: 34838105
PMCID: PMC8626736
DOI: 10.1186/s13063-021-05827-z

The Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program: protocol for a two-arm parallel partially nested randomized controlled feasibility trial with progression to full-scale trial

Julia Nordlund et al. Trials. 2021.

. 2021 Nov 27;22(1):856.

doi: 10.1186/s13063-021-05827-z.

Authors

Collaborators

SPIN Investigators:
Robert Riggs, Shervin Assassi, Ghassan El-Baalbaki, Carolyn Ells, Cornelia van den Ende, Kim Fligelstone, Amy Gietzen, Geneviève Guillot, Daphna Harel, Monique Hinchcliff, Christelle Nguyen, François Rannou, Michelle Richard, Anne A Schouffoer, Christian Agard, Nassim Ait Abdallah, Alexandra Albert, Marc André, Elana J Bernstein, Sabine Berthier, Lyne Bissonnette, Alessandra Bruns, Patricia Carreira, Marion Casadevall, Benjamin Chaigne, Chase Correia, Benjamin Crichi, Christopher Denton, Robyn Domsic, James V Dunne, Bertrand Dunogue, Regina Fare, Dominique Farge-Bancel, Paul R Fortin, Brigitte Granel-Rey, Genevieve Gyger, Eric Hachulla, Ariane L Herrick, Sabrina Hoa, Alena Ikic, Niall Jones, Suzanne Kafaja, Nader Khalidi, Marc Lambert, David Launay, Hélène Maillard, Nancy Maltez, Joanne Manning, Isabelle Marie, Maria Martin, Thierry Martin, Ariel Masetto, François Maurier, Arsene Mekinian, Sheila Melchor, Mandana Nikpour, Louis Olagne, Vincent Poindron, Susanna Proudman, Alexis Régent, Sébastien Rivière, David Robinson, Esther Rodriguez, Sophie Roux, Perrine Smets, Vincent Sobanski, Robert Spiera, Virginia Steen, Evelyn Sutton, Carter Thorne, John Varga, Pearce Wilcox, Angelica Bourgeault, Mara Cañedo Ayala, Andrea Carboni Jiménez, Marie-Nicole Discepola, Maria Gagarine, Nora Østbø

Affiliations

¹ Lady Davis Institute of the Jewish General Hospital, 3755 Côte-Sainte-Catherine Road, Montreal, Quebec, H3T 1E2, Canada.
² Department of Psychiatry, McGill University, Montreal, Quebec, Canada.
³ Department of Clinical Psychology, Radboud University, Nijmegen, The Netherlands.
⁴ Department of Medical Psychology, Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, The Netherlands.
⁵ Centre for Prognosis Research, School of Medicine, Keele University, Keele, Staffordshire, UK.
⁶ St. Joseph's Health Care, London, Ontario, Canada.
⁷ Department of Medicine, McGill University, Montreal, Quebec, Canada.
⁸ Centre for Outcomes Research and Evaluation, Research Institute of the McGill University Health Centre, Montreal, Quebec, Canada.
⁹ Scleroderma Foundation, Michigan Chapter, Southfield, MI, USA.
¹⁰ Scleroderma Society of Ontario, Hamilton, Ontario, Canada.
¹¹ Department of Medicine, University of Western Ontario, London, Ontario, Canada.
¹² Department of Internal Medicine, University of Utah, Salt Lake City, UT, USA.
¹³ California School of Professional Psychology/Alliant, Los Angeles, CA, USA.
¹⁴ Toronto Scleroderma Program, Mount Sinai Hospital & Toronto Western Hospital, Toronto, Ontario, Canada.
¹⁵ Institute of Health Policy, Management, and Evaluation, University of Toronto, Toronto, Ontario, Canada.
¹⁶ Private practice - Nutrition, Milton, Ontario, Canada.
¹⁷ Department of Psychology, Jewish General Hospital, Montreal, Quebec, Canada.
¹⁸ Department of Medicine, Hospital for Special Surgery, New York City, NY, USA.
¹⁹ Department of Medicine, Stanford University, Palo Alto, CA, USA.
²⁰ Department of Medicine, Palo Alto VA Health Care System, Palo Alto, CA, USA.
²¹ Faculty of Health Sciences, Simon Fraser University, Burnaby, British Columbia, Canada.
²² Department of Psychiatry, University of British Columbia, Vancouver, British Columbia, Canada.
²³ Department of Biostatistics, Harvard T. H. Chan School of Public Health, Boston, MA, USA.
²⁴ NVLE Dutch patient organization for systemic autoimmune diseases, Utrecht, The Netherlands.
²⁵ Ottawa Scleroderma Support Group, Ottawa, Ontario, Canada.
²⁶ University of Minho, Braga, Portugal.
²⁷ Scleroderma Foundation, Los Angeles, CA, USA.
²⁸ Scleroderma Canada, Hamilton, Ontario, Canada.
²⁹ Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, Mexico City, Mexico.
³⁰ Department of Health Research Methods, Evidence and Impact, McMaster University, Hamilton, Ontario, Canada.
³¹ Faculty of Behavioural and Movement Sciences, VU University, Amsterdam, The Netherlands.
³² Department of General Internal Medicine, University of Texas MD Anderson Cancer Center, Houston, TX, USA.
³³ School of Kinesiology, University of the Fraser Valley, Chilliwack, British Columbia, Canada.
³⁴ Faculty of Kinesiology, University of Calgary, Calgary, Alberta, Canada.
³⁵ Department of Oncology, Cumming School of Medicine, Calgary, Canada.
³⁶ Department of Psychosocial Resources, Tom Baker Cancer Centre, Alberta Health Services, Calgary, Alberta, Canada.
³⁷ Department of Psychology, San Diego State University, San Diego, CA, USA.
³⁸ Joint Doctoral Program in Clinical Psychology, San Diego State University/University of California San Diego, San Diego, CA, USA.
³⁹ Department of Internal Medicine, University of Texas McGovern School of Medicine, Houston, TX, USA.
⁴⁰ Université de Paris, Centre of Research Epidemiology and Statistics (CRESS), Inserm, INRA, Paris, France.
⁴¹ Centre d'Épidémiologie Clinique, Assistance Publique-Hôpitaux de Paris (AP-HP), Hôpital Hôtel Dieu, Paris, France.
⁴² Service de Médecine Interne, Centre de Référence Maladies Autoimmunes Systémiques Rares d'Ile de France, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (AP-HP), Paris, France.
⁴³ APHP-CUP, Hôpital Cochin, Université de Paris, F-75014, Paris, France.
⁴⁴ Department of Epidemiology, Biostatistics, and Occupational Health, McGill University, Montreal, Quebec, Canada.
⁴⁵ Respiratory Epidemiology and Clinical Research Unit, McGill University Health Centre, Montreal, Quebec, Canada.
⁴⁶ Lady Davis Institute of the Jewish General Hospital, 3755 Côte-Sainte-Catherine Road, Montreal, Quebec, H3T 1E2, Canada. brett.thombs@mcgill.ca.
⁴⁷ Department of Psychiatry, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁴⁸ Department of Medicine, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁴⁹ Department of Epidemiology, Biostatistics, and Occupational Health, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁵⁰ Respiratory Epidemiology and Clinical Research Unit, McGill University Health Centre, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁵¹ Department of Psychology, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁵² Department of Educational and Counselling Psychology, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.
⁵³ Biomedical Ethics Unit, McGill University, Montreal, Quebec, Canada. brett.thombs@mcgill.ca.

^# Contributed equally.

PMID: 34838105
PMCID: PMC8626736
DOI: 10.1186/s13063-021-05827-z

Abstract

Background: Systemic sclerosis (scleroderma; SSc) is a rare autoimmune connective tissue disease. We completed an initial feasibility trial of an online self-administered version of the Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program using the cohort multiple randomized controlled trial (RCT) design. Due to low intervention offer uptake, we will conduct a new feasibility trial with progression to full-scale trial, using a two-arm parallel, partially nested RCT design. The SPIN-SELF Program has also been revised to include facilitator-led videoconference group sessions in addition to online material. We will test the group-based intervention delivery format, then evaluate the effect of the SPIN-SELF Program on disease management self-efficacy (primary) and patient activation, social appearance anxiety, and functional health outcomes (secondary).

Methods: This study is a feasibility trial with progression to full-scale RCT, pending meeting pre-defined criteria, of the SPIN-SELF Program. Participants will be recruited from the ongoing SPIN Cohort ( http://www.spinsclero.com/en/cohort ) and via social media and partner patient organizations. Eligible participants must have SSc and low to moderate disease management self-efficacy (Self-Efficacy for Managing Chronic Disease (SEMCD) Scale score ≤ 7.0). Participants will be randomized (1:1 allocation) to the group-based SPIN-SELF Program or usual care for 3 months. The primary outcome in the full-scale trial will be disease management self-efficacy based on SEMCD Scale scores at 3 months post-randomization. Secondary outcomes include SEMCD scores 6 months post-randomization plus patient activation, social appearance anxiety, and functional health outcomes at 3 and 6 months post-randomization. We will include 40 participants to assess feasibility. At the end of the feasibility portion, stoppage criteria will be used to determine if the trial procedures or SPIN-SELF Program need important modifications, thereby requiring a re-set for the full-scale trial. Otherwise, the full-scale RCT will proceed, and outcome data from the feasibility portion will be utilized in the full-scale trial. In the full-scale RCT, 524 participants will be recruited.

Discussion: The SPIN-SELF Program may improve disease management self-efficacy, patient activation, social appearance anxiety, and functional health outcomes in people with SSc. SPIN works with partner patient organizations around the world to disseminate its programs free-of-charge.

Trial registration: ClinicalTrials.gov NCT04246528 . Registered on 27 January 2020.

Keywords: Patient activation; Randomized controlled trial; Scleroderma; Self-efficacy; Self-management; Systemic sclerosis; e-Health.

PubMed Disclaimer

Conflict of interest statement

All authors declare that they have no competing interests.

Figures

**Fig. 1**
SPIN Cohort and non-SPIN Cohort participants: trial flow diagram

**Fig. 2**
Schedule of enrollment, interventions, and assessments

**Fig. 3**
Health care professional discussing the SPIN-SELF Program

**Fig. 4**
Person with SSc discussing their experience with self-management

**Fig. 5**
Nine-item quiz providing guidance to modules most relevant to participants’ symptoms and disease management challenges

**Fig. 6**
Menu of the SPIN-SELF Program modules

**Fig. 7**
Example of the goal-setting feature in the SPIN-SELF Program

**Fig. 8**
Example of the worksheet features in the SPIN-SELF Program

**Fig. 9**
Goals and progress tracking tool

**Fig. 10**
Living with SSc stories page

See this image and copyright information in PMC

References

1. EURORDIS Rare Diseases Europe. [http://www.eurordis.org/content/what-rare-disease] Accessed 10 June 2021.
1. Mayes MD. Systemic sclerosis: clinical features. In: Klippel JH, Stone JH, Crofford LJ, White PH, editors. Primer on the Rheumatic Diseases. 13. New York: Springer and Arthritis Foundation; 2008. pp. 343–350.
1. Allanore Y, Simms R, Distler O, Trojanowska M, Pope J, Denton CP, Varga J. Systemic sclerosis. Nat Rev Dis Primers. 2015;1(1):15002. doi: 10.1038/nrdp.2015.2. - DOI - PubMed
1. Thombs BD, van Lankveld W, Bassel M, Baron M, Buzza R, Haslam S, Haythornthwaite JA, Hudson M, Jewett LR, Knafo R, Kwakkenbos L, Malcarne VL, Milette K, Motivala SJ, Newton EG, Nielson WR, Pacy M, Razykov I, Schieir O, Taillefer S, Worron-Sauve M. Psychological health and well-being in systemic sclerosis: State of the science and consensus research agenda. Arthritis Care Res. 2010;62(8):1181–1189. doi: 10.1002/acr.20187. - DOI - PubMed
1. Bassel M, Hudson M, Taillefer SS, Schieir O, Baron M, Thombs BD. Frequency and impact of symptoms experienced by patients with systemic sclerosis: results from a Canadian National Survey. Rheumatology. 2011;50(4):762–767. doi: 10.1093/rheumatology/keq310. - DOI - PubMed

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The Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program: protocol for a two-arm parallel partially nested randomized controlled feasibility trial with progression to full-scale trial

Collaborators

Affiliations

The Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program: protocol for a two-arm parallel partially nested randomized controlled feasibility trial with progression to full-scale trial

Authors

Collaborators

Affiliations

Abstract

Conflict of interest statement

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Grants and funding

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