Scleroderma Patient-centered Intervention Network-Scleroderma Support group Leader EDucation (SPIN-SSLED) program: non-randomised feasibility trial

Brett D Thombs, Laura Dyas, Mia Pépin, Kylene Aguila, Marie-Eve Carrier, Lydia Tao, Sami Harb, Vanessa L Malcarne, Ghassan El-Baalbaki, Sandra Peláez, Maureen Sauve, Marie Hudson, Robert W Platt, SPIN-SSLED Patient Advisory Team, Kerri Connolly, Stephen Elrod, Catherine Fortuné, Karen Gottesman, Karen Nielsen, Ken Rozee, Geneviève Guillot, Amy Gietzen, Michelle Richard, Nancy Stephens, Brett D Thombs, Laura Dyas, Mia Pépin, Kylene Aguila, Marie-Eve Carrier, Lydia Tao, Sami Harb, Vanessa L Malcarne, Ghassan El-Baalbaki, Sandra Peláez, Maureen Sauve, Marie Hudson, Robert W Platt, SPIN-SSLED Patient Advisory Team, Kerri Connolly, Stephen Elrod, Catherine Fortuné, Karen Gottesman, Karen Nielsen, Ken Rozee, Geneviève Guillot, Amy Gietzen, Michelle Richard, Nancy Stephens

Abstract

Objectives: The Scleroderma Patient-centered Intervention Network-Scleroderma Support group Leader EDucation (SPIN-SSLED) Programme was designed to improve confidence and self-efficacy and to reduce burden for support group leaders. Objectives were to (1) evaluate feasibility of programme delivery, including required resources, management issues and scientific aspects (eg, performance of outcome measures) and (2) assess user satisfaction and identify any modifications needed to improve programme content or delivery based on participant feedback.

Design: Non-randomised feasibility trial.

Setting: North American patient organisations.

Participants: Current support group leaders or potential new leaders referred by patient organisations.

Intervention: The programme included 13 modules delivered live via videoconference over 3 months (April to July 2018) in 60 to 90 min sessions.

Outcome measures: (1) Elements of feasibility, including enrolment and consent procedures, percentage of referred group leaders who consented to participate, session attendance and technical support requirements; (2) programme usability, understandability, organisation and clarity; (3) leader satisfaction with the programme and (4) planned trial outcome measures, including support group leader self-efficacy, burnout, emotional distress and physical function.

Results: All 12 referred potential participants consented to enrol, and 10 were included in two training groups of five participants each. Participants attended 95% of sessions. Required technical support was minimal, and videoconferencing technology functioned well. Overall programme satisfaction rating was 9.4/10. Mean item rating on the eight items of the Client Satisfaction Questionnaire-8 was 3.83 (1=low satisfaction; 4=high satisfaction). Pre-post scores on the Scleroderma Support Group Leader Self-efficacy Scale increased by 1.7 SDs (large effect); scores on burnout, emotional distress and physical function improved by 0.44, 0.38 and 0.45 SDs (moderate effects).

Conclusion: The SPIN-SSLED Programme was feasibly delivered, including management, resource and scientific aspects. Participant satisfaction was high. The programme is ready to be tested in a full-scale randomised controlled trial.

Trial registration number: NCT03508661.

Keywords: feasibility trial; patient education; peer support; scleroderma; support groups; systemic sclerosis.

Conflict of interest statement

Competing interests: None declared.

© Author(s) (or their employer(s)) 2019. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.

References

    1. Kralik D. The quest for ordinariness: transition experienced by midlife women living with chronic illness. J Adv Nurs 2002;39:146–54. 10.1046/j.1365-2648.2000.02254.x
    1. Karasz A, Ouellette SC. Role strain and psychological well-being in women with systemic lupus erythematosus. Women Health 1995;23:41–57. 10.1300/J013v23n03_03
    1. Kole A, Faurisson F. The voice of 12,000 patients: experiences and expectations of rare disease patients on diagnosis and care in Europe, 2009. Available: [Accessed 10 Feb 2019].
    1. European Organisation for Rare Diseases Rare diseases: understanding this public health priority, 2005. Available: [Accessed 10 Feb 2019].
    1. Cohen JS, Biesecker BB. Quality of life in rare genetic conditions: a systematic review of the literature. Am J Med Genet A 2010;152A:1136–56. 10.1002/ajmg.a.33380
    1. Huyard C. What, if anything, is specific about having a rare disorder? patients' judgements on being ill and being rare. Health Expect 2009;12:361–70. 10.1111/j.1369-7625.2009.00552.x
    1. Nettleton S, Watt I, O'Malley L, et al. . Understanding the narratives of people who live with medically unexplained illness. Patient Educ Couns 2005;56:205–10. 10.1016/j.pec.2004.02.010
    1. van Walsem MR, Howe EI, Iversen K, et al. . Unmet needs for healthcare and social support services in patients with Huntington's disease: a cross-sectional population-based study. Orphanet J Rare Dis 2015;10:124 10.1186/s13023-015-0324-8
    1. Anderson M, Elliott EJ, Zurynski YA. Australian families living with rare disease: experiences of diagnosis, health services use and needs for psychosocial support. Orphanet J Rare Dis 2013;8:22 10.1186/1750-1172-8-22
    1. Dwyer AA, Quinton R, Morin D, et al. . Identifying the unmet health needs of patients with congenital hypogonadotropic hypogonadism using a web-based needs assessment: implications for online interventions and peer-to-peer support. Orphanet J Rare Dis 2014;9:83 10.1186/1750-1172-9-83
    1. Kasparian NA, Rutstein A, Sansom-Daly UM, et al. . Through the looking glass: an exploratory study of the lived experiences and unmet needs of families affected by von Hippel-Lindau disease. Eur J Hum Genet 2015;23:34–40. 10.1038/ejhg.2014.44
    1. Holtzclaw Williams P. Policy framework for rare disease health disparities. Policy Polit Nurs Pract 2011;12:114–8. 10.1177/1527154411404243
    1. Newman S, Steed L, Mulligan K. Self-Management interventions for chronic illness. The Lancet 2004;364:1523–37. 10.1016/S0140-6736(04)17277-2
    1. Davison KP, Pennebaker JW, Dickerson SS. Who talks? the social psychology of illness support groups. Am Psychol 2000;55:205–17. 10.1037/0003-066X.55.2.205
    1. Kwakkenbos L, Jewett LR, Baron M, et al. . 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. BMJ Open 2013;3:eoo3563 10.1136/bmjopen-2013-003563
    1. Reimann A, Bend J, Dembski B. [Patient-centred care in rare diseases. A patient organisations' perspective]. Bundesgesundheitsblatt Gesundheitsforschung Gesundheitsschutz 2007;50:1484–93. 10.1007/s00103-007-0382-8
    1. Gumuchian ST, Delisle VC, Kwakkenbos L, et al. . Reasons for attending support groups and organizational preferences: the European scleroderma support group members survey. Disabil Rehabil 2019;41:974–82. 10.1080/09638288.2017.1416497
    1. Delisle VC, Gumuchian ST, Peláez S, et al. . Reasons for non-participation in scleroderma support groups. Clin Exp Rheumatol 2016;100:56–62.
    1. Gumuchian ST, Delisle VC, Peláez S, et al. . Reasons for not participating in scleroderma patient support groups: a cross‐sectional study. Arthritis Care Res 2018;70:275–83. 10.1002/acr.23220
    1. Delisle VC, Gumuchian ST, El-Baalbaki G, et al. . Training and support needs of scleroderma support group facilitators: the North American scleroderma support group facilitators survey. Disabil Rehabil 2018;25:1–6.
    1. Aymé S, Kole A, Groft S. Empowerment of patients: lessons from the rare diseases community. The Lancet 2008;371:2048–51. 10.1016/S0140-6736(08)60875-2
    1. Barg FK, Gullatte MM. Cancer support groups: meeting the needs of African Americans with cancer. Semin Oncol Nurs 2001;17:171–8. 10.1053/sonu.2001.25946
    1. Seibold J. Scleroderma : Harris ED, Budd RC, Firestein GS, Kelly’s Textbook of Rheumatology. Philadelphia: Elsevier, 2005: 1279–308.
    1. Mayes M. Systemic sclerosis: clinical features : Klippel JH, Stone JH, Crafford LJ, Primer on the rheumatic diseases. 13th edn New York: Springer and Arthritis Foundation, 2008: 343–50.
    1. Wigley FM, Hummers LK. Clinical features of systemic sclerosis : Hochberg MC, Silman AJ, Smolen JS, Rheumatology. 3rd edn Philadelphia: Mosby, 2003: 1463–80.
    1. Kwakkenbos L, Delisle VC, Fox RS, et al. . Psychosocial aspects of scleroderma. Rheum Dis Clin North Am 2015;41:519–28. 10.1016/j.rdc.2015.04.010
    1. Scleroderma Canada Find a support group. Available: [Accessed 10 Feb 2019].
    1. Scleroderma Foundation Support groups. Available: [Accessed 10 Feb 2019].
    1. Scleroderma & Raynaud’s UK Find support. Available: [Accessed 10 Feb 2019].
    1. Scleroderma Association of New South Wales Regional support groups. Available: [Accessed 10 Feb 2019].
    1. Delisle VC, Gumuchian ST, Kloda LA, et al. . Effect of support group peer facilitator training programmes on peer facilitator and support group member outcomes: a systematic review. BMJ Open 2016;6:e013325 10.1136/bmjopen-2016-013325
    1. Zordan R, Butow P, Kirsten L, et al. . Supporting the supporters: a randomized controlled trial of interventions to assist the leaders of cancer support groups. J Community Psychol 2015;43:261–77. 10.1002/jcop.21677
    1. Eldridge SM, Chan CL, Campbell MJ, et al. . Consort 2010 statement: extension to randomised pilot and feasibility trials. BMJ 2016;355:i5239 10.1136/bmj.i5239
    1. Boud D, Feletti G. The challenge of problem-based learning. 2nd edn London: Kogan Page, 1997.
    1. Duch BJ, Groh SE, Allen DE. Why problem-based learning? A case study of institutional change in undergraduate education : Duch BJ, Groh SE, Allen DE, The power or problem-based learning. Sterling, VA: Stylus, 2001: 3–11.
    1. Hmelo-Silver CE. Problem-Based learning: what and how do students learn? Educ Psychol Rev 2004;16:235–66. 10.1023/B:EDPR.0000034022.16470.f3
    1. Torp L, Sage S. Problems as possibilities: problem-based learning for K-16 education. 2nd edn Alexandria, VA: Association for Supervision and Curriculum Development, 2002.
    1. Hay-Hansson AW, Eldevik S. Training discrete trials teaching skills using videoconference. Res Autism Spectr Disord 2013;7:1300–9. 10.1016/j.rasd.2013.07.022
    1. Seibert DC, Guthrie JT, Adamo G. Improving learning outcomes: integration of standardized patients & telemedicine technology. Nurs Educ Perspect 2004;25:232–7.
    1. Xavier K, Shepherd L, Goldstein D. Clinical supervision and education via videoconference: a feasibility project. J Telemed Telecare 2007;13:206–9. 10.1258/135763307780907996
    1. Marziali E, Donahue P. Caring for others: Internet video-conferencing group intervention for family caregivers of older adults with neurodegenerative disease. Gerontologist 2006;46:398–403. 10.1093/geront/46.3.398
    1. Fisher WW, Luczynski KC, Hood SA, et al. . Preliminary findings of a randomized clinical trial of a virtual training program for applied behavior analysis technicians. Res Autism Spectr Disord 2014;8:1044–54. 10.1016/j.rasd.2014.05.002
    1. Hommel KA, Gray WN, Hente E, et al. . The telehealth enhancement of adherence to medication (team) in pediatric IBD trial: design and methodology. Contemp Clin Trials 2015;43:105–13. 10.1016/j.cct.2015.05.013
    1. Williams LK, McCarthy MC, Burke K, et al. . Addressing behavioral impacts of childhood leukemia: a feasibility pilot randomized controlled trial of a group videoconferencing parenting intervention. Eur J Oncol Nurs 2016;24:61–9. 10.1016/j.ejon.2016.08.008
    1. Shoemaker SJ, Wolf MS, Brach C. Development of the patient education materials assessment tool (PEMAT): a new measure of understandability and actionability for print and audiovisual patient information. Patient Educ Couns 2014;96:395–403. 10.1016/j.pec.2014.05.027
    1. Pal NE, Gumuchian ST, Delisle VC, et al. . Development and preliminary validation of the scleroderma support group leader self-efficacy scale. J Scleroderma Relat Disord 2018;3:106–11. 10.5301/jsrd.5000260
    1. Page BJ, Pietrzak DR, Lewis TF. Development of the group leader self-efficacy instrument. JSGW 2001;26:168–84. 10.1080/01933920108415736
    1. Zordan RD, Juraskova I, Butow PN, et al. . Exploring the impact of training on the experience of Australian support group leaders: current practices and implications for research. Health Expect 2010;13:427–40. 10.1111/j.1369-7625.2010.00592.x
    1. Butow PN, Ussher J, Kirsten L, et al. . Sustaining leaders of cancer support groups: the role, needs, and difficulties of leaders. Soc Work Health Care 2005;42:39–55.
    1. Demerouti E, Bakker AB, Vardakou I, et al. . The convergent validity of two burnout instruments: a multitrait-multimethod analysis. Eur J Psychol Assess 2002;18:296–307.
    1. Halbesleben JRB, Demerouti E. The construct validity of an alternative measure of burnout: investigating the English translation of the Oldenburg burnout inventory. Work Stress 2005;19:208–20. 10.1080/02678370500340728
    1. Reis D, Xanthopoulou D, Tsaousis I. Measuring job and academic burnout with the Oldenburg burnout inventory (OLBI): factorial invariance across samples and countries. Burn Res 2015;2:8–18. 10.1016/j.burn.2014.11.001
    1. Kroenke K, Strine TW, Spitzer RL, et al. . The PHQ-8 as a measure of current depression in the general population. J Affect Disord 2009;114:163–73. 10.1016/j.jad.2008.06.026
    1. Milette K, Hudson M, Baron M, et al. . Comparison of the PHQ-9 and CES-D depression scales in systemic sclerosis: internal consistency reliability, convergent validity and clinical correlates. Rheumatology 2010;49:789–96. 10.1093/rheumatology/kep443
    1. Kwakkenbos L, Thombs BD, Khanna D, et al. . Performance of the patient-reported outcomes measurement information System-29 in scleroderma: a scleroderma patient-centered intervention network cohort study. Rheumatology 2017;56:1302–11. 10.1093/rheumatology/kex055
    1. Larsen DL, Attkisson CC, Hargreaves WA, et al. . Assessment of client/patient satisfaction: development of a general scale. Eval Program Plann 1979;2:197–207. 10.1016/0149-7189(79)90094-6
    1. Kelly PJ, Kyngdon F, Ingram I, et al. . The client satisfaction Questionnaire-8: psychometric properties in a cross-sectional survey of people attending residential substance abuse treatment. Drug Alcohol Rev 2018;37:79–86. 10.1111/dar.12522
    1. Cohen J. Statistical Power Analysis for the Behavioral Sciences. 2nd edn Hillsdale, NJ: Lawrence Erlbaum, 1988.
    1. Zwarenstein M, Treweek S, Gagnier JJ, et al. . Improving the reporting of pragmatic trials: an extension of the CONSORT statement. BMJ 2008;337:a2390 10.1136/bmj.a2390
    1. Roland M, Torgerson DJ. Understanding controlled trials: what are pragmatic trials? BMJ 1998;316:285 10.1136/bmj.316.7127.285
    1. Lohr S, Schochet PZ, Sanders E. Partially nested randomised controlled trials in educational research: a guide to design and analysis. NCER 2014-2000. Washington, DC: National Center for Education Research, 2014.
    1. Bauer DJ, Sterba SK, Hallfors DD. Evaluating group-based interventions when control participants are ungrouped. Multivariate Behav Res 2008;43:210–36. 10.1080/00273170802034810
    1. Schweig JD, Pane JF. Intention-To-Treat analysis in partially nested randomized controlled trials with real-world complexity. IJRES 2016;39:268–86. 10.1080/1743727X.2016.1170800

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