Effectiveness and cost-effectiveness of a self-management group program to improve social participation in patients with neuromuscular disease and chronic fatigue: protocol of the Energetic study

Yvonne Veenhuizen, Edith H C Cup, Jan T Groothuis, Jan C M Hendriks, Eddy M M Adang, Baziel G M van Engelen, Alexander C H Geurts, Yvonne Veenhuizen, Edith H C Cup, Jan T Groothuis, Jan C M Hendriks, Eddy M M Adang, Baziel G M van Engelen, Alexander C H Geurts

Abstract

Background: Chronic fatigue is present in more than 60% of the patients with a neuromuscular disease and can be their most disabling symptom. In combination with other impairments, fatigue often results in low levels of physical activity and decreased social participation, leading to high societal costs. 'Energetic' is a self-management group program aimed at improving social participation, physical endurance and alleviating fatigue in these patients. The primary aim of this study is to evaluate the effectiveness and cost-effectiveness of the Energetic program.

Methods/design: A multicentered, assessor-blinded, two-armed randomized controlled trial is conducted with evaluations at inclusion and four, seven and fifteen months later. The study includes patients with a neuromuscular disease and chronic fatigue and, when present, their caregivers. The participants are randomized (ratio 1:1) to either an intervention group, receiving the Energetic program, or a control group, receiving usual care (i.e., no specific intervention). The Energetic program covers four months and includes four modules: 1) individually tailored aerobic exercise training; 2) education about aerobic exercise; 3) self-management training in applying energy conservation strategies; and 4) implementation and relapse prevention in daily life. Two months after cessation of the program a booster session is provided. The primary outcome is the perceived performance score of the Canadian Occupational Performance Measure (COPM). Secondary outcomes include the COPM-satisfaction score, and measures of fatigue, physical endurance, activity engagement, mood, and self-efficacy. Caregiver burden is also evaluated as a secondary outcome. Health-related quality of life and medical and societal costs are assessed to estimate cost-effectiveness of the program.

Discussion: The Energetic study is the first randomized controlled trial to evaluate the effectiveness and cost-effectiveness of a combined physical and self-management group training program for improving social participation, physical endurance and alleviating fatigue in patients with neuromuscular diseases. It will generate new insights in (cost-)effective rehabilitation strategies for these incurable conditions.

Trial registration: Clinicaltrials.gov NCT02208687 .

Figures

Figure 1
Figure 1
Flow chart of the Energetic study.

References

    1. Lou JS, Weiss MD, Carter GT. Assessment and management of fatigue in neuromuscular disease. Am J Hosp Palliat Care. 2010;27(2):145–57. doi: 10.1177/1049909109358420.
    1. Kalkman JS, Schillings ML, van der Werf SP, Padberg GW, Zwarts MJ, van Engelen BG, et al. Experienced fatigue in facioscapulohumeral dystrophy, myotonic dystrophy, and HMSN-I. J Neurol Neurosurg Psychiatry. 2005;76(10):1406–9. doi: 10.1136/jnnp.2004.050005.
    1. Needham M, Mastaglia FL. Inclusion body myositis: current pathogenetic concepts and diagnostic and therapeutic approaches. Lancet Neurol. 2007;6(7):620–31. doi: 10.1016/S1474-4422(07)70171-0.
    1. Wilbers J, Frants RR, van Engelen BG, Padberg GW, Van der Maarel SM. [Facioscapulohumeral muscular dystrophy] Ned Tijdschr Tandheelkd. 2010;117(1):11–4.
    1. Gustavsson A, Svensson M, Jacobi F, Allgulander C, Alonso J, Beghi E, et al. Cost of disorders of the brain in Europe 2010. Eur Neuropsychopharmacol. 2011;21(10):718–79. doi: 10.1016/j.euroneuro.2011.08.008.
    1. Voet N, Bleijenberg G, Hendriks J, de Groot I, Padberg G, van Engelen B, et al. Both aerobic exercise and cognitive-behavioral therapy reduce chronic fatigue in FSHD: an RCT. Neurology. 2014;83(21):1914–22. doi: 10.1212/WNL.0000000000001008.
    1. Kalkman JS, Schillings ML, Zwarts MJ, van Engelen BG, Bleijenberg G. The development of a model of fatigue in neuromuscular disorders: a longitudinal study. J Psychosom Res. 2007;62(5):571–9. doi: 10.1016/j.jpsychores.2006.11.014.
    1. Nierse CJ, Abma TA, Horemans AM, van Engelen BG. Research priorities of patients with neuromuscular disease. Disabil Rehabil. 2013;35(5):405–12. doi: 10.3109/09638288.2012.694964.
    1. Voet NB, van der Kooi EL, Riphagen II, Lindeman E, Van Engelen BG, Geurts AC. Strength training and aerobic exercise training for muscle disease. Cochrane Database Syst Rev. 2013;7:CD003907.
    1. Cup EH, Pieterse AJ, Ten Broek-Pastoor JM, Munneke M, van Engelen BG, Hendricks HT, et al. Exercise therapy and other types of physical therapy for patients with neuromuscular diseases: a systematic review. Arch Phys Med Rehabil. 2007;88(11):1452–64. doi: 10.1016/j.apmr.2007.07.024.
    1. Mathiowetz VG, Finlayson ML, Matuska KM, Chen HY, Luo P. Randomized controlled trial of an energy conservation course for persons with multiple sclerosis. Mult Scler. 2005;11(5):592–601. doi: 10.1191/1352458505ms1198oa.
    1. Mathiowetz VG, Matuska KM, Finlayson ML, Luo P, Chen HY. One-year follow-up to a randomized controlled trial of an energy conservation course for persons with multiple sclerosis. Int J Rehabil Res. 2007;30(4):305–13. doi: 10.1097/MRR.0b013e3282f14434.
    1. Lorig KR, Sobel DS, Ritter PL, Laurent D, Hobbs M. Effect of a self-management program on patients with chronic disease. Eff Clin Pract. 2001;4(6):256–62.
    1. Cup E, Nab N, Maas D, De Roode M, De Groot IJM, Smeenge M, et al. ENERGETIC, a multidisciplinary self-management group program for reconditioning and managing fatigue. Neuromuscul Disord. 2012;22(9-10):896–7. doi: 10.1016/j.nmd.2012.06.308.
    1. Copeland L, McNamara R, Kelson M, Simpson S. Mechanisms of change within motivational interviewing in relation to health behaviors outcomes: a systematic review. Patient Educ Couns. 2015;98(4):401–11. doi: 10.1016/j.pec.2014.11.022.
    1. Rademakers J, Nijman J, van der Hoek L, Heijmans M, Rijken M. Measuring patient activation in The Netherlands: translation and validation of the American short form Patient Activation Measure (PAM13) BMC Public Health. 2012;12:577. doi: 10.1186/1471-2458-12-577.
    1. Cup EH, Pieterse AJ, Knuijt S, Hendricks HT, van Engelen BG, Oostendorp RA, et al. Referral of patients with neuromuscular disease to occupational therapy, physical therapy and speech therapy: usual practice versus multidisciplinary advice. Disabil Rehabil. 2007;29(9):717–26. doi: 10.1080/09638280600926702.
    1. McColl MA, Law M, Baptiste S, Pollock N, Carswell A, Polatajko HJ. Targeted applications of the Canadian Occupational Performance Measure. Can J Occup Ther. 2005;72(5):298–300. doi: 10.1177/000841740507200506.
    1. Dedding C, Cardol M, Eyssen IC, Dekker J, Beelen A. Validity of the Canadian Occupational Performance Measure: a client-centred outcome measurement. Clin Rehabil. 2004;18(6):660–7. doi: 10.1191/0269215504cr746oa.
    1. Eyssen IC, Beelen A, Dedding C, Cardol M, Dekker J. The reproducibility of the Canadian Occupational Performance Measure. Clin Rehabil. 2005;19(8):888–94. doi: 10.1191/0269215505cr883oa.
    1. Vercoulen JH, Swanink CM, Fennis JF, Galama JM, van der Meer JW, Bleijenberg G. Dimensional assessment of chronic fatigue syndrome. J Psychosom Res. 1994;38(5):383–92. doi: 10.1016/0022-3999(94)90099-X.
    1. Enright PL. The six-minute walk test. Respir Care. 2003;48(8):783–5.
    1. Jong AM, van Nes FA, Lindeboom R. The Dutch Activity Card Sort institutional version was reproducible, but biased against women. Disabil Rehabil. 2012;34(18):1550–5. doi: 10.3109/09638288.2011.647232.
    1. Foster ER, Golden L, Duncan RP, Earhart GM. Community-based Argentine tango dance program is associated with increased activity participation among individuals with Parkinson’s disease. Arch Phys Med Rehabil. 2013;94(2):240–9. doi: 10.1016/j.apmr.2012.07.028.
    1. Spinhoven P, Ormel J, Sloekers PP, Kempen GI, Speckens AE, Van Hemert AM. A validation study of the Hospital Anxiety and Depression Scale (HADS) in different groups of Dutch subjects. Psychol Med. 1997;27(2):363–70. doi: 10.1017/S0033291796004382.
    1. Romppel M, Herrmann-Lingen C, Wachter R, Edelmann F, Dungen HD, Pieske B, et al. A short form of the General Self-Efficacy Scale (GSE-6): Development, psychometric properties and validity in an intercultural non-clinical sample and a sample of patients at risk for heart failure. Psychosoc Med. 2013;10:Doc01.
    1. Van Durme T, Macq J, Jeanmart C, Gobert M. Tools for measuring the impact of informal caregiving of the elderly: a literature review. Int J Nurs Stud. 2012;49(4):490–504. doi: 10.1016/j.ijnurstu.2011.10.011.
    1. Aaronson NK, Muller M, Cohen PD, Essink-Bot ML, Fekkes M, Sanderman R, et al. Translation, validation, and norming of the Dutch language version of the SF-36 Health Survey in community and chronic disease populations. J Clin Epidemiol. 1998;51(11):1055–68. doi: 10.1016/S0895-4356(98)00097-3.
    1. Health Economics and Decision Science, SF-6D []
    1. Sturkenboom IH, Graff MJ, Hendriks JC, Veenhuizen Y, Munneke M, Bloem BR, et al. Efficacy of occupational therapy for patients with Parkinson’s disease: a randomised controlled trial. Lancet Neurol. 2014;13(6):557–66. doi: 10.1016/S1474-4422(14)70055-9.
    1. Carswell A, McColl MA, Baptiste S, Law M, Polatajko H, Pollock N. The Canadian Occupational Performance Measure: a research and clinical literature review. Can J Occup Ther. 2004;71(4):210–22. doi: 10.1177/000841740407100406.
    1. Geerdink Y, Aarts P, Geurts AC. Motor learning curve and long-term effectiveness of modified constraint-induced movement therapy in children with unilateral cerebral palsy: a randomized controlled trial. Res Dev Disabil. 2013;34(3):923–31. doi: 10.1016/j.ridd.2012.11.011.
    1. Cup EH, Scholte OP, Reimer WJ, Thijssen MC, Van Kuyk-Minis MA. Reliability and validity of the Canadian Occupational Performance Measure in stroke patients. Clin Rehabil. 2003;17(4):402–9. doi: 10.1191/0269215503cr635oa.

Source: PubMed

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