Cost-Effectiveness and Cost-Utility of Internet-Delivered Exposure Therapy for Fibromyalgia: Results From a Randomized, Controlled Trial

Published:August 11, 2018DOI:


      Fibromyalgia (FM) is a prevalent and debilitating chronic pain disorder associated with a substantial economic burden. Although there are several studies investigating the effectiveness of psychological treatments such as cognitive-behavioral therapy for FM, studies on cost-effectiveness are scarce. The aim of the present study was to investigate the cost-effectiveness of Internet-delivered exposure therapy (iExp) for FM. We used health economic data from a recently conducted randomized, controlled trial, where 140 participants were randomized to either iExp or a waitlist control (WLC) condition. Economic data were collected at pre-treatment, post-treatment, and at the 1-year follow-up. Treatment effectiveness in relation to costs were analyzed using both a societal perspective (including all direct and indirect costs) and a health care unit perspective (including only the direct treatment costs). Bootstrapped net benefit regression analyses were also conducted, comparing the difference in costs and effects between iExp and WLC, within different willingness-to-pay scenarios. Results showed that the incremental cost-effectiveness ratio was –$15,295, indicating that iExp was highly cost-effective as each successfully treated case (treatment responder) was associated with a substantial net reduction in costs. The robustness of the results was tested in 2 different sensitivity analyses, where iExp remained cost-effective, even in a willingness-to-pay-scenario of $0. We conclude that iExp is a cost-effective treatment that generates large societal cost savings.


      Health-economic evaluations of psychological interventions for FM are scarce. This study is a cost-effectiveness analysis of Internet-delivered exposure therapy for patients with FM. Results showed that iExp was highly cost-effective compared with no treatment, where each successfully treated case generated a substantial societal cost saving.

      Key words

      To read this article in full you will need to make a payment

      Purchase one-time access:

      Academic & Personal: 24 hour online accessCorporate R&D Professionals: 24 hour online access
      One-time access price info
      • For academic or personal research use, select 'Academic and Personal'
      • For corporate R&D use, select 'Corporate R&D Professionals'


      Subscribe to The Journal of Pain
      Already a print subscriber? Claim online access
      Already an online subscriber? Sign in
      Institutional Access: Sign in to ScienceDirect


        • Alaoui El S
        • Lindefors N
        Combining time-driven activity-based costing with clinical outcome in cost-effectiveness analysis to measure value in treatment of depression.
        PLoS One. 2016; 11: 1-15
        • Andersson E
        • Hedman E
        • Ljótsson B
        • Wikström M
        • Elveling E
        • Lindefors N
        • Andersson G
        • Kaldo V
        • Rück C
        Cost-effectiveness of internet-based cognitive behavior therapy for obsessive-compulsive disorder: Results from a randomized controlled trial.
        J Obsessive Compuls Relat Disord. 2015; 4: 47-53
        • Andersson E
        • Ljótsson B
        • Hedman E
        • Mattson S
        • Enander J
        • Andersson G
        • Kaldo V
        • Lindefors N
        • Rück C
        Cost-effectiveness of an internet-based booster program for patients with obsessive-compulsive disorder: Results from a randomized controlled trial.
        J Obsessive Compuls Relat Disord. 2015; 4: 14-19
        • Andersson G
        Using the Internet to provide cognitive behaviour therapy.
        Behav Res Ther. 2009; 47: 175-180
        • Bennett R
        The Fibromyalgia Impact Questionnaire (FIQ): A review of its development, current version, operating characteristics and uses.
        Clin Exp Rheumatol. 2005; 23: S154-S162
        • Bennett RM
        • Bushmakin AG
        • Cappelleri JC
        • Zlateva G
        • Sadosky AB
        Minimal clinically important difference in the fibromyalgia impact questionnaire.
        J Rheumatol. 2009; 36: 1304-1311
        • Berger A
        • Dukes E
        • Martin S
        • Edelsberg J
        • Oster G
        Characteristics and healthcare costs of patients with fibromyalgia syndrome.
        Int J Clin Pract. 2007; 61: 1498-1508
        • Bernardy K
        • Klose P
        • Busch AJ
        • Choy EHS
        • Hauser W
        Cognitive behavioural therapies for fibromyalgia.
        Cochrane Database Syst Rev (9), CD009796, 2013
        • Boonen A
        • van den Heuvel R
        • van Tubergen A
        • Goossens M
        • Severens JL
        • van der Heijde D
        • van der Linden S
        Large differences in cost of illness and wellbeing between patients with fibromyalgia, chronic low back pain, or ankylosing spondylitis.
        Ann Rheum Dis. 2005; 64: 396-402
        • Bouwmans C
        • De Jong K
        • Timman R
        • Ziljstra-Vlasveld M
        • Van der Feltz-Cornelis C
        • Swan Tan S
        • Hakkart-van Roijen L
        Feasibility, reliability and validity of a questionnaire on healthcare consumption and productivity loss in patients with a psychiatric disorder (TiC-P).
        BMC Health Serv Res. 2013; 13: 1-9
        • Briones-Vozmediano E
        • Vives-Cases C
        • Ronda-Pérez E
        • Gil-González D
        Patients’ and professionals’ views on managing fibromyalgia.
        Pain Res Manag. 2013; 18: 1-6
        • Burckhardt C
        • Clark S
        • Bennett R
        The fibromyalgia impact questionnaire: Development and validation.
        J Rheumatol. 1991; 18: 728-733
        • Cohen J
        Statistical power analysis for the behavioral sciences.
        in: Hillsdale NJ, 2nd ed. 1988 (Lawrence Erlbaum Associates)
        • Cuijpers P
        • Smit F
        • Oostenbrink J
        • de Graaf R
        • Have ten M
        • Beekman A
        Economic costs of minor depression: A population-based study.
        Acta Psychiatr Scand. 2007; 115: 229-236
        • Drummond MF
        • Sculpher MJ
        • Torrance GW
        • O'Brien BJ
        Methods for the Economic Evaluation of Health Care Programmes.
        Oxford University Press, Oxford2005
        • Efron B
        • Tibshirani RJ
        An Introduction to the Bootstrap.
        Chapman & Hall, New York, NY1993
        • Epstein SA
        • Kay G
        • Clauw D
        • Heaton R
        • Klein D
        • Krupp L
        • Kuck J
        • Leslie V
        • Masur D
        • Wagner M
        • Waid R
        • Zisook S
        Psychiatric disorders in patients with fibromyalgia.
        Psychosomatics. 1999; 40: 57-63
        • Friesen LN
        • Hadjistavropoulos HD
        • Schneider LH
        • Alberts NM
        • Titov N
        • Dear BF
        Examination of an internet-delivered cognitive behavioural pain management course for adults with fibromyalgia: A randomized controlled trial.
        Pain. 2016; (Dec 15 [Epub ahead of print])
        • Goossens ME
        • Rutten-van Mölken MPMH
        • Leidl RM
        • Bos SGPM
        • Vlaeyen JWS
        • Teeken-Gruben NJG
        Cognitive-educational treatment of fibromyalgia: A randomized clinical trial. II. Economic evaluation.
        J Rheumatol. 1996; 23: 1246-1254
        • Grosse SD
        Assessing cost-effectiveness in healthcare: History of the $50,000 per QALY threshold.
        Expert Rev Pharm Out. 2014; 8: 165-178
        • Haakart-van Roijen L
        Manual Trimbos/iMTA Questionnaire for Costs Associated With Psychiatric Illness.
        Institute for Medical Technology Assessment, Rotterdam, Netherlands2002
        • Hedman E
        • Ljótsson B
        • Lindefors N
        Cognitive behavior therapy via the Internet: A systematic review of applications, clinical efficacy and cost-effectiveness.
        Expert Rev Pharm Out. 2012; 12: 745-764
        • Hedman-Lagerlöf M
        • Hedman-Lagerlöf E
        • Axelsson E
        • Ljótsson B
        • Engelbrektsson J
        • Hultkrantz S
        • Lundbäck K
        • Björkander D
        • Wicksell R
        • Flink I
        • Andersson E
        Internet-based exposure therapy for fibromyalgia: A randomized controlled trial.
        Clin J Pain. 2018; 34: 532-542
        • Hollander den M
        • Goossens M
        • de Jong J
        • Ruijgrok J
        • Oosterhof J
        • Onghena P
        • Smeets R
        • Vlaeyen JWS
        Expose or protect? A randomized controlled trial of exposure in vivo vs pain-contingent treatment as usual in patients with complex regional pain syndrome type 1.
        Pain. 2016; 157: 2318-2329
        • Hudson JI
        • Goldenberg DL
        • Pope Jr, HG
        • Keck Jr, PE
        • Schlesinger L
        Comorbidity of fibromyalgia with medical and psychiatric disorders.
        Am J Med. 1992; 92: 363-367
        • Jacobson NS
        • Truax P
        Clinical significance: A statistical approach to defining meaningful change in psychotherapy research.
        J Consult Clin Psychol. 1991; 59: 12-19
        • Leeuw M
        • Goossens MEJB
        • van Breukelen GJP
        • de Jong JR
        • Heuts PHTG
        • Smeets RJEM
        • Koke AJA
        • Vlaeyen JWS
        Exposure in vivo versus operant graded activity in chronic low back pain patients: Results of a randomized controlled trial.
        Pain. 2008; 138: 192-207
        • Linton SJ
        • Boersma K
        • Jansson M
        • Overmeer T
        • Lindblom K
        • Vlaeyen JWS
        A randomized controlled trial of exposure in vivo for patients with spinal pain reporting fear of work-related activities.
        Eur J Pain. 2008; 12: 722-730
        • Linton SJ
        Correspondence of back pain patients' self-reports of sick leave and Swedish National Insurance authority register.
        Percept Mot Skills. 2011; 112: 133-137
        • Ljótsson B
        • Atterlöf E
        • Hedman-Lagerlöf M
        • Andersson E
        • Jernelöv S
        • Hedman E
        • Kemani M
        • Wicksell RK
        Internet-delivered acceptance and values-based exposure treatment for fibromyalgia: A pilot study.
        Cogn Behav Ther. 2014; 43: 93-104
        • Luciano JV
        • Amico F
        • Lafont M
        • Penarrubia-Maria MT
        • Knapp M
        • Cuesta-Vargas AI
        • Serrano-Blanco A
        • Garcia-Campayo J
        Cost-utility of cognitive behavioral therapy versus U.S. Food and Drug Administration recommended drugs and usual care in the treatment of patients with fibromyalgia: An economic evaluation alongside a 6-month randomized controlled trial.
        Arthritis Res Ther. 2014; 16: 451
        • Luciano JV
        • D'Amico F
        • Feliu-Soler A
        • McCracken LM
        • Aguado J
        • Peñarrubia-María MT
        • Knapp M
        • Serrano-Blanco A
        • Garcia-Campayo J
        Cost-utility of group acceptance and commitment therapy for fibromyalgia versus recommended drugs: An economic analysis alongside a 6-month randomised controlled trial conducted in Spain (EFFIGACT study).
        J Pain. 2017; 18: 868-880
        • Luciano JV
        • Sabes-Figuera R
        • Cardeñosa E
        • Peñarrubia-María MT
        • Fernández-Vergel R
        • Garcia-Campayo J
        • Knapp M
        • Serrano-Blanco A
        • Serran
        Cost-utility of a psychoeducational intervention in fibromyalgia patients compared with usual care.
        Clin J Pain. 2013; 29: 702-711
        • Marciniak M
        • Lage MJ
        • Landbloom RP
        • Dunayevich E
        • Bowman L
        Medical and productivity costs of anxiety disorders: Case control study.
        Depress Anxiety. 2004; 19: 112-120
        • Miró E
        • Lupiáñez J
        • Martínez MP
        • Sánchez AI
        • Díaz-Piedra C
        • Guzmán MA
        • Buela-Casal G
        Cognitive-behavioral therapy for insomnia improves attentional function in fibromyalgia syndrome: A pilot, randomized controlled trial.
        J Health Psychol. 2011; 16: 770-782
        • Mohr DC
        • Spring B
        • Freedland KE
        • Beckner V
        • Arean P
        • Hollon SD
        • Ockene J
        • Kaplan R
        The selection and design of control conditions for randomized controlled trials of psychological interventions.
        Psychother Psychosom. 2009; 78: 275-284
        • Montgomery SA
        • Asberg M
        A new depression scale designed to be sensitive to change.
        Br J Psychiatry. 1979; 134: 382-389
        • Nordgren LB
        • Hedman E
        • Etienne J
        • Bodin J
        • Kadowaki Å
        • Eriksson S
        • Lindkvist E
        • Andersson G
        • Carlbring P
        Effectiveness and cost-effectiveness of individually tailored Internet-delivered cognitive behavior therapy for anxiety disorders in a primary care population: A randomized controlled trial.
        Behav Res Ther. 2014; 59: 1-11
        • Nüesch E
        • Hauser W
        • Bernardy K
        • Barth J
        • Jüni P
        Comparative efficacy of pharmacological and non-pharmacological interventions in fibromyalgia syndrome: Network meta-analysis.
        Ann Rheum Dis. 2013; 72: 955-962
        • Patel A
        • Rendu A
        • Moran P
        • Leese M
        • Mann A
        • Knapp M
        A comparison of two methods of collecting economic data in primary care.
        Fam Pract. 2005; 22: 323-327
        • Proudfoot J
        • Goldberg D
        • Mann A
        • Everitt B
        • Marks I
        • Gray JA
        Computerized, interactive, multimedia cognitive-behavioural program for anxiety and depression in general practice.
        Psychol Med. 2003; 33: 217-227
        • Rabin R
        • Charro F de
        EQ-SD: A measure of health status from the EuroQol Group.
        Ann Med. 2009; 33: 337-343
        • Rivera J
        • Rejas J
        • Esteve-Vives J
        • Vallejo M
        • ICAF
        Resource utilisation and health care costs in patients diagnosed with fibromyalgia in Spain.
        Clin Exp Rheumatol. 2009; 27: S39-S45
        • Robinson RL
        • Jones ML
        In search of pharmacoeconomic evaluations for fibromyalgia treatments: A review.
        Expert Opin Pharmacother. 2006; 7: 1027-1039
      1. Sicras-Mainar A, Rejas J, Navarro R, Blanca M, Morcillo Á, Larios R, Velasco S, Villarroya C: Treating patients with fibromyalgia in primary care settings under routine medical practice: A claim database cost and burden of illness study. Arthritis Res Ther 11: R54, 2009

        • Smit F
        • Willemse G
        • Koopmanschap M
        • Onrust S
        • Cuijpers P
        • Beekman A
        Cost-effectiveness of preventing depression in primary care patients.
        Br J Psychiatry. 2006; 188: 330-336
        • Soares JJF
        • Grossi G
        A randomized, controlled comparison of educational and behavioural interventions for women with fibromyalgia.
        Scand J Occup Ther. 2002; 9: 35-45
        • Soeteman DI
        • Hakkart-van Roijen L
        • Verheul R
        • Busschbach JJV
        The economic burden of personality disorders in mental health care.
        J Clin Psychiatry. 2008; 69: 259-265
      2. Statistics Sweden: Average basic salary, monthly salary and women´s salary as a percentage of men´s salary by sector, occupation (SSYK 2012), sex and educational level (SUN). Year 2014-2015. Available at: Accessed January 30, 2017.

        • Thieme K
        • Gromnica-Ihle E
        • Flor H
        Operant behavioral treatment of fibromyalgia: A controlled study.
        Arthritis Rheum. 2003; 49: 314-320
        • White KP
        • Speechley M
        • Harth M
        • Ostbye T
        Comparing self-reported function and work disability in 100 community cases of fibromyalgia syndrome versus controls in London, Ontario: The London Fibromyalgia Epidemiology Study.
        Arthritis Rheum. 1999; 42: 76-83
        • White LA
        • Birnbaum HG
        • Kaltenboeck A
        • Tang J
        • Mallett D
        • Robinson RL
        Employees with fibromyalgia: Medical comorbidity, healthcare costs, and work loss.
        J Occup Environ Med. 2008; 50: 13-24
        • Wicksell RK
        • Kemani M
        • Jensen K
        • Kosek E
        • Kadetoff D
        • Sorjonen K
        • Ingvar M
        • Olsson GL
        Acceptance and commitment therapy for fibromyalgia: A randomized controlled trial.
        Eur J Pain. 2012; 17: 599-611
        • Wigers SH
        • Stiles TC
        • Vogel PA
        Effects of aerobic exercise versus stress management treatment in fibromyalgia.
        Scand J Rheumatol. 2009; 25: 77-86
        • Wolfe F
        • Ross K
        • Anderson J
        • Russell IJ
        • Hebert L
        The prevalence and characteristics of fibromyalgia in the general population.
        Arthritis Rheum. 1995; 38: 19-28

      Linked Article

      • Erratum
        The Journal of PainVol. 20Issue 12
        • Preview
          In the article “Cost-Effectiveness and Cost-Utility of Internet-Delivered Exposure Therapy for Fibromyalgia: Results From A Randomized, Controlled Trial,” published in the January 2019 issue of The Journal of Pain (2019; 20:47-59), the median value for direct medical costs for iExp at post-treatment was incorrectly displayed in Table 2. The correct table appears below.
        • Full-Text
        • PDF