Economic Evaluations of Educational, Physical, and Psychological Treatments for Fibromyalgia

A Systematic Review With Meta-Analysis

Cristina Maria Nunes Cabral; Gisela Cristiane Miyamoto; Katherinne Ferro Moura Franco; Judith Ekkina Bosmans


Pain. 2021;162(9):2331-2345. 

In This Article


The aim of this study was to perform a systematic review of economic evaluations of educational, physical, and psychological interventions in the treatment of patients with fibromyalgia. Psychological interventions[14,25,26,28,35] can be considered cost-effective compared with different comparator treatments. Meta-analyses showed that healthcare costs in the psychological intervention group were statistically significantly lower than in usual care and pharmacological intervention group after 6 months, and than usual care after 12 months. Finally, for QALYs, aquatic exercise was cost-effective compared with usual care, and for impact of fibromyalgia, whether aerobic exercise is considered cost-effective compared with usual care depends on the willingness to pay of decision makers.

Some strengths of this systematic review can be highlighted. We used a search strategy including broad terms for fibromyalgia and economic evaluations, without searching for specific treatments.[7] All types of economic evaluations were included in this review, although we only identified cost-effectiveness and cost-utility analyses conducted alongside randomized controlled trials. Finally, by calculating ICERs for impact of fibromyalgia in 3 studies[8,14,28] and in a study that only reported costs and effects,[49] results of this review are of interest and use for patients, clinicians, and decision makers alike. On the other hand, the adoption of different interventions and time horizons in the economic evaluations limited our possibility to pool data, which was performed only for 2 studies per meta-analysis. This is not a specific limitation of our systematic review but of the research in the field of economic evaluations on interventions for fibromyalgia. Data of costs and outcomes stratified by the group were missing in some studies, which also limited our meta-analyses. Efforts were made to obtain complementary data from 2 studies,[10,25] but no response was acquired from one study,[10] and the sample size was different for QALYs and costs from another study.[25]

Despite the economic burden of fibromyalgia,[23] the evidence base on cost-effective treatments is limited. A recent systematic review[53] on the cost-effectiveness of interventions for functional somatic syndromes including fibromyalgia showed similar results to ours. However, our search has been conducted more recently resulting in the addition of 3 recently published studies.[8,14,35] Moreover, we presented novel findings on costs and effects in 4 studies[8,14,28,49] that we used to calculate ICERs for impact of fibromyalgia, which are important to allow the comparison of ICERs for the same health condition[30] and more meaningful for decision makers.[47] Impact of fibromyalgia is an umbrella term that encompasses outcomes, such as health-related quality of life, pain, fatigue, stiffness, and physical function, and is commonly assessed in randomized controlled trials with intervention for fibromyalgia.[6,12,19] Therefore, we considered it important to include this outcome in our systematic review, whereas the previous review[53] did not. Finally, we could perform 3 meta-analyses, to improve precision with bigger sample sizes and to answer different questions,[16] especially related to which intervention presented lower healthcare and societal costs.

Two other systematic reviews also assessed the cost-effectiveness of interventions for patients with chronic nonmalignant pain[43] and medically unexplained symptoms,[21] which are health conditions that typically include fibromyalgia. Because of different inclusion criteria, these systematic reviews included only one study[10] also included in our systematic review and presented general results for chronic nonmalignant pain[43] and medically unexplained symptoms.[21] These differences prevented a detailed comparison between our results and the results of these systematic reviews.

The reporting quality of the economic evaluations according to the CHEERS statement was sufficient. Information, such as target population and comparators, was rarely complete in the economic evaluation study, because reporting all important details requires substantial space. Word limits prevent doing this and are a known challenge for both authors and editors.[18] To overcome this issue, we did not only extract data from the included studies, but also from supporting studies, ie, design and effectiveness studies.[1,13,24,27,32,36,48,50,54] On the other hand, the risk of bias of the randomized controlled trial for all included studies was high. The domain judged as having high risk of bias in all studies was bias in measurement of the outcomes, because blinding of outcome assessors is difficult for patient-reported outcomes.[16] In addition, the high overall risk of bias found in the included studies of this systematic review may lead to an overestimation or underestimation of the true effectiveness of the interventions,[40] which can compromise the validity of the results presented here.

The results of this systematic review show that psychological interventions are dominant over other active interventions and usual care or control groups. Cost-effective psychological interventions mainly consist of psychoeducative program, cognitive behavioural therapy, acceptance and commitment therapy, exposure therapy, and attachment-based compassion therapy. Future studies should indicate which specific elements of the interventions are effective in fibromyalgia. However, economic evaluations of other interventions recommended for the treatment of fibromyalgia, such as acupuncture, exercises, meditative, and multicomponent therapies,[5,12,29,42] are missing and need to be performed. We found only 2 economic evaluations on the cost-effectiveness of physical exercise in the treatment of patients with fibromyalgia,[11,49] and, thus, definitive conclusions cannot be drawn about this type of treatment. The risk of bias of the randomized controlled trials should be reduced, especially concerning bias due to missing outcome data and in measurement of the outcome. Finally, the time horizon was less than 12 months in 5 studies.[8,11,14,26,28] Considering the chronic nature of fibromyalgia symptoms, future research in this field should consider longer time horizons with a minimum of 12 months, which is comparable with studies on chronic spinal disorders,[22] a chronic health condition similar to fibromyalgia. Results from high-quality economic evaluations can be used to develop health economic decision analytical models. These models synthesize information from multiple sources, including economic evaluations alongside randomized controlled trials, and apply mathematical techniques to estimate cost-effectiveness.[37] Using such models allows for indirect comparison of different interventions groups and longer time horizons than feasible in randomized controlled trials. At this time, only economic evaluations alongside randomized controlled trials with educational, physical, and psychological interventions were found.