Cost-effectiveness of interventions for medically unexplained symptoms: A systematic review

Wortman MSH1,2, Lokkerbol J3,4, van der Wouden JC2, Visser B1, van der Horst HE2, Olde Hartman TC5. PLoS One. 2018 Oct 15;13(10):e0205278. doi: 10.1371/journal.pone.0205278. eCollection 2018.

Author information

1 ACHIEVE - Centre of Applied Research, Faculty of Health, Amsterdam University of Applied Sciences, Amsterdam, The Netherlands.

2 Department of General Practice and Elderly Care Medicine, Amsterdam UMC, Vrije Universiteit, Amsterdam, The Netherlands.

3 Centre of Economic Evaluation, Trimbos Institute (Netherlands Institute of Mental Health and Addiction), Utrecht, The Netherlands.

4 Department of Health Care Policy, Harvard Medical School, Boston, Massachusetts, United States of America.

5 Department of Primary and Community Care, Radboud University Medical Center, Nijmegen, The Netherlands.


BACKGROUND: In primary and secondary care medically unexplained symptoms (MUS) or functional somatic syndromes (FSS) constitute a major burden for patients and society with high healthcare costs and societal costs. Objectives were to provide an overview of the evidence regarding the cost-effectiveness of interventions for MUS or FSS, and to assess the quality of these studies.

METHODS: We searched the databases PubMed, PsycINFO, the National Health Service Economic Evaluation Database (NHS-EED) and the CEA registry to conduct a systematic review. Articles with full economic evaluations on interventions focusing on adult patients with undifferentiated MUS or fibromyalgia (FM), irritable bowel syndrome (IBS) and chronic fatigue syndrome (CFS), with no restrictions on comparators, published until 15 June 2018, were included. We excluded preventive interventions. Two reviewers independently extracted study characteristics and cost-effectiveness data and used the Consensus on Health Economic Criteria Checklist to appraise the methodological quality.

RESULTS: A total of 39 studies out of 1,613 articles met the inclusion criteria. Twenty-two studies reported costs per quality-adjusted life year (QALY) gained and cost-utility analyses (CUAs). In 13 CUAs the intervention conditions dominated the control conditions or had an incremental cost-effectiveness ratio below the willingness-to-pay threshold of € 50,000 per QALY, meaning that the interventions were (on average) cost-effective in comparison with the control condition. Group interventions focusing on MUS (n = 3) or FM (n = 4) might be more cost-effective than individual interventions. The included studies were heterogeneous with regard to the included patients, interventions, study design, and outcomes.

CONCLUSION: This review provides an overview of 39 included studies of interventions for patients with MUS and FSS and the methodological quality of these studies. Considering the limited comparability due to the heterogeneity of the studies, group interventions might be more cost-effective than individual interventions.

REGISTRATION: Study methods were documented in an international prospective register of systematic reviews (PROSPERO) protocol, registration number: CRD42017060424.

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