Systematic Review of the Epidemiological Burden of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome Across Europe: Current Evidence and EUROMENE Research Recommendations for Epidemiology

Fernando Estévez-López¹, Kathleen Mudie², Xia Wang-Steverding³, Inger Johanne Bakken⁴, Andrejs Ivanovs⁵, Jesús Castro-Marrero⁶, Luis Nacul², Jose Alegre⁶, Paweł Zalewski⁷, Joanna Słomko⁷, Elin Bolle Strand^{8

9}, Derek Pheby¹⁰, Evelina Shikova¹¹, Lorenzo Lorusso¹², Enrica Capelli¹³, Slobodan Sekulic¹⁴, Carmen Scheibenbogen¹⁵, Nuno Sepúlveda^{2

16}, Modra Murovska¹⁷, Eliana Lacerda²

Affiliations

¹ Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC University Medical Center, P.O. Box 2040, 3000 CA Rotterdam, The Netherlands.
² Department of Clinical Research, Faculty of Infectious & Tropical Disease, London School of Hygiene & Tropical Medicine, London WC1E 7HT, UK.
³ Warwick Medical School, University of Warwick, Coventry CV4 7HL, UK.
⁴ Centre for Fertility and Health (CeFH), Norwegian Institute of Public Health, 0456 Oslo, Norway.
⁵ Statistics Unit, Riga Stradins University, LV-1007 Riga, Latvia.
⁶ ME/CFS Unit, Division of Rheumatology, Vall d'Hebron Hospital Research Institute (VHIR), Universitat Autònoma de Barcelona, 08035 Barcelona, Spain.
⁷ Department of Hygiene, Epidemiology, Ergonomics and Postgraduate Education, Collegium Medicum in Bydgoszcz, Nicolaus Copernicus University in Toruń, 85-094 Bydgoszcz, Poland.
⁸ Faculty of Health Studies, VID Specialized University, 0370 Oslo, Norway.
⁹ Norway & National Advisory Unit on CFS/ME, Oslo University Hospital, 0424 Oslo, Norway.
¹⁰ Faculty of Health and Society, Buckinghamshire New University, High Wycombe HP11 3JZ, UK.
¹¹ Department of Virology, National Center of Infectious and Parasitic Diseases, Sofia, Bulgaria & The National Specialized Hospital for Active Treatment in Haematological Diseases, 1233 Sofia, Bulgaria.
¹² Neurology Department, ASST-Lecco, 23807 Merate, Italy.
¹³ Department of Earth and Environmental Sciences and Centre for Health Technologies, University of Pavia, 27100 Pavia, Italy.
¹⁴ Department of Neurology, Medical Faculty Novi Sad, University of Novi Sad, 21000 Novi Sad, Serbia.
¹⁵ Institute for Medical Immunology, Charité-Universitätsmedizin Berlin, 13353 Berlin, Germany.
¹⁶ Centre of Statistics and Its Applications, University of Lisbon, 1749-016 Lisbon, Portugal.
¹⁷ Institute of Microbiology and Virology, Riga Stradins University, LV-1007 Riga, Latvia.

PMID: 32455633
PMCID: PMC7290765
DOI: 10.3390/jcm9051557

Review

Systematic Review of the Epidemiological Burden of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome Across Europe: Current Evidence and EUROMENE Research Recommendations for Epidemiology

Fernando Estévez-López et al. J Clin Med. 2020.

. 2020 May 21;9(5):1557.

doi: 10.3390/jcm9051557.

Authors

Affiliations

¹ Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC University Medical Center, P.O. Box 2040, 3000 CA Rotterdam, The Netherlands.
² Department of Clinical Research, Faculty of Infectious & Tropical Disease, London School of Hygiene & Tropical Medicine, London WC1E 7HT, UK.
³ Warwick Medical School, University of Warwick, Coventry CV4 7HL, UK.
⁴ Centre for Fertility and Health (CeFH), Norwegian Institute of Public Health, 0456 Oslo, Norway.
⁵ Statistics Unit, Riga Stradins University, LV-1007 Riga, Latvia.
⁶ ME/CFS Unit, Division of Rheumatology, Vall d'Hebron Hospital Research Institute (VHIR), Universitat Autònoma de Barcelona, 08035 Barcelona, Spain.
⁷ Department of Hygiene, Epidemiology, Ergonomics and Postgraduate Education, Collegium Medicum in Bydgoszcz, Nicolaus Copernicus University in Toruń, 85-094 Bydgoszcz, Poland.
⁸ Faculty of Health Studies, VID Specialized University, 0370 Oslo, Norway.
⁹ Norway & National Advisory Unit on CFS/ME, Oslo University Hospital, 0424 Oslo, Norway.
¹⁰ Faculty of Health and Society, Buckinghamshire New University, High Wycombe HP11 3JZ, UK.
¹¹ Department of Virology, National Center of Infectious and Parasitic Diseases, Sofia, Bulgaria & The National Specialized Hospital for Active Treatment in Haematological Diseases, 1233 Sofia, Bulgaria.
¹² Neurology Department, ASST-Lecco, 23807 Merate, Italy.
¹³ Department of Earth and Environmental Sciences and Centre for Health Technologies, University of Pavia, 27100 Pavia, Italy.
¹⁴ Department of Neurology, Medical Faculty Novi Sad, University of Novi Sad, 21000 Novi Sad, Serbia.
¹⁵ Institute for Medical Immunology, Charité-Universitätsmedizin Berlin, 13353 Berlin, Germany.
¹⁶ Centre of Statistics and Its Applications, University of Lisbon, 1749-016 Lisbon, Portugal.
¹⁷ Institute of Microbiology and Virology, Riga Stradins University, LV-1007 Riga, Latvia.

PMID: 32455633
PMCID: PMC7290765
DOI: 10.3390/jcm9051557

Abstract

This review aimed at determining the prevalence and incidence of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) in Europe. We conducted a primary search in Scopus, PubMed and Web of Science for publications between 1994 and 15 June 2019 (PROSPERO: CRD42017078688). Additionally, we performed a backward-(reference lists) and forward-(citations) search of the works included in this review. Grey literature was addressed by contacting all members of the European Network on ME/CFS (EUROMENE). Independent reviewers searched, screened and selected studies, extracted data and evaluated the methodological and reporting quality. For prevalence, two studies in adults and one study in adolescents were included. Prevalence ranged from 0.1% to 2.2%. Two studies also included incidence estimates. In conclusion, studies on the prevalence and incidence of ME/CFS in Europe were scarce. Our findings point to the pressing need for well-designed and statistically powered epidemiological studies. To overcome the shortcomings of the current state-of-the-art, EUROMENE recommends that future research is better conducted in the community, reviewing the clinical history of potential cases, obtaining additional objective information (when needed) and using adequate ME/CFS case definitions; namely, the Centers for Disease Control & Prevention-1994, Canadian Consensus Criteria, or Institute of Medicine criteria.

Keywords: central nervous system diseases; infections; muscular diseases; post-exertional malaise; virus diseases.

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Conflict of interest statement

The authors declare no conflict of interest, and the funders of individual researchers had no role in the design of the study; in the collection, analyses, or interpretation of data; in the writing of the manuscript, or in the decision to publish the results.

Figures

**Figure 1**
Flow diagram for study selection. Notes. WOS, web of science.

See this image and copyright information in PMC

References

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1. Institute of Medicine I.O.M. Beyond Myalgic Encephalomyelitis/Chronic Fatigue Syndrome: Redefining an Illness. Natl. Acad. Press. 2015
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Systematic Review of the Epidemiological Burden of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome Across Europe: Current Evidence and EUROMENE Research Recommendations for Epidemiology

Affiliations

Systematic Review of the Epidemiological Burden of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome Across Europe: Current Evidence and EUROMENE Research Recommendations for Epidemiology

Authors

Affiliations

Abstract

Conflict of interest statement

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References

Publication types

Grants and funding

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Full Text Sources

Medical