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

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.

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Long COVID and Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS)—A Systemic Review and Comparison of Clinical Presentation and Symptomatology

Medicina ◽

10.3390/medicina57050418 ◽

2021 ◽

Vol 57 (5) ◽

pp. 418

Author(s):

Timothy L. Wong ◽

Danielle J. Weitzer

Keyword(s):

Chronic Fatigue Syndrome ◽

Literature Search ◽

Clinical Presentation ◽

Chronic Fatigue ◽

Myalgic Encephalomyelitis ◽

Systemic Review ◽

Future Research ◽

Medical Illness ◽

Case Definitions ◽

Fatigue Syndrome

Background and Objectives: Long COVID defines a series of chronic symptoms that patients may experience after resolution of acute COVID-19. Early reports from studies with patients with long COVID suggests a constellation of symptoms with similarities to another chronic medical illness—myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS). A review study comparing and contrasting ME/CFS with reported symptoms of long COVID may yield mutualistic insight into the characterization and management of both conditions. Materials and Methods: A systemic literature search was conducted in MEDLINE and PsycInfo through to 31 January 2021 for studies related to long COVID symptomatology. The literature search was conducted in accordance with PRISMA methodology. Results: Twenty-one studies were included in the qualitative analysis. Long COVID symptoms reported by the included studies were compared to a list of ME/CFS symptoms compiled from multiple case definitions. Twenty-five out of 29 known ME/CFS symptoms were reported by at least one selected long COVID study. Conclusions: Early studies into long COVID symptomatology suggest many overlaps with clinical presentation of ME/CFS. The need for monitoring and treatment for patients post-COVID is evident. Advancements and standardization of long COVID research methodologies would improve the quality of future research, and may allow further investigations into the similarities and differences between long COVID and ME/CFS.

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Current Research Provides Insight into the Biological Basis and Diagnostic Potential for Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS)

Diagnostics ◽

10.3390/diagnostics9030073 ◽

2019 ◽

Vol 9 (3) ◽

pp. 73 ◽

Cited By ~ 3

Author(s):

Sweetman ◽

Noble ◽

Edgar ◽

Mackay ◽

Helliwell ◽

...

Keyword(s):

Chronic Fatigue Syndrome ◽

Chronic Fatigue ◽

Preclinical Study ◽

Myalgic Encephalomyelitis ◽

Institute Of Medicine ◽

Case Definitions ◽

Biological Basis ◽

Fatigue Syndrome ◽

Diagnostic Potential ◽

Long Term Impact

Myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) is a severe fatigue illness that occurs most commonly following a viral infection, but other physiological triggers are also implicated. It has a profound long-term impact on the life of the affected person. ME/CFS is diagnosed primarily by the exclusion of other fatigue illnesses, but the availability of multiple case definitions for ME/CFS has complicated diagnosis for clinicians. There has been ongoing controversy over the nature of ME/CFS, but a recent detailed report from the Institute of Medicine (Academy of Sciences, USA) concluded that ME/CFS is a medical, not psychiatric illness. Importantly, aspects of the biological basis of the ongoing disease have been revealed over the last 2–3 years that promise new leads towards an effective clinical diagnostic test that may have a general application. Our detailed molecular studies with a preclinical study of ME/CFS patients, along with the complementary research of others, have reported an elevation of inflammatory and immune processes, ongoing neuro-inflammation, and decreases in general metabolism and mitochondrial function for energy production in ME/CFS, which contribute to the ongoing remitting/relapsing etiology of the illness. These biological changes have generated potential molecular biomarkers for use in diagnostic ME/CFS testing.

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The evidence base for physiotherapy in myalgic encephalomyelitis/chronic fatigue syndrome when considering post-exertional malaise: a systematic review and narrative synthesis

Journal of Translational Medicine ◽

10.1186/s12967-020-02683-4 ◽

2021 ◽

Vol 19 (1) ◽

Author(s):

Marjon E. A. Wormgoor ◽

Sanne C. Rodenburg

Keyword(s):

Systematic Review ◽

Chronic Fatigue Syndrome ◽

Diagnostic Criteria ◽

Scientific Evidence ◽

Chronic Fatigue ◽

Evidence Base ◽

Myalgic Encephalomyelitis ◽

Current Evidence ◽

Case Definitions ◽

Fatigue Syndrome

Abstract Background Due to the inconsistent use of diagnostic criteria in myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS), it is unsure whether physiotherapeutic management regarded effective in ME/CFS is appropriate for patients diagnosed with criteria that consider post-exertional malaise (PEM) as a hallmark feature. Purpose To appraise current evidence of the effects of physiotherapy on symptoms and functioning in ME/CFS patients in view of the significance of PEM in the applied diagnostic criteria for inclusion. Methods A systematic review of randomized controlled trials published over the last two decades was conducted. Studies evaluating physiotherapeutic interventions for adult ME/CFS patients were included. The diagnostic criteria sets were classified into three groups according to the extent to which the importance of PEM was emphasized: chronic fatigue (CF; PEM not mentioned as a criterion), CFS (PEM included as an optional or minor criterion) or ME (PEM is a required symptom). The main results of included studies were synthesized in relation to the classification of the applied diagnostic criteria. In addition, special attention was given to the tolerability of the interventions. Results Eighteen RCTs were included in the systematic review: three RCTs with CF patients, 14 RCTs with CFS patients and one RCT covering ME patients with PEM. Intervention effects, if any, seemed to disappear with more narrow case definitions, increasing objectivity of the outcome measures and longer follow-up. Conclusion Currently, there is no scientific evidence when it comes to effective physiotherapy for ME patients. Applying treatment that seems effective for CF or CFS patients may have adverse consequences for ME patients and should be avoided.

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The adoption of chronic fatigue syndrome/myalgic encephalomyelitis case definitions to assess prevalence: a systematic review

Annals of Epidemiology ◽

10.1016/j.annepidem.2013.04.003 ◽

2013 ◽

Vol 23 (6) ◽

pp. 371-376 ◽

Cited By ~ 20

Author(s):

Samantha Johnston ◽

Ekua W. Brenu ◽

Donald R. Staines ◽

Sonya Marshall-Gradisnik

Keyword(s):

Systematic Review ◽

Chronic Fatigue Syndrome ◽

Chronic Fatigue ◽

Myalgic Encephalomyelitis ◽

Case Definitions ◽

Fatigue Syndrome

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Case definitions for chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME): a systematic review

BMJ Open ◽

10.1136/bmjopen-2013-003973 ◽

2014 ◽

Vol 4 (2) ◽

pp. e003973 ◽

Cited By ~ 112

Author(s):

Kjetil Gundro Brurberg ◽

Marita Sporstøl Fønhus ◽

Lillebeth Larun ◽

Signe Flottorp ◽

Kirsti Malterud

Keyword(s):

Systematic Review ◽

Chronic Fatigue Syndrome ◽

Chronic Fatigue ◽

Myalgic Encephalomyelitis ◽

Case Definitions ◽

Fatigue Syndrome

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A Rose By Any Other Name?

Canadian Journal of General Internal Medicine ◽

10.22374/cjgim.v10i4.88 ◽

2016 ◽

Vol 10 (4) ◽

Author(s):

Richard Marlin PhD

Keyword(s):

United States ◽

Chronic Fatigue Syndrome ◽

Diagnostic Criteria ◽

Functional Impairment ◽

Chronic Fatigue ◽

The United States ◽

Myalgic Encephalomyelitis ◽

Expert Committee ◽

Institute Of Medicine ◽

Fatigue Syndrome

In February of this year, an expert committee of the United States Institute of Medicine (IOM) released a lengthy report in which its members reviewed diagnostic criteria and proposed a new label for chronic fatigue syndrome, also historically referred to as myalgic encephalomyelitis.1 The committee’s proposed new label for this illness is Systemic Exertion Intolerance Disease. The report refers to the fact that a sizeable population is diagnosed with this illness, which causes considerable suffering and functional impairment. Many patients also feel stigmatized because of the label chronic fatigue syndrome.

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Systematic review of randomized controlled trials for chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME)

Journal of Translational Medicine ◽

10.1186/s12967-019-02196-9 ◽

2020 ◽

Vol 18 (1) ◽

Cited By ~ 8

Author(s):

Do-Young Kim ◽

Jin-Seok Lee ◽

Samuel-Young Park ◽

Soo-Jin Kim ◽

Chang-Gue Son

Keyword(s):

Systematic Review ◽

Chronic Fatigue Syndrome ◽

Randomized Controlled Trials ◽

Chronic Fatigue ◽

Myalgic Encephalomyelitis ◽

Controlled Trials ◽

Pharmacological Interventions ◽

Case Definitions ◽

Randomized Controlled ◽

Fatigue Syndrome

Abstract Background Although medical requirements are urgent, no effective intervention has been proven for chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME). To facilitate the development of new therapeutics, we systematically reviewed the randomized controlled trials (RCTs) for CFS/ME to date. Methods RCTs targeting CFS/ME were surveyed using two electronic databases, PubMed and the Cochrane library, through April 2019. We included only RCTs that targeted fatigue-related symptoms, and we analyzed the data in terms of the characteristics of the participants, case definitions, primary measurements, and interventions with overall outcomes. Results Among 513 potentially relevant articles, 56 RCTs met our inclusion criteria; these included 25 RCTs of 22 different pharmacological interventions, 29 RCTs of 19 non-pharmacological interventions and 2 RCTs of combined interventions. These studies accounted for a total of 6956 participants (1713 males and 5243 females, 6499 adults and 457 adolescents). CDC 1994 (Fukuda) criteria were mostly used for case definitions (42 RCTs, 75.0%), and the primary measurement tools included the Checklist Individual Strength (CIS, 35.7%) and the 36-item Short Form health survey (SF-36, 32.1%). Eight interventions showed statistical significance: 3 pharmacological (Staphypan Berna, Poly(I):poly(C12U) and CoQ10 + NADH) and 5 non-pharmacological therapies (cognitive-behavior-therapy-related treatments, graded-exercise-related therapies, rehabilitation, acupuncture and abdominal tuina). However, there was no definitely effective intervention with coherence and reproducibility. Conclusions This systematic review integrates the comprehensive features of previous RCTs for CFS/ME and reflects on their limitations and perspectives in the process of developing new interventions.

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Does the microbiome and virome contribute to myalgic encephalomyelitis/chronic fatigue syndrome?

Clinical Science ◽

10.1042/cs20171330 ◽

2018 ◽

Vol 132 (5) ◽

pp. 523-542 ◽

Cited By ~ 12

Author(s):

Fiona Newberry ◽

Shen-Yuan Hsieh ◽

Tom Wileman ◽

Simon R. Carding

Keyword(s):

Chronic Fatigue Syndrome ◽

Study Design ◽

Chronic Fatigue ◽

Myalgic Encephalomyelitis ◽

Current Evidence ◽

Fatigue Syndrome ◽

Health And Disease ◽

Host Lysis ◽

Diagnosis Criteria ◽

Endocrine Symptoms

Myalgic encephalomyelitis (ME)/chronic fatigue syndrome (CFS) (ME/CFS) is a disabling and debilitating disease of unknown aetiology. It is a heterogeneous disease characterized by various inflammatory, immune, viral, neurological and endocrine symptoms. Several microbiome studies have described alterations in the bacterial component of the microbiome (dysbiosis) consistent with a possible role in disease development. However, in focusing on the bacterial components of the microbiome, these studies have neglected the viral constituent known as the virome. Viruses, particularly those infecting bacteria (bacteriophages), have the potential to alter the function and structure of the microbiome via gene transfer and host lysis. Viral-induced microbiome changes can directly and indirectly influence host health and disease. The contribution of viruses towards disease pathogenesis is therefore an important area for research in ME/CFS. Recent advancements in sequencing technology and bioinformatics now allow more comprehensive and inclusive investigations of human microbiomes. However, as the number of microbiome studies increases, the need for greater consistency in study design and analysis also increases. Comparisons between different ME/CFS microbiome studies are difficult because of differences in patient selection and diagnosis criteria, sample processing, genome sequencing and downstream bioinformatics analysis. It is therefore important that microbiome studies adopt robust, reproducible and consistent study design to enable more reliable and valid comparisons and conclusions to be made between studies. This article provides a comprehensive review of the current evidence supporting microbiome alterations in ME/CFS patients. Additionally, the pitfalls and challenges associated with microbiome studies are discussed.

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