The Development of a Consistent Europe-Wide Approach to Investigating the Economic Impact of Myalgic Encephalomyelitis (ME/CFS): A Report from the European Network on ME/CFS (EUROMENE)
2. European Network on ME/CFS (EUROMENE)—A European Cost Action Project
3. Challenges in Developing a Consistent Europe-Wide Approach to Measuring the Economic Impact of ME/CFS
3.1. Overview of Challenges
3.2. Case Definition and Prevalence Rates
3.3. Case Ascertainment
3.4. Determination of Costs
3.5. Europe-Wide Comparisons
Conflicts of Interest
|ME/CFS||Myalgic Encephalomyelitys/Chronic Fatigue Syndrome|
|EUROMENE||European Network on ME/CFS|
|COI||Cost of illness|
|EuroQol-5D||An instrument for measuring quality of life|
|CCC||Canadian Consensus Criteria|
|PPP||Purchasing Power Parity|
|ICD-10 CM||The international diagnostic classification standard for reporting diseases, disorders, injuries and health conditions for all clinical and research purposes|
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|1.||Case definition||ME/CFS is a syndrome, defined in terms of its symptomatology rather than its underlying pathology. Work done in this area is therefore dependent on case definitions, which of their very nature are arbitrary. In addition, there are numerous case definitions in use, which vary substantially in sensitivity and specificity, and do not necessarily identify the same population.|
|2.||Incidence and prevalence||Little is known about the incidence or prevalence of ME/CFS. Very little work has been done in this area in Europe, except in the UK. Conclusions drawn from UK experience, or indeed from work done in other countries, in particular the USA and Australia, cannot be readily extrapolated to Europe as a whole, because the extent of natural variation between populations is unknown.|
|3.||Failure to diagnose||A high proportion of doctors, in particular GPs, refuse to recognise ME/CFS as a genuine clinical entity, and as a result do not diagnose it. Even in countries where ME/CFS is officially recognised, this proportion may be as high as 50%. It is not possible therefore to obtain accurate prevalence data through the use of service utilisation data.|
|4.||Determination of costs and losses||Any attempt to determine costs and economic losses attributable to ME/CFS must take into account direct and indirect costs incurred both by healthcare systems, patients and families, as well as productivity losses. This applies equally to patients who have been diagnosed as having ME/CFS and those who have not received a diagnosis, including for the reasons outlined in (iii) above. It is likely to be difficult to identify costs for this latter group, for obvious reasons.|
|5.||Variation within the ME/CFS population||It can be hypothesised that, for example, severely affected people (housebound or bedbound) may incur greater overall costs than mildly or moderately affected people. There is no information available which could shed light on this, which clearly requires further research.|
|6.||Heterogeneity of national economies and health care systems||Against such a background, it is clearly an uphill struggle to reach meaningful conclusions about the costs and losses attributable to ME/CFS across Europe, particularly given the variety of systems of healthcare delivery in Europe, and varying stages of economic development.|
|Area of Concern||Recommendation|
|1||Case definition||That there should be Europe-wide adoption of the Fukuda (CDC-1994) case definition alongside the Canadian Consensus Criteria (CCC).|
|2||Case identification||That a common symptom checklist should be used, capable of being mapped by algorithms onto both the Fukuda case definition and the CCC.|
|3||Prevalence and incidence||Better descriptive epidemiological information is required, as a basis for economic investigation. This should include information concerning the proportion of severely affected people, as there are likely to be different cost implications for such people, in comparison with those with mild or moderate illnesses.|
|4||Economic investigation||Prevalence based cost of illness studies, based on these case definitions, should be carried out in different countries, to determine the overall cost burden attributable to ME/CFS.|
|5||Data items||A list of data items required for cost of illness studies has been identified (though not reported here). Individual participating countries should examine this, to ensure that, insofar as these are derivable from routine data collection, systems are in place to ensure that they are collected.|
|6||Data audit||The availability in participating countries of the relevant data items referred to above which are required for cost-of illness studies should be examined, with a view to achieving convergence, and facilitating international comparisons.|
|7||Relationship between disease severity and economic impact||The EuroQol-5D instrument [52,53] should be used as a generic measure of health status and as a multi-attribute utility instrument to determine the relationship, if any, between disease severity and economic impacts, as in the Italian study reported in this document , and to inform future economic evaluations in ME/CFS. We further recommend that the Italian study be replicated in other countries, to enable international comparisons to be made.|
|8||International comparisons and compilation of Europe-wide statistics||Given the diversity of patterns of health care organisations and health funding, as well as of outcomes and general levels of health, and of national wealth and levels of economic development, we recommend the use of purchasing power parities (PPP) in order both to make valid international comparisons and to collate meaningful statistics at a European level.|
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Pheby, D.F.H.; Araja, D.; Berkis, U.; Brenna, E.; Cullinan, J.; de Korwin, J.-D.; Gitto, L.; Hughes, D.A.; Hunter, R.M.; Trepel, D.; et al. The Development of a Consistent Europe-Wide Approach to Investigating the Economic Impact of Myalgic Encephalomyelitis (ME/CFS): A Report from the European Network on ME/CFS (EUROMENE). Healthcare 2020, 8, 88. https://doi.org/10.3390/healthcare8020088
Pheby DFH, Araja D, Berkis U, Brenna E, Cullinan J, de Korwin J-D, Gitto L, Hughes DA, Hunter RM, Trepel D, et al. The Development of a Consistent Europe-Wide Approach to Investigating the Economic Impact of Myalgic Encephalomyelitis (ME/CFS): A Report from the European Network on ME/CFS (EUROMENE). Healthcare. 2020; 8(2):88. https://doi.org/10.3390/healthcare8020088Chicago/Turabian Style
Pheby, Derek F.H., Diana Araja, Uldis Berkis, Elenka Brenna, John Cullinan, Jean-Dominique de Korwin, Lara Gitto, Dyfrig A Hughes, Rachael M Hunter, Dominic Trepel, and et al. 2020. "The Development of a Consistent Europe-Wide Approach to Investigating the Economic Impact of Myalgic Encephalomyelitis (ME/CFS): A Report from the European Network on ME/CFS (EUROMENE)" Healthcare 8, no. 2: 88. https://doi.org/10.3390/healthcare8020088