Comparative Survey of People with ME/CFS in Italy, Latvia, and the UK: A Report on Behalf of the Socioeconomics Working Group of the European ME/CFS Research Network (EUROMENE).

Background and Objectives: A comparative survey of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) patients was carried out in three countries, with the aim of identifying appropriate policy measures designed to alleviate the burden of disease both on patients and their families, and also on public institutions. The survey addressed demographic features, the economic impact of the disease on household incomes, patterns of medical and social care, specific therapies, social relationships, and the impact of the illness on quality of life. Materials and Methods: Parallel surveys were undertaken in Italy, Latvia, and the UK. There were 88 completed responses from Italy, 75 from Latvia, and 448 from the UK. To facilitate comparisons, 95% confidence intervals were calculated in respect of responses to questions from all three countries. To explore to what extent general practitioners (GPs) manage ME/CFS disease, a separate questionnaire for GPs, with questions about the criteria for granting a diagnosis, laboratory examinations, the involvement of specialists, and methods of treatment, was undertaken in Latvia, and there were 91 completed responses from GPs. Results: The results are presented in respect of sociodemographic information, household income, disease progression and management, perceived effectiveness of treatment, responsibility for medical care, personal care, difficulty explaining the illness, and quality of life. Demographic details were similar in all three countries, and the impact of illness on net household incomes and quality of life. There were significant differences between the three countries in illness progression and management, which may reflect differences in patterns of health care and in societal attitudes. Graded exercise therapy, practiced in the UK, was found to be universally ineffective. Conclusions: There were similarities between respondents in all three countries in terms of demographic features, the impact of the illness on household incomes and on quality of life, and on difficulties experienced by respondents in discussing their illness with doctors, but also differences in patterns of medical care, availability of social care, and societal attitudes to ME/CFS.

The Role of Prevention in Reducing the Economic Impact of ME/CFS in Europe: A Report from the Socioeconomics Working Group of the European Network on ME/CFS (EUROMENE).

This report addresses the extent to which there may be scope for preventive programmes for Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS), and, if so, what economic benefits may accrue from the implementation of such programmes. We consider the economic case for prevention programmes, whether there is scope for preventive programmes for ME/CFS, and what are the health and economic benefits to be derived from the implementation of such programmes. We conclude that there is little scope for primary prevention programmes, given that ME/CFS is attributable to a combination of host and environmental risk factors, with host factors appearing to be most prominent, and that there are few identified modifiable risk factors that could be the focus of such programmes. The exception is in the use of agricultural chemicals, particularly organophosphates, where there is scope for intervention, and where Europe-wide programmes of health education to encourage safe use would be beneficial. There is a need for more research on risk factors for ME/CFS to establish a basis for the development of primary prevention programmes, particularly in respect of occupational risk factors. Secondary prevention offers the greatest scope for intervention, to minimise diagnostic delays associated with prolonged illness, increased severity, and increased costs.

Perceptions of European ME/CFS Experts Concerning Knowledge and Understanding of ME/CFS among Primary Care Physicians in Europe: A Report from the European ME/CFS Research Network (EUROMENE).

Background and Objectives: We have conducted a survey of academic and clinical experts who are participants in the European ME/CFS Research Network (EUROMENE) to elicit perceptions of general practitioner (GP) knowledge and understanding of myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) and suggestions as to how this could be improved. Materials and Methods: A questionnaire was sent to all national representatives and members of the EUROMENE Core Group and Management Committee. Survey responses were collated and then summarized based on the numbers and percentages of respondents selecting each response option, while weighted average responses were calculated for questions with numerical value response options. Free text responses were analysed using thematic analysis. Results: Overall there were 23 responses to the survey from participants across 19 different European countries, with a 95% country-level response rate. Serious concerns were expressed about GPs' knowledge and understanding of ME/CFS, and, it was felt, about 60% of patients with ME/CFS went undiagnosed as a result. The vast majority of GPs were perceived to lack confidence in either diagnosing or managing the condition. Disbelief, and misleading illness attributions, were perceived to be widespread, and the unavailability of specialist centres to which GPs could refer patients and seek advice and support was frequently commented upon. There was widespread support for more training on ME/CFS at both undergraduate and postgraduate levels. Conclusion: The results of this survey are consistent with the existing scientific literature. ME/CFS experts report that lack of knowledge and understanding of ME/CFS among GPs is a major cause of missed and delayed diagnoses, which renders problematic attempts to determine the incidence and prevalence of the disease, and to measure its economic impact. It also contributes to the burden of disease through mismanagement in its early stages.

The implementation of health technology assessment principles in public decisions concerning orphan drugs.

PURPOSE: Over the last few years, the share of public spending for orphan drugs (ODs) has increased in several western countries, raising concern on the exemptions granted to this sector with respect to the implementation of health technology assessment (HTA) principles. The aim of this paper is to shed light on both the HTA criteria adopted and the international agreements implemented in the OD regulation, given the new challenges imposed on western countries by a growing number of therapies for rare diseases. METHODS: We carried out a literature review to analyse the development of the international debate on the adaptability of HTA criteria for the OD assessment and regulation. The time span lies between January 1990 and May 2018, and the policies considered relate to both market authorization and reimbursement decisions within western countries. We focus specifically on HTA criteria in some of the dimensions included in the Core Model of the European net for HTA (EUnetHTA). RESULTS: OD high prices, the absence of clarity on the possible high revenues realized by the distribution of a new OD outside the national borders, the risk that - once marketed - a new OD can be used to treat common diseases, are all issues that raise concern on OD regulation and have to be carefully monitored by policymakers in the next future. CONCLUSIONS: Across western countries, the preferential track granted to ODs in the implementation of HTA principles is not homogeneous, but fragmented and differentiated. The need for common rules at an international level is underlined, with a view to assessing the sustainability of a sector which, due to this regulatory void, can lend itself to producers' strategic and opportunistic behaviours.

A Literature Review of GP Knowledge and Understanding of ME/CFS: A Report from the Socioeconomic Working Group of the European Network on ME/CFS (EUROMENE).

Background and Objectives: The socioeconomic working group of the European myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) Research Network (EUROMENE) has conducted a review of the literature pertaining to GPs' knowledge and understanding of ME/CFS; Materials and Methods: A MEDLINE search was carried out. The papers identified were reviewed following the synthesis without meta-analysis (SWiM) methodology, and were classified according to the focus of the enquiry (patients, GPs, database and medical record studies, evaluation of a training programme, and overview papers), and whether they were quantitative or qualitative in nature; Results: Thirty-three papers were identified in the MEDLINE search. The quantitative surveys of GPs demonstrated that a third to a half of all GPs did not accept ME/CFS as a genuine clinical entity and, even when they did, they lacked confidence in diagnosing or managing it. It should be noted, though, that these papers were mostly from the United Kingdom. Patient surveys indicated that a similar proportion of patients was dissatisfied with the primary medical care they had received. These findings were consistent with the findings of the qualitative studies that were examined, and have changed little over several decades; Conclusions: Disbelief and lack of knowledge and understanding of ME/CFS among GPs is widespread, and the resultant diagnostic delays constitute a risk factor for severe and prolonged disease. Failure to diagnose ME/CFS renders problematic attempts to determine its prevalence, and hence its economic impact.

Analysing outpatient care access for planning purposes: The Basilicata Region experience.

The delivery of healthcare services at regional level should be modeled on population needs, stemming from patients' consumption pattern and trying to include unexpressed needs and exclude excesses of demand due to both moral hazard behaviors and inducements from the supply side. We propose a model able to estimate the frequency of access in outpatient care (OC) based on the characteristics of the population. According to empirical evidence, among the determinants of outpatient access we include variables addressing health, socioeconomic status and place of residence, plus variables related to the supply of services. We run generalized linear models for counting data of the Poisson family with the aim of both identifying the determinants of OC utilization and quantifying the related effects. We use the regional administrative database of Basilicata region, year 2019. Results are consistent with literature findings and provide new insights into the analysis of OC, suggesting that our model could easily be implemented by regional policymakers to plan the supply of ambulatory services on population needs.

Should I care for my mum or for my kid? Sandwich generation and depression burden in Italy.

SETTING AND OBJECTIVE: In Italy, over the last decades, elderly care has been mostly provided by family members, especially adult offspring, and in particular daughters. This paper investigates the relationship between informal caregiving and mental distress among Italians aged 35-59, with a focus on gender effect and parenthood responsibilities. DATA: The dataset is the European Health Interview Survey (EHIS), second wave, year 2015. As far as it is known, the Italian EHIS has not been used for studies on ageing and caregiving. METHODS: Using selected subsamples, a Propensity Score Matching between caregivers and non-caregivers aged 35-59 is implemented, with the aim of measuring the difference in level of depression, if any, between the two groups. RESULTS: Findings show that women providing their frail relatives with informal care are less likely to suffer from mental distress compared to non-carers. However, results change radically if they have children aged less than 15 at home, and a higher probability of being depressed is detected for women overwhelmed by the double responsibility of assisting both dependent relatives and their own children. Results are not significant for men.

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).

We have developed a Europe-wide approach to investigating the economic impact of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS), facilitating acquisition of information on the economic burden of ME/CFS, and international comparisons of economic costs between countries. The economic burden of ME/CFS in Europe appears large, with productivity losses most significant, giving scope for substantial savings through effective prevention and treatment. However, economic studies of ME/CFS, including cost-of-illness analyses and economic evaluations of interventions, are problematic due to different, arbitrary case definitions, and unwillingness of doctors to diagnose it. We therefore lack accurate incidence and prevalence data, with no obvious way to estimate costs incurred by undiagnosed patients. Other problems include, as for other conditions, difficulties estimating direct and indirect costs incurred by healthcare systems, patients and families, and heterogeneous healthcare systems and patterns of economic development across countries. We have made recommendations, including use of the Fukuda (CDC-1994) case definition and Canadian Consensus Criteria (CCC), a pan-European common symptom checklist, and implementation of prevalence-based cost-of-illness studies in different countries using an agreed data list. We recommend using purchasing power parities (PPP) to facilitate international comparisons, and EuroQol-5D as a generic measure of health status and multi-attribute utility instrument to inform future economic evaluations in ME/CFS.

Rich and Well Educated: Are These Requirements Necessary to Claim Healthcare Tax Credits in Italy?

BACKGROUND: The paper investigates the use of healthcare tax credits (HTCs) in Italy through the analysis of a panel data, which provides information on individual income tax from 2008 to 2014. There is evidence of disparities in the per-capita HTCs between Northern and Southern regions, which need to be analyzed and addressed. OBJECTIVE: The aim of the paper is to investigate the socioeconomic determinants in the use of Healthcare Tax Credits in Italy. METHODS: A fixed effects Ordinary Least Square model is run to analyze the impact of selected socioeconomic variables on regional per capita HTCs, with a particular focus on the role of education. RESULTS: The results corroborate literature findings on the regressive effects of HTCs; they also provide highlights on the role of education in explaining the distribution of HTCs among Italian regions. CONCLUSION: Public money is reimbursed to regions where people are, on average, richer and better educated. More equitable objectives could be reached by allocating the same resources in the provision of services covered by the NHS.

To What Extent Is Long-term Care Representative of Elderly Care? A Case Study of Elderly Care Financing in Lombardy, Italy.

The ageing of European population has been rapidly increasing during the last decades, and the problem of elderly care financing has become an issue for policy-makers. Long-term care (LTC) financing is considered a suitable proxy of the resources committed to elderly care by each government, but the preciseness of this approximation depends on the extent to which LTC is representative of elderly care within each country. Since there is a broad heterogeneity in LTC funding, organization and setting among European States, it is difficult to find a common parameter representing the public resources destined to the elderly care. We address these topics employing as a case study an Italian region, Lombardy, which in terms of population, dimension, healthcare organization and economic development could be compared to other European countries. The method we suggest, which consists basically in a careful estimate of all the public resources employed in the provision of services exclusively destined to the elderly, could be applied, with the due differences, to other European countries or regions.

Regional incentives and patient cross-border mobility: evidence from the Italian experience.

BACKGROUND: In recent years, accreditation of private hospitals followed by decentralisation of the Italian National Health Service (NHS) into 21 regional health systems has provided a good empirical ground for investigating the Tiebout principle of "voting with their feet". We examine the infra-regional trade-off between greater patient choice (due to an increase in hospital services supply) and financial equilibrium, and we relate it to the significant phenomenon of Cross-Border Mobility (CBM) between Italian regions. Focusing on the rules supervising the financial agreements between regional authorities and providers of hospital care, we find incentives for private accredited providers in attracting patient inflows. METHODS: The analysis is undertaken from an institutional, regulatory and empirical perspective. We select a sample of five regions with higher positive CBM balance and we examine regional regulations governing the contractual agreements between purchasers and providers of hospital care. According to this sample, we provide a statistical analysis of CBM and apply a Regional Attraction Ability Index (RAAI), aimed at testing patient preferences for private/public accredited providers. RESULTS: We find that this index is systematically higher for private providers, both in the case of distance/boundary patients and of excellence/general hospitals. CONCLUSION: Conclusions address both financial issues regarding the coverage of regional healthcare systems and equity issues on patient healthcare access. They also raise concerns on the new European Union (EU) directive inherent to patient mobility across Europe.

Quasi-market and cost-containment in Beveridge systems: the Lombardy model of Italy.

In the very recent past, the Lombardy health care system - established in 1997 on the quasi market model - has caught the interest of researchers and politicians in different OECD countries(1). Its merits, compared to other Italian regional systems, are the control of health care spending and the balanced budget, in a frame of good quality of services and patient choice. From the theoretical point of view, an appealing aspect of the Lombardy model is its gradual shift from a quasi market (QM) to a "quasi administered" system, which maintains all the typical features of the QM orientation - separation between purchasers and providers, the co-presence of public, not for profit and public providers, and patient free choice - but has deliberately sacrificed competition in order to control health expenditure. Another aspect of the Lombardy model is the sharp presence of private providers: the evidence that private sector is mainly concentrated in the long term care, where risks of complications are lower and financial remuneration is higher, suggests that a closer control should be exerted on hospital activity. Furthermore, possible distortions such as cream skimming and cherry picking by the private providers need more consideration. Another concern is linked to health spending control: equity issues could arise when observing a still relatively high share of private (out of pocket) health care expenditure. The paper stems from a literature review and tries to analyse the evolution of this regional system, the institutional path that brought to the implementation of the model, its theoretical basis, its merits and criticism. The period considered ranges from 1997, when the reform was enacted, to 2010.