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Objectives This study tries to analyze how the crisis generated by severe acute respiratory syndrome coronavirus 2 has affected the reported mental health symptoms of informal caregivers in different European countries. Methods The Survey of Health, Ageing and Retirement in Europe-COVID-19 was used, collecting information from the beginning of June 2020 to August 2020 about individuals’ state of health and the care they received. Several probit regression models were used to analyze the differences in the probability of (1) being sad or depressed, (2) being anxious or nervous, (3) having difficulty sleeping, and (4) feeling lonely, between individuals who provided informal care and individuals who did not. Several subanalyses by geographic area, mortality rates due to coronavirus disease 2019 (COVID-19), and long-term care expenditure were also performed. Results Since the outbreak of COVID-19, informal caregivers have had a higher probability of being sad or depressed of 8 percentage points (p.p.), a 7.1 p.p. higher probability of being anxious or nervous, and a 5.9 p.p. higher probability of having difficulty sleeping than non-caregivers. Informal caregivers in Southern Europe have had an 8 p.p. higher probability of being sad or depressed than non-caregivers. In Eastern Europe, this difference in probability reaches 9.7 p.p. Finally, in countries with higher mortality rates due to COVID-19, there have been greater differences in terms of being sad or depressed between caregivers and non-caregivers, regardless of expenditure on long-term care. Conclusions Since the outbreak of COVID-19, informal caregivers in Europe have had a higher probability of reporting mental health symptoms than non-caregivers.

Background Measurement of muscle mass and function, and thereafter, screening and diagnosis of sarcopenia, is a challenge and a need in hospitalized older adults. However, it is difficult in complex real-world old patients, because usually they are unable to collaborate with clinical, functional, and imaging testing. Ultrasound measurement of quadriceps rectus femoris (QRF) provides a non-invasive, real-time assessment of muscle quantity and quality, and is highly acceptable to participants with excellent inter-rater and intra-rater variability. However, normative data, protocol standardization, and association with longitudinal outcomes, needs further research and consensus. Methods Prospective exploratory multicenter study in older adults admitted to Acute Geriatric Units (AGUs) for medical reasons. 157 subjects from 7 AGUs of Spain were recruited between May 2019 and January 2022. Muscle ultrasound measurements of the anterior vastus of the QRF were acquired on admission and on discharge, using a previously validated protocol, using a Chieson model ECO2 ultrasound system (Chieson Medical Technologies, Co. Ltd, Wimxu District Wuxi, Jiangsu, China). Measurements included the cross-sectional area, muscle thickness in longitudinal view, intramuscular central tendon thickness, echogenicity, and the presence or absence of edema and fasciculations. Functional, nutritional, and DXA measurements were provided. Clinical follow-up was completed at discharge, and 30 and 90 days after discharge. Variations between hospital admission and discharge ultrasound values, and the relationship with clinical variables, will be analyzed using paired t-tests, Wilcoxon tests, or Mc Nemar chi-square tests when necessary. Prevalence of sarcopenia will be calculated, as well as sensitivity and specificity of ultrasound measurements to determine sarcopenia. Kappa analysis will be used to analyze the concordance between measurements, and sensitivity analysis will be conducted for each participating center. Discussion The results obtained will be of great interest to the scientific geriatric community to assess the utility and validity of ultrasound measurements for the detection and follow-up of sarcopenia in hospitalized older adults, and its association with adverse outcomes. Trial registration NCT05113758. Registration date: November 9th 2021. Retrospectively registered.

This study aims to assess the determinants of employment probabilities among people living with Human Immunodeficiency Virus (HIV) during a 15-year period (2001–2016) in Spain, focusing on the possible effects of occurrences such as the 2008 economic crisis. The probability of people living with HIV having a job was evaluated by applying several multivariate probit regression models. Differences between the employment status of people living with HIV and that of the general population were evaluated by applying genetic matching regression models. With respect to the former evaluation, for people living with HIV, the period before the crisis (2001–2007) was associated with a probability of being employed that was 2.43 percentage points (p.p.) higher than during the crisis, and the period after the crisis (2014–2016) with a probability that was 7.58 p.p. lower than during the crisis. Greater effects were also observed among males, the probability of being in employment before the economic crisis being higher (by 2.26 p.p.) and lower after the crisis (− 3.41 p.p.) than among women, and among those infected through drug use (6.18 p.p. and − 7.34 p.p. before and after the crisis, respectively), than among those infected through sex. When analysing the differences with respect to the general population, people living with HIV reported lower probabilities of being employed: by − 18 p.p. before the crisis, by − 15 p.p. during the crisis (years 2008–2013) and by − 10 p.p. after the crisis, implying a convergence in the prospects of employment with the passage of the years. Those differences were greater for people of basic educational level (− 23 to − 16 p.p.), a weaker immune system (− 34 p.p. to − 21 p.p.) and those infected through the use of drugs (− 31 p.p. to − 26 p.p.). Although the results suggest that the economic crisis had a greater effect on the employment prospects of people living with HIV, and that effect is still felt by that group, our findings also point towards a convergence of their employment prospects with those of the general population, over the 15-year period assessed. An analysis of the employment situation of people living with HIV might have helped when designing job-seeking methods and policies on the working environment, especially through the 15-year period considered, when the economic crisis had a greater effect on the job market.

Background The growing number of employed women has been associated with an increase in the prevalence of overweight and obesity in children. We sought to determine whether childhood overweight/obesity in Spain is associated with labour participation of mothers and fathers, and whether the identity of the main caregiver has an influence on child’s weight and unhealthy behaviour. Methods We used microdata from the 2010 and 2014 Health Behaviour in School-Aged Children surveys performed in Spain (n = 32,694). Logistic and linear multi-level regression models were applied to assess the association between parental employment and children’s self-reported weight status, accounting for school effects and controlling for socioeconomic factors. Separated binary models were also fitted for consumption of fruit, sweets, screen viewing and sedentarism. Results In most cases, the significant associations between children’s weight and their parents’ work status disappeared once the models were adjusted for family wealth and education. However, we found persistent associations for some groups. Girls under 13 years-old living in households where the mother was the only employed parent were more likely to be affected by obesity and to report a higher body mass index value. Children in this type of household were more likely to show unhealthy lifestyles related to diet and leisure time activities. Conclusions Parents’ socioeconomic characteristics had a protective effect on their children’s risk of obesity. Unhealthy behaviours were observed in households with a non-working father and a working mother, although the link with obesity was limited to girls. Our results suggest the need for a more equally shared burden of caregiving.

The main data on HIV in high-income countries from the standpoint of health economics are updated and discussed. Specifically, issues surrounding the economic impact of HIV are addressed for health care and occupational perspectives. We review the main epidemiological data on the prevalence of disease and foreseeable changes in patterns within the coming years. Recent research on health care costs of HIV/AIDS and the occupational situation of HIV+ people are discussed. In high-income countries, there is an incipient change in trends that indicate that an increasing percentage of the intermediate-age HIV+ population will age prematurely, presenting more frequently comorbidities and becoming more exposed to frailty situations. Accordingly, health-care systems should have to plan their resources to accommodate new determinants of healthcare costs in the HIV+ population. On the occupational side, the decreased life expectancy of HIV+ persons, coupled with more severe health issues, on average may prompt early -retirement of this population from the labor market.

Las evaluaciones económicas de intervenciones sanitarias se han convertido en los últimos años en una herramienta primordial para informar las decisiones que atañen a la asignación de recursos y la adopción de nuevas tecnologías sanitarias. En el caso de la diabetes mellitus, cuya prevalencia y morbilidad a nivel mundial se ha ido incrementando considerablemente a lo largo de las últimas décadas, resulta primordial desarrollar nuevas estrategias y priorizar aquellas intervenciones que más ayuden a mejorar la salud de la población, haciendo que sea compatible con la sostenibilidad financiera de los sistemas sanitarios públicos. Así, el principal objetivo de este artículo es mostrar al lector las diferentes opciones metodológicas que se deben considerar a la hora de diseñar o de interpretar una evaluación económica en el ámbito de la diabetes mellitus, con el apoyo de ejemplos e investigaciones prácticas llevadas a cabo en el campo de esta enfermedad.

Background: This paper had two aims. Firstly, to provide a broader view of the profile of non-professional caregivers in Europe, and secondly, to estimate the economic value of the non-professional caregiving. Methods: The European Quality of Life Survey 2016/2017, carried out by Eurofound, was used. The target population of the survey was adults who care for a relative or friend in a total of 33 European countries. The opportunity cost method was used to estimate the economic value of caregiving, in which two of the activities forgone were analysed: paid activities (restricted to caregivers who were employed), for which the average gross wage of each country was used; and unpaid activities, for which the minimum gross wage of each country was used. Results: There were more than 76 million non-professional caregivers in Europe that provide care for a relative or friend. This figure represents 12.7% of the population in Europe. The estimated time devoted to non-professional care in Europe reached 72 301.5 million hours in 2016. Sharp differences were found among countries. The economic value of that time is estimated at 576 000 million of euros, which represented about 3.63% of Europe’s gross domestic product (GDP). Conclusion: This study shows the very important number of resources dedicated to the non-professional care of dependent people and their economic valuation. These results may be helpful in prospective analyses estimating future needs on professional and non-professional and for designing of long-term care (LTC) policies in Europe.

Resumen: El objetivo de este artículo es identificar los efectos de la Gran Recesión sobre la salud mental de las personas residentes en España. Tras exponer un marco conceptual sobre los mecanismos por los que las crisis económicas afectan a la salud mental, se describen los principales resultados de 45 trabajos identificados en nuestra búsqueda. Los estudios apuntan a un empeoramiento de la salud mental en España en los años de crisis económica, en especial en los varones. Las condiciones laborales (desempleo, bajos salarios, inestabilidad, precariedad) emergen como uno de los cauces principales a través de los que se pone en riesgo o deteriora la salud mental. Dicho deterioro se produce con intensidad en colectivos particularmente vulnerables, como población inmigrante y familias con cargas económicas. En el caso de los suicidios, no se observan resultados concluyentes. En materia de utilización de servicios sanitarios parece identificarse un aumento en el consumo de ciertos fármacos, si bien las conclusiones de todos los trabajos no son coincidentes. En el caso de las desigualdades sociales en salud mental, estas no parecen haber remitido. Se concluye que es necesario mejorar nuestros sistemas de información para comprender mejor los efectos sobre la salud de las crisis económicas. En materia de políticas públicas, junto con el refuerzo de los servicios sanitarios dirigidos a atender problemas de salud mental, se debería impulsar una red de garantía de rentas para las personas en situación de vulnerabilidad y el desarrollo de políticas dirigidas al medio laboral. Abstract: The objective of this article is to identify the effects of the Great Recession on the mental health of people residing in Spain. After presenting a conceptual framework on the mechanisms through which economic crises affect mental health, we describe the main results of 45 papers identified in our search. Studies indicate a worsening of mental health in Spain in the years of economic crisis, especially in men. Working conditions (unemployment, low wages, instability, precariousness) emerge as one of the main channels through which mental health is put at risk or deteriorates. This deterioration occurs with intensity in particularly vulnerable groups, such as immigrant population and families with economic burdens. In the case of suicides, the results were inconclusive. Regarding the use of health care services, an increase in the consumption of certain drugs seems to be identified, although the conclusions of all the studies are not coincidental. Social inequalities in mental health do not seem to have remitted. We conclude that Spain needs to improve information systems to a better understanding of the health effects of economic crises. In terms of public policies, together with the reinforcement of health services aimed at addressing mental health problems, an income guarantee network for people in vulnerable situations should be promoted, as well as the development of policies aimed at the labour market.

Facilities have been the focus of the greatest impact of COVID-19 in terms of mortality and extreme situations, along with health centers. The main objective of this article is to describe how the arrival of SARS-CoV-2 affected facilities, focusing on Spain during the first pandemic months, and to point out lessons learned. Despite the measures and regulations approved in the first weeks of March 2020, these centers were not prepared for the arrival of an epidemic such as the one experienced. The clearest indicator of this is a strong impact on mortality in residential facilities. The excess of deaths in residences has been estimated at 26,448 people between March 2020 and May 2021 (10.6% of the total number of dependents cared for in residences, with an excess mortality of 43.5%), with deaths concentrated in the first months of the pandemic. However, there are other effects to be considered such as those that affect the mental health and quality of life of residents, family members, and residential facilities staff. Assuming that no two pandemics are possibly alike, it is essential to draw lessons from lived experience that may be useful to prepare for similar future situations and strengthen a long-term care system that was already frail before the arrival of SARS-CoV-2.

Hepatitis C virus (HCV) infection causes a substantial economic burden, not only in terms of healthcare costs, but also in labour productivity losses. The main objective of this study is to provide objective and comparable information about the trend in labour productivity losses caused by premature HCV-associated deaths in Spain in recent years (2009-2018). We used nationwide data from several official sources to create a simulation model based on the human capital approach and to estimate the flows in labour productivity losses due to deaths identified in the period considered. Based on a pessimistic scenario, the annual number of deaths due to HCV infections decreased by 19.7% between 2009 and 2018. The years of potential labour productive life lost (YPLPLL) decreased by 38.1%. That reduction led to a decrease in annual labour productivity losses from €236 million in 2009 to €156 million in 2018 (-33.8%). The aggregate HCV-related labour productivity losses between 2009 and 2018 ranged from €1742 million (optimistic scenario) to €1949 million (pessimistic scenario), with an intermediate estimation of €1846 million (moderately optimistic scenario). These results show a substantial reduction in annual deaths, working-age deaths, YPLPLL, and labour productivity losses associated with HCV infection over this period.

BackgroundThe preferences of citizens are a basic element to incorporate into the decision-making process when planning health policies. Contingent valuation (CV) is a common method for calculating the value for citizens that new technologies, interventions, and the provision of services or policies have. However, choosing the correct CV tool may not be a neutral decision. This work aims to assess the substitution of a healthcare service by comparing valuation differences between the willingness to pay (WTP) for the maintenance of the service versus the willingness to accept compensation (WTA) for its substitution, both of which are related to subject characteristics, with a particular focus on trust in institutions and risk aversion.MethodsA CV study was designed to study Dutch population preferences when physician assistants replace anaesthesiologists. Differences between the distributions of WTA and WTP were compared through full decomposition methods, and conditional quantile regression was performed.ResultsNearly two-thirds of surveyed citizens expressed null values for WTA and WTP. The other third systematically reported a value of WTA higher than that of WTP, which increased further with lower income and the possible presence of a strategic bias. In contrast, being more than 65 years old, having trust in government, and preferring anaesthesiologists decreased the WTA-WTP difference. Risk aversion had no clear association with the WTA-WTP gap.ConclusionsKnown differences between the perceived value of health services from the perspective of gains and losses could be related to people’s characteristics. Trust in government but not aversion to risk was related to the WTA-WTP differences. Identifying a profile of citizens who are averse to losing health services should be considered when designing and implementing health services or interventions or making disinvestment decisions.

Spinal muscular atrophy (SMA) is one of the most common severe hereditary diseases of infancy and early childhood. The progression of this illness causes a high degree of disability, hence, a significant burden is experienced by individuals with this disease and their families. We analyzed the time taken to care for patients suffering from SMA in European countries and the burden on their informal caregivers. We designed a cross-sectional study recording data from France, Germany, Spain and the United Kingdom. The primary caregivers completed a self-administered questionnaire that included questions about the time of care, The Zarit Burden Interview, type of SMA and socio-demographic characteristics. Multivariate analyses were used to study the associations between the type of SMA, time of care and burden supported by informal caregivers. The caregivers provided 10.0 h (SD = 6.7) per day of care (the principal caregivers provided 6.9 h, SD = 4.6). The informal caregivers of patients with type I SMA had a 36.3 point higher likelihood (p &lt, 0.05) of providing more than 10 hours of care per day in comparison with caregivers of patients with type III SMA. The severity of the disease was associated with more time of care and a higher burden on the caregivers.

The aim of this study was to estimate the incremental cost-utility ratio (ICUR) of a multi-modal intervention in frail and pre-frail subjects aged ≥70 years with type-2 diabetes versus usual care g...

Pomacea flagellata is a gastropod conspicuous in freshwater environments, and represents a fishing resource. To assess their abundance, distribution, and secondary production, monthly samplings wer...

Background and Purpose— The aim of this article was to analyze the likelihood of receiving informal care after a stroke and to study the burden and risk of burnout of primary caregivers in Spain. Methods— The CONOCES study is an epidemiological, observational, prospective, multicenter study of patients diagnosed with stroke and admitted to a Stroke Unit in the Spanish healthcare system. At 3 and 12 months post-event, we estimated the time spent caring for the patient and the burden borne by primary caregivers. Several multivariate models were applied to estimate the likelihood of receiving informal caregiving, the burden, and the likelihood of caregivers being at a high risk of burnout. Results— Eighty percent of those still alive at 3 and 12 months poststroke were receiving informal care. More than 40% of those receiving care needed a secondary caregiver at 3 months poststroke. The likelihood of receiving informal care was associated with stroke severity and the individual’s health-related quality of life. When informal care was provided, both the burden borne by caregivers and the likelihood of caregivers being at a high risk of burnout was associated with (1) caregiving hours; (2) the patient’s health-related quality of life; (3) the severity of the stroke measured at discharge; (4) the patient having atrial fibrillation; and (5) the degree of dependence. Conclusions— This study reveals the heavy burden borne by the caregivers of stroke survivors. Our analysis also identifies explanatory and predictive variables for the likelihood of receiving informal care, caregiver burden, and high risk of burnout.

AIMS: To estimate the social-economic costs of Type 1 Diabetes Mellitus (T1DM) in patients aged 0-17 years in Spain from a social perspective. METHODS: We conducted a cross-sectional observational study in 2014 of 275 T1DM pediatric outpatients distributed across 12 public health centers in Spain. Data on demographic and clinical characteristics, healthcare utilization and informal care were collected from medical records and questionnaires completed by clinicians and patients' caregivers. RESULTS: A valid sample of 249 individuals was analyzed. The average annual cost for a T1DM patient was €27,274. Direct healthcare costs were €4070 and direct non-healthcare cost were €23,204. Informal (familial) care represented 83% of total cost, followed by medical material (8%), outpatient and primary care visits (3.1%) and insulin (2.1%). Direct healthcare cost per patient statistically differed by glycated haemoglobin (HbA1c) level [mean cost €4704 in HbA1c ?7.5% (?58mmol/mol) group vs. €3616 in HbA1c

Objectives: To analyze whether the adoption of a societal perspective would alter the results and conclusions of economic evaluations for rare disease–related healthcare technologies. Methods: A search strategy involving all the active substances considered as orphan drugs by the European Medicines Agency plus a list of 76 rare diseases combined with economic-related terms was conducted on Medline and the Cost-Effectiveness Registry from the beginning of 2000 until November 2018. We included studies that considered quality-adjusted life years as an outcome, were published in a scientific journal, were written in English, included informal care costs or productivity losses, and separated the results according to the applied perspective. Results: We found 14 articles that fulfilled the inclusion criteria. Productivity losses were considered in 12 studies, the human capital approach being the method most frequently used. Exclusively, informal care was considered in 2 articles, being valued through the opportunity cost method. The 14 articles selected resulted in 26 economic evaluation estimations, from which incremental cost-utility ratio values changed from cost-effective to dominant in 3 estimates, but the consideration of societal costs only modified the authors’ conclusion in 1 study. Conclusions: The presence of societal costs in the economic evaluation of rare diseases did not affect the conclusions of the studies except in a single specific case. In those studies where the societal perspective was considered, we did not find significant changes in the economic evaluation results due to the higher costs of treatments and the low quality-adjusted life years gained.

Background A large part of the long-term care is provided by non-professional caregivers, generally without any monetary payment but a value economic of time invested. The economic relevance of informal caregivers has been recognized in Spain; however, public provision may still be scarce. The objective of this paper is to estimate the economic burden associated with informal long-term care that should assume the families through a new concept of cost sharing that consider opportunity costs of time provided by informal caregivers. Methods The study sample includes all dependent adults in Spain. Socioeconomic information and the number of hours of informal care was collected through the Spanish Disability and Dependency Survey. The terms of shadow and extended shadow cost sharing were defined as the difference between the maximum potential amount of money that families could receive for the provision of informal care and the amount that actually they received and the value of informal care time with respect to the amount received, respectively. Results 53.87% of dependent persons received an economic benefit associated to informal care. The average weekly hours of care were 71.59 (92.62 without time restrictions). Shadow cost sharing amounted to, on average, two thirds, whereas the State financed the remaining third. In terms of extended shadow cost sharing, the State financed between 3% and 10% of informal care provided by caregivers. Conclusions This study reveals the deficient support received for the provision of informal care in Spain. More than 90% of informal care time is not covered by the economic benefits that families receive from the State.

Although the choice of the comparator is one of the aspects with a highest effect on the results of cost-effectiveness analyses, it is one of the less debated issues in international methodological guidelines. The inclusion of an inappropriate comparator may introduce biases on the outcomes and the recommendations of an economic analysis. Although the rules for cost-effectiveness analyses of sets of mutually exclusive alternatives have been widely described in the literature, in practice, they are hardly ever applied. In addition, there are many cases where the efficiency of the standard of care has never been assessed; or where the standard of care has demonstrated to be cost-effective with respect to a non-efficient option. In all these cases the comparator may lie outside the efficiency frontier, so the result of the CEA may be biased. Through some hypothetical examples, the paper shows how the complementary use of an independent reference may help to identify potential inappropriate comparators and inefficient use of resources.

OBJECTIVE To analyze the differences in health care costs according to glycemic control in people with type 2 diabetes. RESEARCH DESIGN AND METHODS Data on health care resource utilization from 100,391 people with type 2 diabetes were extracted from the electronic database used at the Catalan Health Institute. Multivariate regression models were carried out to test the impact of glycemic control (HbA1c) on total health care, hospital admission, and medication costs; model 1 adjusted for a variety of covariates, and model 2 also included micro- and macrovascular complications. Glycemic control was classified as good for HbA1c RESULTS Mean per patient annual direct medical costs were €3,039 ± SD €6,581. Worse glycemic control was associated with higher total health care costs: compared with good glycemic control, health care costs increased by 18% (€509.82) and 23% (€661.35) in patients with very poor and poor glycemic control, respectively, when unadjusted and by €428.3 and €395.1, respectively, in model 2. Medication costs increased by 12% in patients with fair control and by 28% in those with very poor control (model 2). Patients with poor control had a higher probability of hospitalization than those with good control (5% in model 2) and a greater average cost when hospitalization occurred (€811). CONCLUSIONS Poor glycemic control was directly related to higher total health care, hospitalization, and medication costs. Preventive strategies and good glycemic control in people with type 2 diabetes could reduce the economic impact associated with this disease.

A correction to this paper has been published: https://doi.org/10.1007/s10198-021-01323-y

Stroke is a major social and health problem. However, since the recent incorporation of new advances in its management, little is known about the cost of stroke. The aim of this study is to find out the real cost of stroke in Spain. This is an epidemiological, observational, prospective, multicenter study of patients diagnosed with stroke and admitted to a stroke unit. Patients were recruited from 16 hospitals throughout Spain and followed up for 1 year. Sociodemographic, clinical, and economic data were collected. Costs (€ 2012) were estimated from the social perspective and were divided into direct healthcare (inpatient, outpatient, and medication), direct non-healthcare (mainly formal and informal care) and labor productivity losses. A total of 321 patients were included. Mean age was 72.1 years and 176 patients (54.8 %) were male. Total average cost per patient/year was €27,711. Direct healthcare costs amounted to €8491 per patient/year (68.8 % due to inpatient costs) and non-healthcare costs to an average of €18,643 per patient/year (89.5 % due to informal care). Productivity loss costs per patient/year were €276. Total costs of hemorrhagic strokes were slightly higher than ischemic (€28,895 vs. €27,569 per patient/year, p = 0.550) without significant differences. The main variables associated with higher costs were the presence of hypertension (€30,332 vs. €23,234 per patient/year, p

The aim of this study was to determine the economic burden from a societal perspective and health-related quality of life (HRQOL) of patients with Prader-Willi syndrome (PWS) in Europe. We conducted a cross-sectional study of patients with PWS from Spain, Bulgaria, Hungary, Germany, Italy, the UK, Sweden and France. Data on demographic characteristics, healthcare resource utilisation, informal care, labour productivity losses and HRQOL were collected from questionnaires completed by patients or their caregivers. HRQOL was measured with the EuroQol 5-domain (EQ-5D) questionnaire. A total of 261 patients completed the questionnaire. The average annual costs ranged from € 3937 to € 67,484 between countries; the reference year for unit prices was 2012. Direct healthcare costs ranged from € 311 to € 18,760, direct non-healthcare costs ranged from € 1269 to € 44,035, and loss of labour productivity ranged from € 0 to € 2255. Costs were also shown to differ between children and adults. The mean EQ-5D index score for adult PWS patients ranged between 0.40 and 0.81 and the mean EQ-5D visual analogue scale score ranged between 51.25 and 90.00. The main strengths of this study lie in our bottom-up approach to costing and in the evaluation of PWS patients from a broad societal perspective. This type of analysis is very scarce in the international literature on rare diseases in comparison with other illnesses. We conclude that PWS patients incur considerable societal costs and experience substantial deterioration in HRQOL.

The aim of this study was to determine the economic burden from a societal perspective and the health-related quality of life (HRQOL) of patients with juvenile idiopathic arthritis (JIA) in Europe. We conducted a cross-sectional study of patients with JIA from Germany, Italy, Spain, France, the United Kingdom, Bulgaria, and Sweden. Data on demographic characteristics, healthcare resource utilization, informal care, labor productivity losses, and HRQOL were collected from the questionnaires completed by patients or their caregivers. HRQOL was measured with the EuroQol 5-domain (EQ-5D-5L) questionnaire. A total of 162 patients (67 Germany, 34 Sweden, 33 Italy, 23 United Kingdom, 4 France, and 1 Bulgaria) completed the questionnaire. Excluding Bulgarian results, due to small sample size, country-specific annual health care costs ranged from €18,913 to €36,396 (reference year: 2012). Estimated direct healthcare costs ranged from €11,068 to €22,138; direct non-healthcare costs ranged from €7837 to €14,155 and labor productivity losses ranged from €0 to €8715. Costs are also shown to differ between children and adults. The mean EQ-5D index score for JIA patients was estimated at between 0.44 and 0.88, and the mean EQ-5D visual analogue scale score was estimated at between 62 and 79. JIA patients incur considerable societal costs and experience substantial deterioration in HRQOL in some countries. Compared with previous studies, our results show a remarkable increase in annual healthcare costs for JIA patients. Reasons for the increase are the inclusion of non-professional caregiver costs, a wider use of biologics, and longer hospital stays.

To estimate the social/economic costs of fragile X syndrome (FXS) in Europe and to assess the health-related quality of life (HRQOL) of patients and caregivers. A cross-sectional study was conducted in a sample of European countries. Patients were recruited through patients’ associations. Data on their resource use and absence from the labour market were retrospectively obtained from an online questionnaire. Costs were estimated by a bottom-up approach and the EuroQol-5 Domain (EQ-5D) questionnaire was used to measure patients’ and caregivers’ HRQOL. Five countries were included in the analysis. The mean annual cost of FXS per patient varied from €4951 in Hungary to €58,862 in Sweden. Direct non-healthcare costs represented the majority of costs in all countries but there were differences in the share incurred by formal and informal care among those costs. Costs were also shown to differ between children and adults. Mean EQ-5D utility score for adult patients varied from 0.52 in France (n = 42) to 0.73 in Hungary (n = 2), while for caregivers this score was consistently inferior to 0.87. Our findings underline that, although its prevalence is low, FXS is costly from a societal perspective. They support the development of tailored policies to reduce the consequences of FXS on both patients and their relatives.

The aim of this study was to determine the economic burden from a societal perspective and the health-related quality of life (HRQOL) of patients with systemic sclerosis (SSc; scleroderma) in Europe. We conducted a cross-sectional study of patients with SSc (involving both localised and systemic sclerosis) from Germany, Italy, Spain, France, the UK, Hungary and Sweden. Data on demographic characteristics, healthcare resource utilisation, informal care, labour productivity losses and HRQOL were collected from the questionnaires completed by patients or their caregivers. HRQOL was measured with the EuroQol 5-domain (EQ-5D) questionnaire. A total of 589 patients completed the questionnaire. The rate of patients with localised scleroderma, limited cutan and diffuse cutan SSc were 28, 68 and 4 %, respectively. Average annual costs varied from country to country and ranged from € 4607 to € 30,797 (reference year: 2012). Estimated direct healthcare costs ranged from € 1413 to € 17,300; direct non-healthcare costs ranged from € 1875 to € 4684 and labour productivity losses ranged from € 1701 to € 14,444. The mean EQ-5D index score for adult SSc patients varied from 0.49 to 0.75 and the mean EQ-5D visual analogue scale score was between 58.72 and 65.86. The main strengths of this study lie in our bottom-up approach to costing and our evaluation of SSs patients from a broad societal perspective. This type of analysis is very unusual in the international literature on rare diseases in comparison with other illnesses. We concluded that SSc patients incur considerable societal costs and experience substantial deterioration in HRQOL.

Background/objectivesTo analyze mortality, costs, residents and personnel characteristics, in six long-term care facilities (LTCF) during the outbreak of COVID-19 in Spain.DesignEpidemiological study.SettingSix open LTCFs in Albacete (Spain).Participants198 residents and 190 workers from LTCF A were included, between 2020 March 6 and April 5. Epidemiological data were also collected from six LTCFs of Albacete for the same period of time, including 1,084 residents.MeasurementsBaseline demographic, clinical, functional, cognitive and nutritional variables were collected. 1-month and 3-month mortality was determined, excess mortality was calculated, and costs associated with the pandemics were analyzed.ResultsThe pooled mortality rate for the first month and first three months of the outbreak were 15.3% and 28.0%, and the pooled excess mortality for these periods were 564% and 315% respectively. In facility A, the percentage of probable COVID-19 infected residents were 33.6%. Probable infected patients were older, frail, and with a worse functional situation than those without COVID-19. The most common symptoms were fever, cough and dyspnea. 25 residents were transferred to the emergency department, 21 were hospitalized, and 54 were moved to the facility medical unit. Mortality was higher upon male older residents, with worse functionality, and higher comorbidity. During the first month of the outbreak, 65 (24.6%) workers leaved, mainly with COVID-19 symptoms, and 69 new workers were contracted. The mean number of days of leave was 19.2. Costs associated with the COVID-19 in facility A were estimated at € 276,281/month, mostly caused by resident hospitalizations, leaves of workers, staff replacement, and interventions of healthcare professionals.ConclusionThe COVID-19 pandemic posed residents at high mortality risk, mainly in those older, frail and with worse functional status. Personal and economic costs were high.

espanolLa reciente crisis economica vivida en Espana, pese al sufrimiento y perdida de bienestar ocasionadas, no parece haber afectado de una manera severa, al menos a corto plazo, a la mortalidad y la salud autopercibida de la poblacion general. No obstante, si se ha producido un impacto negativo sobre la salud mental, especialmente entre los varones. Asimismo, hay grupos vulnerables de poblacion cuya salud ha empeorado durante este periodo, intensificandose las desigualdades sociales en la misma. Dada la alarmante evolucion observada en los indicadores sociales de desigualdad de rentas y tasas de pobreza y riesgo de exclusion social, conviene ser cautos con los resultados identificados en el corto plazo y vigilantes sobre la evolucion de la salud de la poblacion en el medio y largo plazo, a la vez que se extraen las conclusiones oportunas sobre las politicas e intervenciones a implementar en futuras situaciones de crisis. EnglishEven though the considerable loss of welfare caused by the recent economic crisis occurred in Spain, it does not seem to have affected heavily, at least in the short term, in terms of mortality and self-perceived health of the general population. However, there has been a negative impact on mental health, especially among men. Likewise, there are several vulnerable population groups whose health has worsened during this period, intensifying social inequalities in health. Given such disturbing evolution observed in the social indicators of income inequality and poverty rates as well as risk of social exclusion, caution is needed when interpreting the results obtained in the short term and attention should be paid on the population’s health evolution in the medium and long term. At the same time, it is necessary to draw the appropriate conclusions so that the policies and interventions could be implemented in future crises.

Background The main objective of this study was to analyse how the inclusion (exclusion) of social costs can alter the results and conclusions of economic evaluations in the field of Alzheimer’s disease interventions. Methods We designed a systematic review that included economic evaluations in Alzheimer’s disease. The search strategy was launched in 2000 and ran until November 2018. The inclusion criteria were: being an original study published in a scientific journal, being an economic evaluation of any intervention related to Alzheimer’s disease, including social costs (informal care costs and/or productivity losses), being written in English, using QALYs as an outcome for the incremental cost–utility analysis, and separating the results according to the perspective applied. Results It was finally included 27 studies and 55 economic evaluations. Around 11% of economic evaluations changed their main conclusions. More precisely, three of them concluded that the new intervention became cost-effective when the societal perspective was considered, whereas when using just the health care payer perspective, the new intervention did not result in a cost–utility ratio below the threshold considered. Nevertheless, the inclusion of social cost can also influence the results, as 37% of the economic evaluations included became the dominant strategy after including social costs when they were already cost-effective in the health care perspective. Conclusions Social costs can substantially modify the results of the economic evaluations. Therefore, taking into account social costs in diseases such as Alzheimer’s can be a key element in making decisions about public financing and pricing of health interventions.

Fifteen years ago, Gaceta Sanitaria published the article entitled "What is an efficient health technology in Spain?" The growing interest in setting the price of new technologies based on the value they provide to health systems and the experience accumulated by the countries in our environment make it opportune to review what constitutes an efficient health intervention in Spain in 2020. Cost-effectiveness analysis continues to be the reference method to maximize social health outcomes with the available resources. The interpretation of its results requires establishing reference values that serve as a guide on what constitutes a reasonable value for the health care system. Efficiency thresholds must be flexible and dynamic, and they need to be updated periodically. Its application should be based on and transparency, and consider other factors that reflect social preferences. Although setting thresholds is down to political decision-makers, in Spain it could be reasonable to use thresholds of 25,000 and 60,000 Euros per QALY. However, currently, in addition to determining exact figures for the threshold, the key question is whether the Spanish National Health System is able and willing to implement a payment model based on value, towards achieving gradual financing decisions and, above all, to improve the predictability, consistency and transparency of the process.

Background The economic evaluation of healthcare technologies has become in many countries a basic tool for reimbursement, pricing and purchasing decisions. Objective The objective of this article is to examine the institutional, legal, and political factors that have impeded the application of economic evaluation and the criterion of efficiency in the process of pricing and reimbursement of new medicines in Spain. Methods Narrative description of the current institutional framework for the use of economic evaluation in pricing and reimbursement in Spain, legal and policy framework in the field of evaluation of new medicines, and stakeholder initiatives and policies related to the use of economic evaluation outside of the pricing and reimbursement process. Results Spain has an institutional framework created and established over the last years that could have facilitated a formal use of economic evaluation in the process of pricing and reimbursement. Nevertheless, the real use of economic evaluation at the central or regional level is still unknown, although application of the efficiency criterion, linking to cost-effectiveness, has been clearly required by Spanish laws and regulations at the national level. We highlight a certain degree of moral hazard from the central government that is not directly responsible for the budget impact of reimbursement and pricing decisions. There are currently a number of ongoing initiatives in the field of economic evaluation by various agents, but they remain uncoordinated. Conclusions Poor governance at the highest level of decision making is the main reason for the lack of interest in economic evaluation. A profound political change, supported by transparency and accountability, is required before the criterion of efficiency can be fully considered in the process of pricing and reimbursement of new medicines in Spain.

This analysis of the Spanish health system reviews recent developments in organization and governance, health financing, health care provision, health reforms and health system performance. Overall health status continues to improve in Spain, and life expectancy is the highest in the European Union. Inequalities in self-reported health have also declined in the last decade, although long-standing disability and chronic conditions are increasing due to an ageing population. The macroeconomic context in the last decade in the country has been characterized by the global economic recession, which resulted in the implementation of health system-specific measures addressed to maintain the sustainability of the system. New legislation was issued to regulate coverage conditions, the benefits package and the participation of patients in the National Health System funding. Despite the budget constraints linked to the economic downturn, the health system remains almost universal, covering 99.1% of the population. Public expenditure in health prevails, with public sources accounting for over 71.1% of total health financing. General taxes are the main source of public funds, with regions (known as Autonomous Communities) managing most of those public health resources. Private spending, mainly related to out-of-pocket payments, has increased over time, and it is now above the EU average. Health care provision continues to be characterized by the strength of primary care, which is the core element of the health system; however, the increasing financing gap as compared with secondary care may challenge primary care in the long-term. Public health efforts over the last decade have focused on increasing health system coordination and providing guidance on addressing chronic conditions and lifestyle factors such as obesity. The underlying principles and goals of the national health system continue to focus on universality, free access, equity and fairness of financing. The evolution of performance measures over the last decade shows the resilience of the health system in the aftermath of the economic crisis, although some structural reforms may be required to improve chronic care management and the reallocation of resources to high-value interventions.

Breast cancer is one of the most common cancers among women worldwide, and is the most common cause of cancer deaths in them. Given that such disease has a significant impact on middle-aged women, it can result in considerable labour productivity losses. The main objective of this study is therefore to analyse labour productivity losses and years of potential working life lost as a result of premature deaths from breast cancer in Spain over a 10-year period (2005–2014). To this end, by combining data on average salary (Salary Structure Survey), occupation (Active Population Survey) and deaths (death registration according to cause of death), a simulation model based on the human capital approach was constructed to estimate productivity losses associated with premature deaths caused by this disease. The results reveal that labour productivity losses in women are estimated to cost 2,137 million euros, as a result of the 22,716 deaths occurring during working age and the 292,848 years of potential productive life lost. This implies that breast cancer is responsible for 10.45% of all estimated female labour productivity losses caused by premature deaths in the reference year. The article concludes by highlighting the major economic impact associated with breast cancer deaths and by pointing out future research areas.

Primary biliary cirrhosis (PBC) is associated to any systemic autoimmune disease (SAD), in particular systemic sclerosis (SSc). To investigate the prevalence of SAD in a cohort of patients with PBC, specifically the prevalence of SSc and its clinical subtypes, and determining the clinical and biological profile of patients with associated PBC and SSc.Observational study of 62 patients with PBC following a protocol that included an anamnesis and physical examination to detect the presence of SAD as well as a nailfold capillaroscopy and an immunological study with specific SSc autoantibodies. A comparative analysis was conducted between patients with isolated PBC and patients with PBC and an associated SAD.SAD was associated to PBC in 22 patients (35,4%), and SSc was the most frequent illness, identified in 13 cases (21%). Five patients (8%) without previous diagnosis of SAD fulfilled pre-scleroderma criteria, according to LeRoy and Medsger criteria. The presence of anticentromere antibodies (54,5% vs. 5%, P.001) was the unique immunological determination identified more frequently in patients with PBC-SAD. The SSc suggestive capillary pattern was visualized in 11 patients (20,4%), mainly the slow pattern. No factors associated with greater morbi-mortality were identified in the PBC-SAD group.It does exist a subgroup of patients with PBC and clinical-biological features suggestive of an SAD, which advise a protocolized study to detect early the association to an SAD.

Resumen Introduccion y objetivos La cirrosis biliar primaria (CBP) se asocia a algunas enfermedades autoinmunes sistemicas (EAS), en particular a la esclerosis sistemica (ES. Determinar la prevalencia de EAS en una cohorte de pacientes con CBP, especificamente la ES y sus diferentes subtipos clinicos, y establecer el perfil clinico-biologico propio de estos pacientes. Metodos Estudio observacional de 62 pacientes con CBP, con un protocolo que incluia una anamnesis y exploracion fisica dirigidas a detectar una EAS, la realizacion de una capilaroscopia ungueal microscopica y un amplio estudio de autoinmunidad, incluido el perfil de anticuerpos especificos de ES. Se realizo un analisis comparativo entre el grupo de pacientes con CBP aislada y los pacientes con CBP y una EAS asociada. Resultados Se asocio una EAS en 22 pacientes (35,4%), y la ES fue la entidad mas frecuente (21%), del subtipo cutaneo limitado (11%). Cinco pacientes (8%) sin EAS previa cumplian criterios de preesclerodermia, segun los criterios de LeRoy y Medsger. Los anticuerpos anticentromero (54,5 vs. 5%, p Conclusiones Existe un subgrupo de pacientes con CBP con caracteristicas clinico-biologicas que sugieren la asociacion con una EAS, con elevada probabilidad, y que recomiendan el estudio protocolizado de estos pacientes con CBP para detectar de forma precoz EAS.

Background Fragile X syndrome (FXS) is the main hereditary cause of intellectual disability. Although the associated burden appears to be considerable, to date no study has comprehensively assessed the cost incurred because of FXS, including its specific impact on health-related quality of life and the burden on caregivers using standardised quantitative tools. The aim of this article is to provide data in order to increase awareness of the repercussions of FXS on patients and caregivers as well as on the health and social care systems in France. Methods A retrospective cross-sectional study was carried out on 145 patients recruited through Le Goeland X-Fragile and Mosaiques, the French FXS patient associations. Data on their demographic characteristics and resource use were obtained from an online questionnaire, and costs were estimated by a bottom-up approach. The EQ-5D health questionnaire was used to measure patients' and caregivers' health-related quality of life. Perceived burden of care was measured using the Zarit Caregiver Burden Interview. The Barthel index, a non-utility-based assessment, was used to measure patients' level of dependence. Results The annual total direct cost of FXS was estimated at €25 800 per patient. The main contributors were informal care provided by the main caregiver (€10 500) and social services (€8400). Healthcare costs, estimated at €2700, represented only a minor share. Mean EQ-5D utility scores were 0.49 for patients and 0.75 for caregivers. The mean burden for caregivers as measured by the Zarit Caregiver Burden Interview was 39.9. Conclusions Fragile X syndrome requires significant resources that are mainly of a non-medical nature and are higher for children than for adults. Compared with related diseases, it constitutes a particularly high burden for caregivers. Using a bottom-up approach and a wide range of standardised measures, this study underscores the need for greater awareness of the burden of FXS as well as an assessment of new and existing interventions to address it.

The aim of this paper was to arrive at an approximation of the value of non-professional (informal) care provided to disabled people living within a household in Spain.We used the Survey on Disabilities, Autonomy and Dependency carried out in 2008 to obtain information about disabled individuals and their informal caregivers. We computed the total number of informal caregiving hours provided by main caregivers in Spain in 2008. The monetary value of informal care time was obtained using three different approaches: the proxy good method, the opportunity cost method and the contingent valuation method.Total hours of informal care provided in 2008 were estimated at 4193 million and the monetary value ranged from EUR23,064 to EUR50,158 million depending on the method used. The value of informal care was estimated at figures equivalent to 1.73-4.90 % of the gross domestic product for that year.Informal care represents a very high social cost regardless of the estimation method considered. A holistic approach to care of dependent people should take into account the role and needs of informal caregivers, promote their social recognition and lead to policies that enhance efficient use of formal and informal resources.

The European crisis has highlighted the role of intra-European payments imbalances for the survival of the EMU. Payment imbalances between the North and the South have contributed to the accumulation of large stock of foreign debt, while flows of foreign capital ceased to finance productive investments which might have contributed to debt repayments—preferring instead to finance consumption and a housing bubble. The dynamic interplay between current account imbalances and the accumulation of debt reveals that, once the system is driven into disequilibrium by a real exchange rate misalignment, the longer a payments imbalance persists the harder the eventual adjustment will be. Capital reversals, by shifting portfolio balances, lead the system toward instability, sovereign default, and the collapse of the exchange rate regime. Replacing private with public creditors may temporarily help us to stay away from the point where the system breaks down. But this is only a temporary expedient because the underlying imbalances need continued and escalating financing until equilibrium is restored by other means. One permanent solution is the ECB's official monetary transactions program, if the potential expansions to the central bank's balance sheet can be tolerated.

In the field of the economic evaluation (EE), there is an open debate on the most appropriate perspective to apply. Despite the relevance of the topic, there are hardly any studies that have analyzed the practical consequences of applying the social perspective vs. health care funder. The aim of this study was performed a systematic review of the available evidence to analyze whether the inclusion/exclusion of informal care and/or loss of productivity influence the results and conclusions of economic evaluations.A systematic review of the literature was carried out using PubMed and Econlit databases, to determine the publications that contained EE on Alzheimer, Parkinson and Stroke. Inclusion criteria were: a) full EE of the diseases under study; b) from January 2006-July 2016; and c) in English or Spanish.6,292 references were identified, of which 245 publications meet the selection criteria and were analyzed. After the full-text analysis, 20 references were selected, of which 27 full EE were obtained (20 corresponding to Alzheimer, 1 to Parkinson and 6 to Stroke). In 20 EE (74.1%) the change of the perspective modifies the results, becoming dominant or increasing the saving of the resources by comparing the alternatives from the social perspective. In the remaining 7, the inclusion of social costs involves an increase in the costs of the intervention evaluated against its control. In a single EE, the conclusions vary when including social costs.The inclusion of social costs can modify the results of EE but their impact on the conclusions is not relevant.En el campo de la evaluación económica (EE), existe un debate abierto sobre la perspectiva más apropiada a aplicar. Pese a la relevancia del tema, apenas hay trabajos que analicen las consecuencias prácticas de aplicar la perspectiva social o la del financiador. El objetivo fue realizar una revisión sistemática para analizar si la inclusión/exclusión de los cuidados informales y/o pérdidas laborales influyen en los resultados y conclusiones de las EE.Se realizó una búsqueda sistemática en PubMed y Econlit para determinar las publicaciones que contenían EE sobre Alzheimer, Parkinson e Ictus. Criterios de inclusión: a) EE completas sobre las enfermedades en estudio; b) periodo enero 2006-julio 2016; y c) en inglés o español.Se obtuvieron 6.292 referencias, seleccionándose 245 artículos que podían cumplir los criterios de inclusión. Tras el análisis a texto completo, se seleccionaron 20 publicaciones de las cuales se obtuvieron 27 EE completas (20 corresponden a Alzheimer, 1 a Parkinson y 6 al Ictus). En 20 EE (74,1%) el cambio de perspectiva modifica los resultados, pasando a ser dominantes o incrementando el ahorro de recursos comparando las alternativas desde la perspectiva social. En las 7 restantes, la inclusión de los costes sociales supone un incremento en los costes de la intervención evaluada frente a su control. Únicamente en una EE varían las conclusiones al incluir los costes sociales.En general, la inclusión de los costes sociales puede modificar los resultados de las EE, pero su impacto sobre las conclusiones de las mismas es poco relevante.

Background: Alzheimer’s disease constitutes one of the leading causes of burden of disease, and it is the third leading disease in terms of economic and social costs. Objective: To analyze the burden and problems borne by informal caregivers of patients who suffer from Alzheimer’s disease in Spain. Data and Methods: We used the Survey on Disabilities, Autonomy and Dependency to obtain information on the characteristics of disabled people with Alzheimer’s disease and the individuals who provide them with personal care. Additionally, statistical multivariate analyses using probit models were performed to analyze the burden placed on caregivers in terms of health, professional, and leisure/social aspects. Results: 46% of informal caregivers suffered from health-related problems as a result of providing care, 90% had leisure-related problems, and 75% of caregivers under 65 years old admitted to suffering from problems related to their professional lives. The probability of a problem arising for an informal caregiver was positively associated with the degree of dependency of the person cared for. In the case of caring for a greatly dependent person, the probability of suffering from health-related problems was 22% higher, the probability of professional problems was 18% higher, and there was a 10% greater probability of suffering from leisure-related problems compared to non-dependents. Conclusions: The results show a part of the large hidden cost for society in terms of problems related to the burden lessened by the caregivers. This information should be a useful tool for designing policies focused toward supporting caregivers and improving their welfare.