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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.

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.

Purpose To determine the impact of non-cardiovascular comorbidities on the health-related quality of life (HRQoL) of patients with chronic heart failure (CHF). Methods A scoping review of the scientific literature published between 2009 and 2019 was carried out. Observational studies which assessed the HRQoL of patients with CHF using validated questionnaires and its association with non-cardiovascular comorbidities were included. Results The search identified 1904 studies, of which 21 fulfilled the inclusion criteria to be included for analysis. HRQoL was measured through specific, generic, or both types of questionnaires in 72.2%, 16.7%, and 11.1% of the studies, respectively. The most common comorbidities studied were diabetes mellitus (12 studies), mental and behavioral disorders (8 studies), anemia and/or iron deficiency (7 studies), and respiratory diseases (6 studies). Across studies, 93 possible associations between non-cardiovascular comorbidities and HRQoL were tested, of which 21.5% regarded anemia or iron deficiency, 20.4% mental and behavioral disorders, 20.4% diabetes mellitus, and 14.0% respiratory diseases. Despite the large heterogeneity across studies, all 21 showed that the presence of a non-cardiovascular comorbidity had a negative impact on the HRQoL of patients with CHF. A statistically significant impact on worse HRQoL was found in 84.2% of associations between mental and behavioral disorders and HRQoL (patients with depression had up to 200% worse HRQoL than patients without depression); 73.7% of associations between diabetes mellitus and HRQoL (patients with diabetes mellitus had up to 21.8% worse HRQoL than patients without diabetes mellitus); 75% of associations between anemia and/or iron deficiency and HRQoL (patients with anemia and/or iron deficiency had up to 25.6% worse HRQoL than between patients without anemia and/or iron deficiency); and 61.5% of associations between respiratory diseases and HRQoL (patients with a respiratory disease had up to 21.3% worse HRQoL than patients without a respiratory disease). Conclusion The comprehensive management of patients with CHF should include the management of comorbidities which have been associated with a worse HRQoL, with special emphasis on anemia and iron deficiency, mental and behavioral disorders, diabetes mellitus, and respiratory diseases. An adequate control of these comorbidities may have a positive impact on the HRQoL of patients.

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.

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.

(1) Background: The aim of this study was (i) to analyze problems faced by informal caregivers in three areas of their life: health, work and finances, and family and social relationships, (ii) to investigate the main determinants of these problems, and (iii) to explore differences between men and women. (2) Methods: The study population consisted of people aged ≥18 years living in a family home who were providing unpaid care to a dependent person in the same or another home and who were registered as caregivers with the Primary Health Care District of Granada or the Provincial Council of Gipuzkoa. Several logistic regression models were built to analyze the likelihood of caregivers experiencing health, work-related, or social problems as a result of their caregiving responsibilities. (3) Results: Informal female caregivers were more likely to experience problems attributed to caregiving than their male counterparts, particularly in the areas of health and work. Additional factors associated with an increased likelihood of problems were low perceived social support, performance of ungratifying tasks, and fewer years as a caregiver. (4) Conclusions: Informal caregivers in Spain face significant problems as a result of their caregiving duties, and the impact on men and women is different. Policies and interventions to mitigate the negative effects of unpaid caregiving should incorporate differential strategies to meet the specific needs of male and female caregivers in different caregiving contexts.

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.

Background: this study aimed to estimate the economic impact and health-related quality of life (HRQOL) of patients with spinal muscular atrophy (SMA) in three European countries. It was used a cross-sectional study carried out in France, Germany, and the United Kingdom. Data were collected from July 2015 to November 2015. Healthcare costs (hospitalizations, emergencies, medical tests, drugs used, visits to general practitioners (GPs) and specialists, medical material and healthcare transport), and non-healthcare costs (social services and informal care) were identified and valued. EuroQol instruments, the Zarit interview, and the Barthel Index were also used to reflect the burden and the social impact of the disease beyond the cost of healthcare. Results: we included 86 children with SMA, 26.7% of them had Type I, and 73.3% Type II or III. The annual average cost associated with SMA reaches &euro, 54,295 in the UK, &euro, 32,042 in France and &euro, 51,983 in Germany. The direct non-healthcare costs ranged between 79&ndash, 86% of the total cost and the informal care costs were the main component of these costs. Additionally, people suffering from this disease have a very low health-related quality of life, and there are large differences between countries. Conclusions: SMA has a high socioeconomic impact in terms of healthcare and social costs. It was also observed that the HRQOL of affected children was extremely reduced. The figures shown in this study may help to design more efficient and equitable policies, with special emphasis on the support provided to the families or on non-healthcare aid.

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...

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.

The aim of this study conducted in Spain was to analyze and compare burden, severe burden, and satisfaction among informal caregivers in relation to health-related quality of life (HRQoL), type and duration of caregiving, perceived social support, and use of social and health care services. We performed multivariate analyses to identify variables associated with caregiver burden, severe burden, and satisfaction with caregiving, stratified by gender. The results showed that secondary or third-level education, performance of ungratifying tasks, negative coping with caregiving, and more years providing care were associated with greater burden. Variables with protective effect were better perceived health of the person being cared for, better caregiver HRQoL, and high perceived social support. Women were 75% more likely to experience severe burden compared with male caregivers. Burden was reduced by high perceived social support in the case of women and by high caregiver HRQoL in the case of men. The main determinant of caregiving satisfaction for both men and women was perceived social support (OR = 3.11 and OR = 6.64). This study shows the need for interventions that promote gender equality and social support as a means of relieving burden and severe burden and improving satisfaction in both male and female caregivers.

The aim of this paper was to estimate the number of premature deaths, years of potential productive life lost (YPPLL) and labour losses attributable to tobacco smoking due to premature death by gender for the Spanish population. The human capital approach was applied. Employment, gross wage and death data were obtained from the Spanish National Institute of Statistics. Relative risks of death due to cigarette smoking and former smoking were applied. The base case used an annual discount rate of 3% and an annual labour productivity growth rate of 1%. Univariate deterministic sensitivity analysis was performed on discount rates and labour productivity growth rates. Between 2002 and 2016, smoking was estimated to cause around 13,171&ndash, 13,781 annual deaths in the population under 65 years of age (legal retirement age) in Spain. This increase was mostly due to female deaths. YPPLLs for females have increased over the years, while for males they have fallen markedly. Labour losses associated with smoking mortality ranged from &euro, 2269 million in 2002 to &euro, 1541 in 2016 (base year 2016). In fact, labour productivity losses have decreased over the years for men (&minus, 39.8%) but increased sharply for women (101.6%). The evolution of monetary value of lost productivity due to smoking mortality shows clearly differentiated trends by gender.

Objective The main aims of this paper are to analyse the monetary value of informal care time using different techniques and to identify significant variables associated with the number of caregiving hours. Data and methods A multicentre study in two Spanish regions in adult caregivers was conducted. A total sample of 604 people was available. A multivariate analysis was performed to identify the variables associated with the number of hours of caregiving time. In the monetary valuation of informal care provided, three approaches were used: replacement cost method, opportunity cost and contingent valuation (willingness to pay and willingness to accept). Results The main determinants of the amount of time of informal care provided were age, gender, the level of care receiver´s dependence and the professional care services received (at home and out of home). The value estimated for informal care time ranges from EUROS 80,247 (replacement cost method) to EUROS 14,325 (willingness to pay), with intermediate values of EUROS 27,140 and EUROS 29,343 (opportunity cost and willingness to accept, respectively). Several sensitivity analyses were performed over the base cases, confirming the previous results. Conclusions Time of informal care represents a great social value, regardless of the applied technique. However, the results can differ strongly depending on the technique chosen. Therefore, the choice of technique of valuation is not neutral. Among the determinants of informal care time, the professional care received at home has a complementary character to informal care, while the formal care outside the home has a substitute character.

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.

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.

Objectives: This study assessed the health-related quality of life (HRQOL) of pediatric patients with type 1 diabetes mellitus (T1DM) and their caregivers.Methods: CHRYSTAL was an observational cross-sectional study conducted in Spain in 2014 on 275 patients under 18 years old diagnosed with T1DM. Patient/caregiver pairs were stratified by patients' HbA1c level (?7.5% versus

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.

BACKGROUND: In spite of its high prevalence and its clinical relevance, the economic impact of malnutrition has not been sufficiently explored.OBJECTIVE: To study whether malnutrition predicts total hospital healthcare costs and costs related to specialist visits, emergency department visits and hospitalization in older adults.METHODS: Concurrent cohort study in Albacete City, Spain. The study sample included 827 subjects aged 70 and over from the FRADEA Study. Mini Nutritional Assessment®-Short Form (MNA®-SF) was recorded at baseline. Use of hospital resources (hospital admissions, emergency visits, and specialist visits), and hospital healthcare costs were recorded at follow-up. Generalized linear models (GLM) adjusted for age, sex, comorbidity, polypharmacy, and disability in basic activities of daily living were used to estimate the impact of nutritional factors on total healthcare costs per person/year (€ base year 2013) as well as specialist visit costs, emergency department visit costs and hospitalization costs.RESULTS: The average cost associated with the use of health resources was 1922€/year. Subjects with MNA®-SF between 0 and 7 had an average total health cost of 3492€/year, 2744€/year in those with MNA®-SF between 8 and 11, and 1542€/year in those with MNA®-SF between 12 and 14. Of the total health cost, 67.2% was associated with hospital admission costs. Adjusted healthcare costs were 714€/year greater in subjects with malnutrition or nutritional risk. Subjects with malnutrition or nutritional risk presented an increased adjusted risk of hospitalization (OR1.72, 95% CI 1.22-2.43).CONCLUSIONS: Malnutrition assessed by MNA®-SF is a prognostic factor of high healthcare cost and use of resources in older adults.

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.

a b s t r a c t Introduction: Stem cell transplantation has been used for many years to treat haematological malig- nancies that could not be cured by other treatments. Despite this medical breakthrough, mortality rates remain high. Our purpose was to evaluate labour productivity losses associated with premature mortality due to blood cancer in recipients of stem cell transplantations. Methods: We collected primary data from the clinical histories of blood cancer patients who had under- gone stem cell transplantation between 2006 and 2011 in two Spanish hospitals. We carried out a descriptive analysis and calculated the years of potential life lost and years of potential productive life lost. Labour productivity losses due to premature mortality were estimated using the Human Capital method. An alternative approach, the Friction Cost method, was used as part of the sensitivity analysis. Results: Our findings suggest that, in a population of 179 transplanted and deceased patients, males and people who die between the ages of 30 and 49 years generate higher labour productivity losses. The estimated loss amounts to over D 31.4 million using the Human Capital method (D 480,152 using the Friction Cost method), which means an average of D 185,855 per death. The highest labour productivity losses are produced by leukaemia. However, lymphoma generates the highest loss per death. Conclusions: Further efforts are needed to reduce premature mortality in blood cancer patients under- going transplantations and reduce economic losses.

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.

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 The aim of this study was to determine the economic burden and health-related quality of life (HRQOL) of patients with Spinal Muscular Atrophy (SMA) and their caregivers in Spain. Methods This was a cross-sectional and retrospective study of patients diagnosed with SMA in Spain. We adopted a bottom up, prevalence approach design to study patients with SMA. The patient’s caregivers completed an anonymous questionnaire regarding their socio-demographic characteristics, use of healthcare services and non-healthcare services. Costs were estimated from a societal perspective (including healthcare costs and non-healthcare costs), and health-related quality of life (HRQOL) was assessed using the EQ-5D questionnaire. The main caregivers also answered a questionnaire on their characteristics and on their HRQOL. Results A total of 81 caregivers of patients with different subtypes of SMA completed the questionnaire. Based on the reference unitary prices for 2014, the average annual costs per patient were € 33,721. Direct healthcare costs were € 10,882 (representing around 32.3% of the total cost) and the direct non-healthcare costs were € 22,839 (67.7% of the total cost). The mean EQ-5D social tariff score for patients was 0.16, and the mean score of the EQ-5D visual analogue scale was 54. The mean EQ-5D social tariff score for caregivers was 0.49 and their mean score on the EQ-5D visual analogue scale was 69. Conclusion The results highlight the burden that SMA has in terms of costs and decreased HRQOL, not only for patients but also for their caregivers. In particular, the substantial social/economic burden is mostly attributable to the high direct non-healthcare costs.

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.

A B S T R A C T Introduction and objectives: Chronic heart failure is associated with high mortality and utilization of health care and social resources. The objective of this study was to quantify the use of health care and nonhealth care resources and identify variables that help to explain variability in their costs in Spain. Methods: This prospective, multicenter, observational study with a 12-month follow-up period included 374 patients with symptomatic heart failure recruited from specialized cardiology clinics. Information was collected on the socioeconomic characteristics of patients and caregivers, health status, health care resources, and professional and nonprofessional caregiving. The monetary cost of the resources used in caring for the health of these patients was evaluated, differentiating among functional classes. Results: The estimated total cost for the 1-year follow-up ranged from s 12 995 to s 18 220, depending on the scenario chosen (base year, 2010). The largest cost item was informal caregiving (59.1%-69.8% of the total cost), followed by health care costs (26.7%- 37.4%), and professional care (3.5%). Of the total health care costs, the largest item corresponded to hospital costs, followed by medication. Total costs differed significantly between patients in functional class II and those in classes III or IV. Conclusions: Heart failure is a disease that requires the mobilization of a considerable amount of resources. The largest item corresponds to informal care. Both health care and nonhealth care costs are higher in the population with more advanced disease.

To estimate the monetary value of informal care to people with Alzheimer’s disease (AD) in Spain and to identify the main determinants of the time of informal care. We used the Survey on Disabilities, Autonomy and Dependency carried out in Spain in 2008 to obtain information on disabled individuals with Alzheimer and their informal caregivers. Assessment of informal care time was performed using three different approaches: the proxy good method, the opportunity cost method and the contingent valuation method. A statistical multivariate analysis—an ordered probit model—was performed to study the determinants of informal care provided. The average number of required informal hours per year was 4,151. The cost per caregiver ranged from 31,839 to 52,760 euros under the proxy good method; it was estimated at 20,053 euros under the opportunity cost method; and it ranged between 18,680 and 29,057 euros under the contingent valuation method. The degree of dependency and the formal care services were the main variables that explained the variability of the informal time provided. When a person presents a very high level of dependency, the probability of receiving more than 100 h of informal care per week is 25.8 % higher than a non-dependent person with AD. Formal in-home care complements informal caregiving, while formal care outside the home replaces it. Informal care represents a high social cost in people with AD, regardless of the estimation method considered. A higher level of dependence is associated with more hours of informal care provided.

BackgroundTo analyse and compare the impact of cardiovascular risk factors and disease on health-related quality of life (HRQoL) in people with and without diabetes living in the community.MethodsWe used data of 1,905 people with diabetes and 19,031 people without diabetes from the last Spanish National Health Survey (years 2011 +/- 2012). The HRQoL instrument used was the EuroQol 5D-5L, based on time trade-off scores. Matching methods were used to assess any differences in the HRQoL in people with and without diabetes with the same characteristics (age, gender, education level, and healthy lifestyle), according to cardiovascular risk factors and diseases. Disparities were also analysed for every dimension of HRQoL: mobility, daily activities, personal care, pain/discomfort, and anxiety/depression.ResultsThere were no significant differences in time trade-off scores between people with and without diabetes when cardiovascular risk factors or established cardiovascular disease were not present. However, when cardiovascular risk factors were present, the HRQoL score was significantly lower in people with diabetes than in those without. This difference was indeed greater when cardiovascular diseases were present. More precisely, people with diabetes and any of the cardiovascular risk factors, who have not yet developed any cardiovascular disease, report lower HRQoL, 0.046 TTO score points over 1 (7.93 over 100 in the VAS score) compared to those without diabetes, and 0.14 TTO score points of difference (14.61 over 100 in the VAS score) if cardiovascular diseases were present. In fact, when the three risk factors were present in people with diabetes, HRQoL was significantly lower (0.10 TTO score points over 1 and 10.86 points over 100 in VAS score), obesity being the most influential risk factor.ConclusionsThe presence of established cardiovascular disease and/or cardiovascular risk factors, specially obesity, account for impaired quality of life in people with diabetes.

Objectives Cost-sharing scheme for pharmaceuticals in Spain changed in July 2012. Our aim was to assess the impact of this change on adherence to essential medication in patients with acute coronary syndrome (ACS) in the region of Valencia. Methods Population-based retrospective cohort of 10 563 patients discharged alive after an ACS in 2009-2011. We examined a control group (low-income working population) that did not change their coinsurance status, and two intervention groups: pensioners who moved from full coverage to 10% coinsurance and middle-income to high-income working population, for whom coinsurance rose from 40% to 50% or 60%. Weekly adherence rates measured from the date of the first prescription. Days with available medication were estimated by linking prescribed and filled medications during the follow-up period. Results Cost-sharing change made no significant differences in adherence between intervention and control groups for essential medications with low price and low patient maximum coinsurance, such as antiplatelet and beta-blockers. For costlier ACE inhibitor or an angiotensin II receptor blocker (ACEI/ARB) and statins, it had an immediate effect in the proportion of adherence in the pensioner group as compared with the control group (6.8% and 8.3% decrease of adherence, respectively, p

There is a growing interest in incorporating informal care in cost-of-illness studies as a relevant part of the economic impact of some diseases. The aim of this paper was to review the recent literature valuating the costs of informal care in a group of selected diseases from 2005 to 2015. We carried out a systematic review on the economic impact of informal care, focusing on six selected diseases: arthritis or osteoarthritis, cancer, dementia, mental diseases, multiple sclerosis and stroke. We selected 91 cost-of-illness articles. The average weight attributed to the informal care cost over the total cost was highly relevant for dementia, stroke, mental diseases, cancer and multiple sclerosis. The most frequent valuation method applied was the opportunity cost method, followed by the proxy good method. The annual cost of informal care presented a high variability depending on the disease and geographic location. Distinguishing by type of illness, the disease with the highest annual value of informal caregiving was dementia, followed by mental illness and multiple sclerosis. The average hourly unit cost was €11.43 (2015 values), varying noticeably depending on the geographic location. This paper identifies several aspects that should be enhanced to promote comparability between studies and countries, and it sends key messages for incorporating informal care costs to adequately measure the economic impact of diseases.

At the end of 2006, a new System for Promotion of Personal Autonomy and Assistance for Persons in a Situation of Dependency (SAAD) was established in Spain through the approval of the Act 39/2006 of 14th December (the Dependency Act, DA). The DA acknowledged the universal entitlement of Spanish citizens to social services. The recent economic crisis added degrees of uncertainty to several dimensions of the SAAD implementation process. Firstly, the political consensus on which its foundation rested upon has weakened. Secondly, implementation of the SAAD was hampered by several challenges that emerged in the context of the economic crisis. Thirdly, the so-called “dependency limbo” (i.e. the existence of a large number of people eligible for benefits but who do not receive them) has become a structural feature of the system. Finally, contrary to the spirit of the DA, monetary benefits have become the norm rather than a last resort. High heterogeneity across regions regarding the number of beneficiaries covered and services provided reveal the existence of regional inequity in access to long-term care services in the country. Broadly, the current evidence on the state of the SAAD suggests the need to improve the quality of governance, to enhance coordination between health and social systems, to increase the system's transparency, to foster citizens’ participation in decision-making and to implement a systematic monitoring of the system.

Background HIV/AIDS (Human immunodeficiency virus/Acquired immune deficiency syndrome) not only has a strong impact on the health of the worldwide population but also affects the labour status of HIV-positive people. The primary aim of this paper is to compare the labour participation of people living with HIV (PlwHIV) with the labour participation of the general population along the last business cycle in Spain. Method The data used are from the Hospital Survey on HIV-AIDS, with a total sample size of 4,651 PlwHIV and the Labour Force Survey from 2001 to 2010, with a total sample size of 660,674 individuals as general population. Propensity Score Matching method was used to analyse the differences between the labour participation of PlwHIV and the general population. Additionally, several specific models categorised into different subgroups (gender, education, source of infection and level of defences) were also performed. Results We identified a convergence in labour participation across the period in the two populations considered: PlwHIV was 23% less likely to have a job than the general population during 2001–2002 and 14% less likely during 2009–2010. This convergence is mainly explained by two facts: first, the positive evolution of people infected by sex; second, the change in the PlwHIV population composition with a decreasing weight of people infected by drug use throughout the decade. Thereby, at the end of period, there was no statistical difference in the employment rate between PlwHIV infected through sex and the general population but there was strongly difference in PlwHIV infected through drugs. Conclusion Inmunological status, source of infection and level of education play a relevant role among the PlwHIV population when comparing their labour participation with the general population. In spite of this positive result, the likelihood of being employed in HIV-positive people continues to be different from that of non-carriers. Our study shows that institutional features of labour markets are relevant and should be considered in comparison between countries.

Objective: This study explores the relationship between the employment status of human immunodeficiency virus (HIV)-positive individuals and socioeconomic and health characteristics in Spain. Methods: Data were obtained from four waves of the HIV/AIDS Hospital Survey from the period 2001 to 2004. The sample was composed of 3376 individuals between 16 and 64 years. Assessment of employment participation was performed using a probit model on a wide range of socioeconomic and lifestyle-related variables. Results: The main variables explaining the probability of participation in the labour market were age, gender, education level, CD4 cell count, health status, time since HIV diagnosis, psychological impact of contracting the disease or its progression, the most likely means of transmission and intravenous drug use (IDU). The significance of each of these variables differs for men and women as well as for injecting drug users (IDUs) and non-IDUs. Conclusions: The employment status of HIV-positive individuals is directly related to their health status and other personal characteristics. Policies to improve the well-being of HIV-positive individuals should not be limited to any one sector as their needs require strategies with a multidisciplinary approach. Copyright © 2009 John Wiley & Sons, Ltd.

The aims of this study were to estimate medical expenditures on human immunodeficiency virus (HIV) treatment and to identify significant associated variables. We performed a retrospective multi-centre study in the Canary Islands using a sample of 569 patients recruited at outpatient visits. The study examined demographic and clinical variables, health-related quality of life (HRQOL), and health care resources. Clinical data was obtained from medical records and patient interviews. Several empirical models for identifying the relationship between health care costs and independent variables were developed. The greatest expense came from pharmaceutical expenditure (82.1% of direct costs), while hospital costs only represented 4.6% of total expenditure. The data showed a statistically significant association between health care costs and the CD4 count of the previous year. HRQOL was also a significant variable. Therefore, CD4 cell count can be used to predict health care costs in patients. Policymakers could use this information to help guide their decisions in allocating limited health care resources to HIV treatments.

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 histiocytosis in Europe. We conducted a cross-sectional study of patients with histiocytosis from France, Germany, Italy, Spain, Bulgaria, the UK, and Sweden. Data on demographic characteristics, health resource utilisation, informal care, loss of labour productivity 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 134 patients (35 France, 32 Germany, 30 Italy, 24 Spain, 7 Bulgaria, 4 UK and 2 Sweden) completed the questionnaire. The average annual costs ranged from € 6832 to € 33,283 between countries, the year of reference being 2012. Estimated direct healthcare costs ranged from € 1698 to € 18,213; direct nonhealthcare costs ranged from € 2936 to € 17,622 and labour productivity losses ranged from € 1 to € 8855. The mean EQ-5D score for adult histiocytosis patients was estimated at between 0.32 and 0.85, and the mean EQ-5D visual analogue scale score was estimated at between 50.00 and 66.50. The main strengths of this study lie in our bottom-up approach to costing and in the evaluation of histiocytosis patients from a broad perspective (societal costs). This type of analysis is very scarce in international literature for rare diseases in comparison with other illnesses. We conclude that histiocytosis patients incur considerable societal costs and experience substantial deterioration in HRQOL.

Although there is no single commonly accepted definition worldwide for rare diseases [1], the European Commission has agreed that rare diseases are lifethreatening or chronically debilitating conditions affecting no more than 5 in 10,000 people in the European Union (EU) [2]. Despite the low frequency of each single rare disease, it is estimated that between 5000 and 8000 rare diseases have been described, affecting 6–8 % of the population in the course of their lives, for a total number of people ranging between 27 and 36 million in the EU [3–5]. While the effects of rare diseases are varied, many have serious health consequences, a high proportion being degenerative and life-threatening. About 80 % of rare diseases have a genetic origin. Approximately 50 % have a childhood onset and it is estimated that over one-third of deaths of children under the age of 1 year are due to rare diseases [6]. It is also worth noting that due to their low prevalence, the correct diagnosis of rare diseases is complex and subject to significant delays. Moreover, despite significant advances brought about by specific health policies in recent years [2, 7], not only do most rare diseases have no cure but, for many, there is no effective treatment available or, if treatments exist, there is no guarantee of improvement in life expectancy or quality of life. The combination of these elements—severity of illness, diagnostic uncertainty, lack of effective treatments— has a strong social impact that rests largely on patients and their families. Two insufficiently examined issues with rare diseases are (i) the economic impact caused to society, and (ii) the loss of health-related quality of life (HRQOL) for affected patients and their caregivers. Although some countryspecific research on a limited set of rare diseases has been done on HRQOL and cost-of-illness [8–12], cross-national research on the socio-economic impact of rare diseases is still lacking in the EU. The ‘‘Social Economic Burden and Health-Related Quality of Life in Patients with Rare Diseases in Europe’’ (BURQOL-RD) project was a 3-year project under the framework of the Second Programme of Community Action in the Field of Public Health, which began in April 2010, and was promoted by the Directorate General for Health and Consumer Affairs (DG Sanco) [13]. Its objective was to develop a disease-based model capable of quantifying the socio-economic burden and HRQOL of patients suffering from ten rare diseases and their caregivers in Europe (reference year: 2012). Hence, BURQOLRD can help to set the basis for an integrated and harmonised approach to assess public policies and interventions for rare diseases in the EU. The availability of these approaches and their application will be crucial to define the current consequences of rare diseases on society, and to assess the effectiveness of new policies and interventions [14], opening a channel for studying the cost-effectiveness of new treatments (e.g. orphan drugs). & Julio Lopez-Bastida julio.lopezbastida@uclm.es

The development of the economic evaluation of health care interventions has become a support tool in making decisions on pricing and reimbursement of new health interventions. The increasingly extensive application of these techniques has led to the identification of particular situations in which, for various reasons, it may be reasonable to take into account special considerations when applying the general principles of economic evaluation. In this article, which closes a series of three, we will discuss, using the Metaplan technique, about the economic evaluation of health interventions in special situations such as rare diseases and end of life treatments, as well as consideration of externalities in assessments, finally pointing out some research areas to solve the main problems identified in these fields.

In this second article of a series of three, we will discuss using the Metaplan technique on controversial issues of health outcomes in economic evaluation of health care interventions. The four-discussion areas focus on: choice of health outcomes measures, where any outcome measure is superior to another; extrapolation and transferability of health outcomes measures, which should not be assumed the results of an EEIS of one country to another without making certain adjustments; appropriate instruments to measure quality of life in Spain, where the EQ-5D was indicated as convenient due to its widespread international use; and, indirect comparisons, where the combination of both comparisons, direct and indirect, it would be advisable if the test for indirect estimates is consistent and has been validated. Finally, research lines to try to overcome the identified discrepancies were identified in each of these areas, some of those are: doing studies of correlation between scores of specific and generic instruments measuring quality of life; update or create a database of economic evaluations in Spain; estimating utilities for the Spanish population by existing generic and specific instruments; or, establish a common way to show the results of a meta-analysis network.

Our goal was to provide data on the economic burden and health-related quality of life (HRQOL) of patients with cystic fibrosis (CF) and their caregivers in Europe.A cross-sectional study was carried out on adults and children with CF in eight European countries. Patients completed an anonymous questionnaire regarding their socio-demographic characteristics, use of healthcare services and presence of a caregiver. Costs were calculated with a bottom-up approach using unit costs from each participating country, and HRQOL was assessed using EQ-5D. The principal caregiver also answered a questionnaire on their characteristics, HRQOL and burden.A total of 905 patients with CF was included (399 adults and 506 children). The total average annual cost per patient varied from €21,144 in Bulgaria to €53,256 in Germany. Adults had higher direct healthcare costs than children, but children had much higher informal care costs (P 0.0001). Total costs increased with patients' level of dependence. In adults, mean utility fell between 0.640 and 0.870, and the visual analogue scale ranged from 46.0 to 69.7. There was no difference in caregiver HRQOL regardless of whether they cared for an adult or a child. However, caregivers who looked after a child had a significantly higher burden (P = 0.0013).Our study highlights the burden of CF in terms of costs and decreased HRQOL for both patients and their caregivers throughout Europe.