Social/economic costs and health-related quality of life of mucopolysaccharidosis patients and their caregivers in Europe

Péntek, Márta; Gulàcsi, László; Brodszky, Valentin; Baji, Petra; Boncz, I; Pogány, Gábor; López-Bastida, Julio; Linertová, Renata; Oliva‐Moreno, Juan; Serrano‐Aguilar, Pedro; Paz, Manuel Posada de la; Taruscio, Domenica; Искров, Георги; Schieppati, Arrigo; Schulenburg, J.-Matthias Graf von der; Kanavos, Panos; Chevreul, Karine; Persson, Ulf; Fattore, Giovanni

doi:10.1007/s10198-016-0787-0

Cited by 46 publications

(45 citation statements)

References 26 publications

(30 reference statements)

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“…Duchenne muscular dystrophy, epidermolysis bullosa, Prader–Willi syndrome, cystic fibrosis, and haemophilia were studied in two countries (Hungary and Bulgaria) applying the same methodology in a European Commission founded rare disease study (BURQOL-RD project). Prader–Willi syndrome was the least costly (Bulgaria: €3842 Hungary: €12,532) and mucopolysaccharidosis was the most costly rare disease (Bulgaria: €77,414; Hungary: €25,326) [ 28 , 29 ].…”

Section: Methodsmentioning

confidence: 99%

Cost-of-illness studies in nine Central and Eastern European countries

et al. 2019

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Background To date, a multi-country review evaluating the cost-of-illness (COI) studies from the Central and Eastern European (CEE) region has not yet been published. Our main objective was to provide a general description about published COI studies from CEE. Methods A systematic search was performed between 1 January 2006 and 1 June 2017 in Medline, EMBASE, The Cochrane Library, CINAHL, and Web of Science to identify all relevant COI studies from nine CEE countries. COI studies reporting costs without any restrictions by age, co-morbidities, or treatment were included. Methodology, publication standards, and cost results were analysed. Results We identified 58 studies providing 83 country-specific COI results: Austria ( n = 9), Bulgaria ( n = 16), Croatia ( n = 3), the Czech Republic ( n = 10), Hungary ( n = 24), Poland ( n = 11), Romania ( n = 3), Slovakia ( n = 3), and Slovenia ( n = 4). Endocrine, nutritional, and metabolic diseases (18%), neoplasms (12%), infections (11%), and neurological disorders (11%) were the most frequently studied clinical areas, and multiple sclerosis was the most commonly studied disease. Overall, 57 (98%) of the studies explicitly stated the source of resource use data, 45 (78%) the study perspective, 34 (64%) the costing method, and 24 (58%) reported at least one unit costs. Regardless of methodological differences, a positive relationship was observed between costs of diseases and countries’ per capita GDP. Conclusions Cost-of-illness studies varied considerably in terms of methodology, publication practice, and clinical areas. Due to these heterogeneities, transferability of the COI results is limited across Central and Eastern European countries. Electronic supplementary material The online version of this article (10.1007/s10198-019-01066-x) contains supplementary material, which is available to authorized users.

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Section: Methodsmentioning

confidence: 99%

Cost-of-illness studies in nine Central and Eastern European countries

et al. 2019

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“…Available studies mostly focus on informal care volume (i.e,. share of people receiving informal care, care time in hours per week) in specific diseases based on self-reports of the patients [6][7][8][9][10][11][12][13]. Less is known about the overall burden of informal caregiving among the general population, especially from the caregivers' perspective and including QoL effects of caregiving.…”

Section: Introductionmentioning

confidence: 99%

The burden of informal caregiving in Hungary, Poland and Slovenia: results from national representative surveys

et al. 2019

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Background We aimed to investigate the burden of informal care in Hungary (HU), Poland (PL) and Slovenia (SI). Methods A cross-sectional online survey was performed involving representative samples of 1000 respondents per country. Caregiving situations were explored; health status of informal caregivers/care recipients and care-related quality of life were assessed using the EQ-5D-5L and CarerQol-7D. Results The proportion of caregivers was (HU/PL/SI) 14.9, 15.0 and 9.6%, respectively. Their mean age was 56.1, 45.6 and 48.0, and the average time spent on informal care was 27.6, 35.5 and 28.8 h/week. Chronic care was dominant (> 1 year: 78.5%, 72.0%, 74.0%) and care recipients were mainly (own/in-law) parents. Average EQ-5D-5L scores of care recipients were 0.53, 0.49 and 0.52. For Poland and Slovenia, EQ-5D-5L scores of informal care providers were significantly lower than of other respondents. Average CarerQol-7D scores were (HU/PL/SI) 76.0, 69.6 and 70.9, and CarerQol-VAS was 6.8, 6.4 and 6.6, respectively. Overall, 89, 87, and 84% of caregivers felt some or a lot fulfilment related to caring. Problems with combining tasks with daily activities were most important in Hungary and Slovenia. Women had a higher probability of being a caregiver in Hungary. CarerQol-7D scores were significantly associated with caregivers' EQ-5D-5L scores. In Hungary and Poland, living in a larger household was positively, while caring for patients with mental health problems was negatively associated with CarerQol-7D scores. Conclusions These first results from the Central and Eastern European region using preference-based measures for the evaluation of informal care can serve as a valuable input for health economic analyses.

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“…According to a review by Jurecka et al [46], the prevalence of MPS (all types) per 100,000 live births was 4.5 in the Netherlands, 3.7 in the Czech Republic, 3.5 in Germany, 3.1 in Norway, 1.8 in Denmark, 1.8 in Sweden and 1.8 in Poland. Pentek et al [47] estimated a mean yearly total cost per patient with MPS that ranged from €24,520 (Hungary) to €209,420 (Germany). Direct healthcare costs ranged from €275 per patient in France to €206,283 in Germany and direct non-healthcare costs ranged from €24,066 per patient in Italy to €201,371 in Sweden.…”

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confidence: 99%

Social/economic costs and health-related quality of life in patients with rare diseases in Europe

et al. 2016

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Social/economic costs and health-related quality of life of mucopolysaccharidosis patients and their caregivers in Europe

Abstract: MPS patients experience substantial loss of HRQOL and their families take a remarkable part in their care. Although utilization of health and social care resources varies significantly across countries, MPS incurs considerable societal costs in all the countries studied.

Cited by 46 publications

References 26 publications

Cost-of-illness studies in nine Central and Eastern European countries

Cost-of-illness studies in nine Central and Eastern European countries

The burden of informal caregiving in Hungary, Poland and Slovenia: results from national representative surveys

Social/economic costs and health-related quality of life in patients with rare diseases in Europe

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