| Title |
Social/economic costs and health-related quality of life of mucopolysaccharidosis patients and their caregivers in Europe
|
|---|---|
| Published in |
HEPAC Health Economics in Prevention and Care, April 2016
|
| DOI | 10.1007/s10198-016-0787-0 |
| Pubmed ID | |
| Authors |
Márta Péntek, László Gulácsi, Valentin Brodszky, Petra Baji, Imre Boncz, Gábor Pogány, Julio López-Bastida, Renata Linertová, Juan Oliva-Moreno, Pedro Serrano-Aguilar, Manuel Posada-de-la-Paz, Domenica Taruscio, Georgi Iskrov, Arrigo Schieppati, Johann Matthias Graf von der Schulenburg, Panos Kanavos, Karine Chevreul, Ulf Persson, Giovanni Fattore, BURQOL-RD Research Network |
| Abstract |
To assess the health-related quality of life (HRQOL) of patients with mucopolysaccharidosis (MPS) and their caregivers and to quantify the disease-related costs from a societal perspective. In the context of a multi-country study of rare diseases (BURQOL-RD project), a cross-sectional survey was performed among MPS patients in seven European countries. Data on demographic characteristics, health resource utilization, informal care, and loss of labor productivity were collected. The EQ-5D, Barthel index (BI), and Zarit burden interview (ZBI) questionnaires were used to assess patients' and their informal caregivers' quality of life, patients' functional ability, and caregivers' burden, respectively. Altogether, 120 patients (children 62 %, females 40 %) and 66 caregivers completed the questionnaire. Patients' mean age was 16.5 years and median age at diagnosis was 3 years. Adult patients' average EQ-5D and EQ VAS scores varied across countries from 0.13 to 0.43 and 30.0 to 62.2, respectively, mean BI was 46.7, and ZBI was 32.7. Mean informal care time was 51.3 h/week. The mean total annual cost per patient (reference year 2012) was €24,520 in Hungary, €25,993 in France, €84,921 in Italy, €94,384 in Spain, and €209,420 in Germany. Costs are also shown to differ between children and adults. Direct costs accounted for most of the costs in all five countries (80, 100, 99, 98, and 93 %, respectively). 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. |
X Demographics
The data shown below were collected from the profiles of 3 X users who shared this research output. Click here to find out more about how the information was compiled.
Geographical breakdown
| Country | Count | As % |
|---|---|---|
| United States | 1 | 33% |
| Spain | 1 | 33% |
| Unknown | 1 | 33% |
Demographic breakdown
| Type | Count | As % |
|---|---|---|
| Members of the public | 2 | 67% |
| Scientists | 1 | 33% |
Mendeley readers
The data shown below were compiled from readership statistics for 144 Mendeley readers of this research output. Click here to see the associated Mendeley record.
Geographical breakdown
| Country | Count | As % |
|---|---|---|
| Unknown | 144 | 100% |
Demographic breakdown
| Readers by professional status | Count | As % |
|---|---|---|
| Researcher | 22 | 15% |
| Student > Master | 17 | 12% |
| Student > Bachelor | 16 | 11% |
| Student > Ph. D. Student | 13 | 9% |
| Other | 11 | 8% |
| Other | 23 | 16% |
| Unknown | 42 | 29% |
| Readers by discipline | Count | As % |
|---|---|---|
| Medicine and Dentistry | 26 | 18% |
| Nursing and Health Professions | 14 | 10% |
| Pharmacology, Toxicology and Pharmaceutical Science | 12 | 8% |
| Economics, Econometrics and Finance | 10 | 7% |
| Agricultural and Biological Sciences | 8 | 6% |
| Other | 30 | 21% |
| Unknown | 44 | 31% |
Attention Score in Context
This research output has an Altmetric Attention Score of 2. This is our high-level measure of the quality and quantity of online attention that it has received. This Attention Score, as well as the ranking and number of research outputs shown below, was calculated when the research output was last mentioned on 19 May 2017.
All research outputs
#16,387,941
of 25,870,940 outputs
Outputs from HEPAC Health Economics in Prevention and Care
#868
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Outputs of similar age
#175,077
of 316,802 outputs
Outputs of similar age from HEPAC Health Economics in Prevention and Care
#21
of 34 outputs
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