Social/economic costs and health-related quality of life in patients with Prader-Willi syndrome in Europe
Objective 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. Methods We conducted a cross-sectional study of patients with PWS from Spain, Bulgaria, Hungary, Germany...
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| Veröffentlicht in: | The European journal of health economics 2016-04, Vol.17 (Suppl 1), p.S99-S108 |
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| creator | López-Bastida, Julio Linertová, Renata Oliva-Moreno, Juan Posada-de-la-Paz, Manuel Serrano-Aguilar, Pedro Kanavos, Panos Taruscio, Domenica Schieppati, Arrigo Iskrov, Georgi Baji, Petra Delgado, Claudia von der Schulenburg, Johann Matthias Graf Persson, Ulf Chevreul, Karine Fattore, Giovanni |
| description | Objective 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. Methods 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. Results 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. Conclusion 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. |
| doi_str_mv | 10.1007/s10198-016-0788-z |
| format | Article |
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Methods 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. Results 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. Conclusion 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.</description><identifier>ISSN: 1618-7598</identifier><identifier>EISSN: 1618-7601</identifier><identifier>DOI: 10.1007/s10198-016-0788-z</identifier><identifier>PMID: 27038627</identifier><language>eng</language><publisher>Berlin/Heidelberg: Springer</publisher><subject>Adolescent ; Adult ; Average total cost ; Caregivers ; Child ; Cost of Illness ; Costs ; Cross-Sectional Studies ; Direct costs ; Direct labor costs ; Direct service costs ; Economic Policy ; Economics ; Europe ; Female ; Health care ; Health care costs ; Health Care Costs - statistics & numerical data ; Health care expenditures ; Health Care Management ; Health Economics ; Hospital costs ; Humans ; Illnesses ; Labor productivity ; Male ; Medical research ; Medicine ; Medicine & Public Health ; Middle Aged ; Original Paper ; Patient Care - economics ; Patients ; Pharmacoeconomics and Health Outcomes ; Prader Willi syndrome ; Prader-Willi Syndrome - economics ; Prader-Willi Syndrome - psychology ; Productivity ; Public Finance ; Public Health ; Quality of Life ; Questionnaires ; Rare diseases ; Sick Leave - economics ; Sickness Impact Profile ; Socioeconomic Factors ; Surveys and Questionnaires ; United Kingdom ; Visual Analog Scale ; Young Adult</subject><ispartof>The European journal of health economics, 2016-04, Vol.17 (Suppl 1), p.S99-S108</ispartof><rights>Springer-Verlag Berlin Heidelberg 2016</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c427t-2dab39ee149c50e12a40e40da88bb9c9c4f692e6f6c0b8cecd4979b00eed7afe3</citedby><cites>FETCH-LOGICAL-c427t-2dab39ee149c50e12a40e40da88bb9c9c4f692e6f6c0b8cecd4979b00eed7afe3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.jstor.org/stable/pdf/24774157$$EPDF$$P50$$Gjstor$$H</linktopdf><linktohtml>$$Uhttps://www.jstor.org/stable/24774157$$EHTML$$P50$$Gjstor$$H</linktohtml><link.rule.ids>314,780,784,803,27924,27925,41488,42557,51319,58017,58250</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/27038627$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>López-Bastida, Julio</creatorcontrib><creatorcontrib>Linertová, Renata</creatorcontrib><creatorcontrib>Oliva-Moreno, Juan</creatorcontrib><creatorcontrib>Posada-de-la-Paz, Manuel</creatorcontrib><creatorcontrib>Serrano-Aguilar, Pedro</creatorcontrib><creatorcontrib>Kanavos, Panos</creatorcontrib><creatorcontrib>Taruscio, Domenica</creatorcontrib><creatorcontrib>Schieppati, Arrigo</creatorcontrib><creatorcontrib>Iskrov, Georgi</creatorcontrib><creatorcontrib>Baji, Petra</creatorcontrib><creatorcontrib>Delgado, Claudia</creatorcontrib><creatorcontrib>von der Schulenburg, Johann Matthias Graf</creatorcontrib><creatorcontrib>Persson, Ulf</creatorcontrib><creatorcontrib>Chevreul, Karine</creatorcontrib><creatorcontrib>Fattore, Giovanni</creatorcontrib><creatorcontrib>The BURQOL-RD Research Network</creatorcontrib><creatorcontrib>BURQOL-RD Research Network</creatorcontrib><title>Social/economic costs and health-related quality of life in patients with Prader-Willi syndrome in Europe</title><title>The European journal of health economics</title><addtitle>Eur J Health Econ</addtitle><addtitle>Eur J Health Econ</addtitle><description>Objective 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. Methods 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. Results 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. Conclusion 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.</description><subject>Adolescent</subject><subject>Adult</subject><subject>Average total cost</subject><subject>Caregivers</subject><subject>Child</subject><subject>Cost of Illness</subject><subject>Costs</subject><subject>Cross-Sectional Studies</subject><subject>Direct costs</subject><subject>Direct labor costs</subject><subject>Direct service costs</subject><subject>Economic Policy</subject><subject>Economics</subject><subject>Europe</subject><subject>Female</subject><subject>Health care</subject><subject>Health care costs</subject><subject>Health Care Costs - statistics & numerical data</subject><subject>Health care expenditures</subject><subject>Health Care Management</subject><subject>Health Economics</subject><subject>Hospital costs</subject><subject>Humans</subject><subject>Illnesses</subject><subject>Labor productivity</subject><subject>Male</subject><subject>Medical research</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Middle Aged</subject><subject>Original Paper</subject><subject>Patient Care - economics</subject><subject>Patients</subject><subject>Pharmacoeconomics and Health Outcomes</subject><subject>Prader Willi syndrome</subject><subject>Prader-Willi Syndrome - economics</subject><subject>Prader-Willi Syndrome - psychology</subject><subject>Productivity</subject><subject>Public Finance</subject><subject>Public Health</subject><subject>Quality of Life</subject><subject>Questionnaires</subject><subject>Rare diseases</subject><subject>Sick Leave - economics</subject><subject>Sickness Impact Profile</subject><subject>Socioeconomic Factors</subject><subject>Surveys and Questionnaires</subject><subject>United Kingdom</subject><subject>Visual Analog Scale</subject><subject>Young Adult</subject><issn>1618-7598</issn><issn>1618-7601</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2016</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><recordid>eNp9kU9rFTEUxQdRbK1-ABdKwI2b2JtM3iRZSql_oKCg4jJkkju-PDKT12SG8vrpzXNaFReuciG_c-7lnKZ5zuANA5DnhQHTigLrKEil6O2D5pR1TFHZAXt4P2-0OmmelLID4Fzy9nFzwiW0quPytAlfkgs2nqNLUxqDIy6VuRA7ebJFG-ctzRjtjJ5cLzaG-UDSQGIYkISJ7O0ccKr4TZi35HO2HjP9HmIMpBwmn9P4C7tcctrj0-bRYGPBZ3fvWfPt3eXXiw_06tP7jxdvr6gTXM6Ue9u3GpEJ7TaAjFsBKMBbpfpeO-3E0GmO3dA56JVD54WWugdA9NIO2J41r1fffU7XC5bZjKE4jNFOmJZiWE1qI3grZEVf_YPu0pKnet2RkhqgxlwptlIup1IyDmafw2jzwTAwxx7M2oOpPZhjD-a2al7eOS_9iP634j74CvAVKPVr-oH5r9X_cX2xinZlTvmPqZBSsI1sfwI4kZ6g</recordid><startdate>20160401</startdate><enddate>20160401</enddate><creator>López-Bastida, Julio</creator><creator>Linertová, Renata</creator><creator>Oliva-Moreno, Juan</creator><creator>Posada-de-la-Paz, Manuel</creator><creator>Serrano-Aguilar, Pedro</creator><creator>Kanavos, Panos</creator><creator>Taruscio, Domenica</creator><creator>Schieppati, Arrigo</creator><creator>Iskrov, Georgi</creator><creator>Baji, Petra</creator><creator>Delgado, Claudia</creator><creator>von der Schulenburg, Johann Matthias Graf</creator><creator>Persson, Ulf</creator><creator>Chevreul, Karine</creator><creator>Fattore, Giovanni</creator><general>Springer</general><general>Springer Berlin Heidelberg</general><general>Springer Nature B.V</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7WY</scope><scope>7WZ</scope><scope>7X7</scope><scope>7XB</scope><scope>87Z</scope><scope>88C</scope><scope>88E</scope><scope>8AO</scope><scope>8C1</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>8FL</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>BEZIV</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FRNLG</scope><scope>FYUFA</scope><scope>F~G</scope><scope>GHDGH</scope><scope>K60</scope><scope>K6~</scope><scope>K9.</scope><scope>L.-</scope><scope>M0C</scope><scope>M0S</scope><scope>M0T</scope><scope>M1P</scope><scope>PQBIZ</scope><scope>PQBZA</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PYYUZ</scope><scope>Q9U</scope><scope>7X8</scope></search><sort><creationdate>20160401</creationdate><title>Social/economic costs and health-related quality of life in patients with Prader-Willi syndrome in Europe</title><author>López-Bastida, Julio ; Linertová, Renata ; Oliva-Moreno, Juan ; Posada-de-la-Paz, Manuel ; Serrano-Aguilar, Pedro ; Kanavos, Panos ; Taruscio, Domenica ; Schieppati, Arrigo ; Iskrov, Georgi ; Baji, Petra ; Delgado, Claudia ; von der Schulenburg, Johann Matthias Graf ; Persson, Ulf ; Chevreul, Karine ; Fattore, Giovanni</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c427t-2dab39ee149c50e12a40e40da88bb9c9c4f692e6f6c0b8cecd4979b00eed7afe3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2016</creationdate><topic>Adolescent</topic><topic>Adult</topic><topic>Average total cost</topic><topic>Caregivers</topic><topic>Child</topic><topic>Cost of Illness</topic><topic>Costs</topic><topic>Cross-Sectional Studies</topic><topic>Direct costs</topic><topic>Direct labor costs</topic><topic>Direct service costs</topic><topic>Economic Policy</topic><topic>Economics</topic><topic>Europe</topic><topic>Female</topic><topic>Health care</topic><topic>Health care costs</topic><topic>Health Care Costs - statistics & numerical data</topic><topic>Health care expenditures</topic><topic>Health Care Management</topic><topic>Health Economics</topic><topic>Hospital costs</topic><topic>Humans</topic><topic>Illnesses</topic><topic>Labor productivity</topic><topic>Male</topic><topic>Medical research</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Middle Aged</topic><topic>Original Paper</topic><topic>Patient Care - economics</topic><topic>Patients</topic><topic>Pharmacoeconomics and Health Outcomes</topic><topic>Prader Willi syndrome</topic><topic>Prader-Willi Syndrome - economics</topic><topic>Prader-Willi Syndrome - psychology</topic><topic>Productivity</topic><topic>Public Finance</topic><topic>Public Health</topic><topic>Quality of Life</topic><topic>Questionnaires</topic><topic>Rare diseases</topic><topic>Sick Leave - economics</topic><topic>Sickness Impact Profile</topic><topic>Socioeconomic Factors</topic><topic>Surveys and Questionnaires</topic><topic>United Kingdom</topic><topic>Visual Analog Scale</topic><topic>Young Adult</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>López-Bastida, Julio</creatorcontrib><creatorcontrib>Linertová, Renata</creatorcontrib><creatorcontrib>Oliva-Moreno, Juan</creatorcontrib><creatorcontrib>Posada-de-la-Paz, Manuel</creatorcontrib><creatorcontrib>Serrano-Aguilar, Pedro</creatorcontrib><creatorcontrib>Kanavos, Panos</creatorcontrib><creatorcontrib>Taruscio, Domenica</creatorcontrib><creatorcontrib>Schieppati, Arrigo</creatorcontrib><creatorcontrib>Iskrov, Georgi</creatorcontrib><creatorcontrib>Baji, Petra</creatorcontrib><creatorcontrib>Delgado, Claudia</creatorcontrib><creatorcontrib>von der Schulenburg, Johann Matthias Graf</creatorcontrib><creatorcontrib>Persson, Ulf</creatorcontrib><creatorcontrib>Chevreul, Karine</creatorcontrib><creatorcontrib>Fattore, Giovanni</creatorcontrib><creatorcontrib>The BURQOL-RD Research Network</creatorcontrib><creatorcontrib>BURQOL-RD Research Network</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Access via ABI/INFORM (ProQuest)</collection><collection>ABI/INFORM Global (PDF only)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>ABI/INFORM Global (Alumni Edition)</collection><collection>Healthcare Administration Database (Alumni)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Public Health Database</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ABI/INFORM Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central</collection><collection>Business Premium Collection</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Business Premium Collection (Alumni)</collection><collection>Health Research Premium Collection</collection><collection>ABI/INFORM Global (Corporate)</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Business Collection (Alumni Edition)</collection><collection>ProQuest Business Collection</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>ABI/INFORM Professional Advanced</collection><collection>ABI/INFORM Global</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Healthcare Administration Database</collection><collection>Medical Database</collection><collection>ProQuest One Business</collection><collection>ProQuest One Business (Alumni)</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ABI/INFORM Collection China</collection><collection>ProQuest Central Basic</collection><collection>MEDLINE - Academic</collection><jtitle>The European journal of health economics</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>López-Bastida, Julio</au><au>Linertová, Renata</au><au>Oliva-Moreno, Juan</au><au>Posada-de-la-Paz, Manuel</au><au>Serrano-Aguilar, Pedro</au><au>Kanavos, Panos</au><au>Taruscio, Domenica</au><au>Schieppati, Arrigo</au><au>Iskrov, Georgi</au><au>Baji, Petra</au><au>Delgado, Claudia</au><au>von der Schulenburg, Johann Matthias Graf</au><au>Persson, Ulf</au><au>Chevreul, Karine</au><au>Fattore, Giovanni</au><aucorp>The BURQOL-RD Research Network</aucorp><aucorp>BURQOL-RD Research Network</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Social/economic costs and health-related quality of life in patients with Prader-Willi syndrome in Europe</atitle><jtitle>The European journal of health economics</jtitle><stitle>Eur J Health Econ</stitle><addtitle>Eur J Health Econ</addtitle><date>2016-04-01</date><risdate>2016</risdate><volume>17</volume><issue>Suppl 1</issue><spage>S99</spage><epage>S108</epage><pages>S99-S108</pages><issn>1618-7598</issn><eissn>1618-7601</eissn><abstract>Objective 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. Methods 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. Results 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. Conclusion 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.</abstract><cop>Berlin/Heidelberg</cop><pub>Springer</pub><pmid>27038627</pmid><doi>10.1007/s10198-016-0788-z</doi><tpages>10</tpages></addata></record> |
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| subjects | Adolescent Adult Average total cost Caregivers Child Cost of Illness Costs Cross-Sectional Studies Direct costs Direct labor costs Direct service costs Economic Policy Economics Europe Female Health care Health care costs Health Care Costs - statistics & numerical data Health care expenditures Health Care Management Health Economics Hospital costs Humans Illnesses Labor productivity Male Medical research Medicine Medicine & Public Health Middle Aged Original Paper Patient Care - economics Patients Pharmacoeconomics and Health Outcomes Prader Willi syndrome Prader-Willi Syndrome - economics Prader-Willi Syndrome - psychology Productivity Public Finance Public Health Quality of Life Questionnaires Rare diseases Sick Leave - economics Sickness Impact Profile Socioeconomic Factors Surveys and Questionnaires United Kingdom Visual Analog Scale Young Adult |
| title | Social/economic costs and health-related quality of life in patients with Prader-Willi syndrome in Europe |
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