Top

Published in:

Open Access 01-06-2015 | Research article

Social and economic costs and health-related quality of life in non-institutionalised patients with cystic fibrosis in the United Kingdom

Authors: Aris Angelis, Panos Kanavos, Julio López-Bastida, Renata Linertová, Elena Nicod, Pedro Serrano-Aguilar, BURQOL-RD Research Network

Published in: BMC Health Services Research | Issue 1/2015

Abstract

Background

This study aimed to determine the societal economic burden and health-related quality of life (HRQOL) of cystic fibrosis (CF) patients in the UK.

Methods

A bottom-up cost-of-illness, cross-sectional, retrospective analysis of 74 patients was conducted aiming to estimate the economic impact of CF. Data on demographic characteristics, health resource utilisation, informal care, productivity losses and HRQOL were collected from questionnaires completed by patients or their caregivers. HRQOL was measured with the EuroQol 5-domain (EQ-5D) instrument.

Results

Using unit costs for 2012 we found that the average annual cost for a CF patient was €48,603, with direct health care costs amounting to €20,854 (42.9 % of total costs), direct non-health care costs being €21,528 (44.3 %) and indirect costs attributable to productivity losses being €6,222 (12.8 %). On average, the largest expenditures by far were accounted for by informal care (44.1 %), followed by medications (14.5 %), acute hospitalisations (13.9 %), early retirement (9.1 %) and outpatient and primary health care visits (7.9 %). Sharp differences existed depending on whether CF patients were in need of caregiver help (€76,271 versus €26,335). In adult CF patients, mean EQ-5D index scores were 0.64 (0.93 in the general population) and mean EQ-5D visual analogue scale scores were 62.23 (86.84 in the general population); among caregivers, these scores were 0.836 and 80.85, respectively.

Discussion

Our analysis highlights the importance of the economic and quality of life consequences of CF from a societal perspective. The results highlight that beyond conventional costs such as acute hospitalisations, medication and outpatient and primary care visits, indirect costs related to informal care and early retirement, have significant societal implications. Similarly, our analysis showed that the average EQ-5D index score of adult CF patients was significantly lower than in the general population, an indication that a methodological bias may exist in using the latter in economic analyses.

Conclusion

CF poses a significant cost burden on UK society, with non-health care and indirect costs representing 57 % of total average costs, and HRQOL being considerably lower than in the general population.

Available only for authorised users

Orphanet: Cystic fibrosis. [http://www.orpha.net]. Accessed on 24 May 2013.

NHS Newborn Blood SPOT Screening Programme. [http://newbornbloodspot.screening.nhs.uk/cf]. Accessed on 24 May 2013.

Rowe SM, Miller S, Sorscher EJ. Cystic fibrosis. N Engl J Med. 2005;352(19):1992–2001.CrossRefPubMed

Roberts HJ, Hubbard R. Trends in bronchiectasis mortality in England and Wales. Respir Med. 2010;104(7):981–5.CrossRefPubMed

Ratjen F, Doring G. Cystic fibrosis. Lancet. 2003;361(9358):681–9.CrossRefPubMed

Bellis G, Cazes MH, Parant A, Gaimard M, Travers C, Le Roux E, et al. Cystic fibrosis mortality trends in France. J Cyst Fibros. 2006;6:179–86.CrossRefPubMed

Dodge JA, Lewis PA. Cystic fibrosis is no longer an important cause of childhood death in the UK. Arch Dis Child. 2005;90(5):547–a.CrossRefPubMedPubMedCentral

Joish VN, Spilsbury-Cantalupo M, Operschall E, Luong B, Boklage S. Economic burden of non-cystic fibrosis bronchiectasis in the first year after diagnosis from a US health plan perspective. Appl Health Econ Health Policy. 2013;11(3):299–304.CrossRefPubMed

van Gool K, Norman R, Delatycki MB, Hall J, Massie J. Understanding the costs of care for cystic fibrosis: an analysis by age and health state. Value Health. 2013;16(2):345–55.CrossRefPubMed

10.

Kianifar HR, Bakhshoodeh B, Hebrani P, Behdani F. Quality of life in cystic fibrosis children. Iran J Pediatr. 2013;23(2):149–53.PubMedPubMedCentral

11.

Tluczek A, Becker T, Grieve A, Laxova A, Rock MJ, Gershan WM, et al. Health-related quality of life in children and adolescents with cystic fibrosis: convergent validity with parent-reports and objective measures of pulmonary health. J Dev Behav Pediatr. 2013;34(4):252–61.CrossRefPubMedPubMedCentral

12.

Tepper LA, Utens EM, Caudri D, Bos AC, Gonzalez-Graniel K, Duivenvoorden HJ, et al. Impact of bronchiectasis and trapped air on quality of life and exacerbations in cystic fibrosis. Eur Respir J. 2013;42(2):371–9.CrossRefPubMed

13.

Weiner JR, Toy EL, Sacco P, Duh MS. Costs, quality of life and treatment compliance associated with antibiotic therapies in patients with cystic fibrosis: a review of the literature. Expert Opin Pharmacother. 2008;9(5):751–66.CrossRefPubMed

14.

Groeneveld IF, Sosa ES, Pérez M, Fiuza-Luces C, Gonzalez-Saiz L, Gallardo C, et al. Health-related quality of life of Spanish children with cystic fibrosis. Qual Life Res. 2012;21:1837–45.CrossRefPubMedPubMedCentral

15.

Dewitt EM, Grussemeyer CA, Friedman JY, Dinan MA, Lin L, Schulman KA, et al. Resource use, costs, and utility estimates for patients with cystic fibrosis with mild impairment in lung function: analysis of data collected alongside a 48-week multicenter clinical trial. Value Health. 2012;15:277–83.CrossRefPubMed

16.

Gu, Y., Perez, S.G., Massie, J., van Gool, K. Cost of care for cystic fibrosis: an investigation of cost determinants using national registry data. Eur J Health Econ in press.

17.

Angelis A, Tordrup D, Kanavos P. Socio-Economic Burden of Rare Diseases: A Systematic Review of Cost of Illness Evidence. Health Policy 2015, DOI: 10.1016/j.healthpol.2014.12.016

18.

Linertová R, Serrano-Aguilar P, Posada-de-la-Paz M, Hens-Pérez M, Kanavos P, Taruscio D, et al. Delphi approach to select rare diseases for a European representative survey. The BURQOL-RD study. Health Policy. 2012;108(1):19–26.CrossRefPubMed

19.

Lopez-Bastida J, Monton-Alvarez F, Serrano-Aguilar P, Perestelo-Perez L. The social-economic costs in patients with ataxias cerebelosas in Spain. Mov Disord. 2008;23:212–7.CrossRefPubMed

20.

Payment by Results in the NHS: tariff for 2012 to 2013. [https://www.gov.uk/government/publications/confirmation-of-payment-by-results-pbr-arrangements-for-2012-13]. Accessed on 24 May 2013.

21.

Curtis, L. Unit Costs of Health and Social Care 2012. [http://www.pssru.ac.uk/project-pages/unit-costs/2012/]. Accessed on 24 May 2013.

22.

NHS reference costs 2012 to 2013. [https://www.gov.uk/government/publications/nhs-reference-costs-2012-to-2013]. Accessed on 24 May 2013.

23.

NHS Drug Tariff. [http://www.ppa.org.uk/ppa/edt_intro.htm]. Accessed on 24 May 2013.

24.

British National Formulary. [http://www.bnf.org/bnf/index.htm]. Accessed on 24 May 2013.

25.

McDaid D. Estimating the costs of informal care for people with Alzheimer’s disease: methodological and practical challenges. Int J of Geriatr Psychiatry. 2001;16:400–5.CrossRef

26.

Hodgson TA, Meiners MR. Cost-of-illness methodology: a guide to assessment practices and procedures. Milbank Mem Fund Q. 1982;60:429–91.CrossRef

27.

Annual Survey of Hours and Earnings, 2012. [http://www.ons.gov.uk/ons/rel/ashe/annual-survey-of-hours-and-earnings/2012-provisional-results/index.html]. Accessed on 24 May 2013.

28.

Brooks R. EuroQol: the current state of play. Health Policy. 1996;37:53–72.CrossRefPubMed

29.

Collin C, Wade DT, Davies S, Horne V. The Barthel ADL Index: a reliability study. Int Disabil Stud. 1988;10(2):61–3.CrossRefPubMed

30.

Hérbert R, Bravo G, Préville M. Reliability, validity, and reference values of the Zarit Burden Interview for assessing informal caregivers of community-dwelling older persons with dementia. Can J Aging. 2000;494–507.

31.

Dolan P. Modeling valuations for EuroQol health states. Med Care. 1997;35:1095–108.CrossRefPubMed

32.

Kind P, Hardman G, Macran S. UK Population Norms for EQ-5D. Discussion paper 172, The University of York Centre of Health Economics.

33.

Heimeshoff M, Hollmeyer H, Schreyögg J, Tiemann O, Staab D. Cost of illness of cystic fibrosis in Germany. Pharmacoeconomics. 2012;30:763–77.CrossRefPubMed

34.

Robson M, Abbott J, Webb K, Dodd M, Walsworth-Bell J. A cost description of an adult cystic fibrosis unit and cost analyses of different categories of patients. Thorax. 1992;47:684–9.CrossRefPubMedPubMedCentral

35.

Baltin CT, Smaczny C, Wagner TO. Drug treatment of cystic fibrosis – cost patterns and savings potential for outpatient treatment. Med Klin (Munich). 2010;105(12):887–900.CrossRef

36.

Tur-Kaspa I, Aljadeff G, Rechitsky S, Grotjan HE, Verlinsky Y. PGD for all cystic fibrosis carrier couples: novel strategy for preventive medicine and cost analysis. Reprod Biomed Online. 2010;21(2):186–95.CrossRefPubMed

37.

Huot L, Durieu I, Bourdy S, Ganne C, Bellon G, Colin C, et al. Evolution of costs of care for cystic fibrosis patients after clinical guidelines implementation in a French network. J Cyst Fibros. 2008;7(5):403–8.CrossRefPubMed

38.

Eidt-Koch D, Mittendorf T, Greiner W. Cross-sectional validity of the EQ-5D-Y as a generic health outcome instrument in children and adolescents with cystic fibrosis in Germany. BMC Pediatr. 2009;9:55.CrossRefPubMedPubMedCentral

39.

Ghatnekar O, Glenngård AH, Olofsson S, Persson U. A literature review of instruments for measuring health-related quality of life in rare diseases. The Swedish Institute for Health Economics. Internal report of BURQOL-RD; April 2011.

Title: Social and economic costs and health-related quality of life in non-institutionalised patients with cystic fibrosis in the United Kingdom
Authors: Aris Angelis
Panos Kanavos
Julio López-Bastida
Renata Linertová
Elena Nicod
Pedro Serrano-Aguilar
BURQOL-RD Research Network
Publication date: 01-06-2015
Publisher: BioMed Central
Published in: BMC Health Services Research / Issue 1/2015
Electronic ISSN: 1472-6963
DOI: https://doi.org/10.1186/s12913-015-1061-3

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Social and economic costs and health-related quality of life in non-institutionalised patients with cystic fibrosis in the United Kingdom

Abstract

Background

Methods

Results

Discussion

Conclusion

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Background

Methods

Results

Discussion

Conclusion

Please log in to get access to this content

Other articles of this Issue 1/2015

Consensus building to improve the physical health of people with severe mental illness: a qualitative outcome mapping study

Assessment of primary care facilities for cardiovascular disease preparedness in Madhya Pradesh, India

Facilitators and barriers to implementing electronic referral and/or consultation systems: a qualitative study of 16 health organizations

Validation of the Adherence Barriers Questionnaire – an instrument for identifying potential risk factors associated with medication-related non-adherence

The effect of cost-sharing in private health insurance on the utilization of health care services between private insurance purchasers and non-purchasers: a study of the Korean health panel survey (2008–2012)

Health and use of health services of people who are homeless and at risk of homelessness who receive free primary health care in Dublin