Abstract
Introduction: Cystic Fibrosis (CF) is a heritable chronic condition. Due to the genetic and progressive nature of CF, a number of interventions are available for the condition. In the United Kingdom (U.K.) average annual cost of CF treatment is between €49,000 to €76,000 (2012) per patient [1]. A review of health economic modelling studies is warranted to provide decision makers and researchers with an in depth understanding of modelling practices in CF and guidance for future research. Methods: Online searches were performed in the 5 databases, studies were included if they were: a) Model based economic evaluation for management of Cystic Fibrosis. Articles were restricted to English language only, but no restriction was applied on publication year. Results: Nine studies were reviewed, most were Markov cohort models. Models evaluated pharmaceutical interventions and drug adherence. Modelling structure was consistent across most articles and a range of sources were used to populate the models. Cost and utility data were based on different sources and elicitation methods respectively. The majority of models failed to incorporate significant health events which impact both cost and disease progression. Conclusion: In our review we observed a lack of, application of European Medicines Agency (EMA) guidelines for clinical trial endpoints, model structure justifications and lastly, health-related quality of life derived utility information around important clinical events. Future work around conceptual modelling of CF progression, utility valuation of significant health events and meeting EMA guidelines for trial reporting is encouraged.
Original language | English |
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Pages (from-to) | 452-460 |
Number of pages | 9 |
Journal | Journal of Cystic Fibrosis |
Volume | 18 |
Issue number | 4 |
Early online date | 7 Feb 2019 |
DOIs | |
Publication status | Published - 2 Jul 2019 |
Keywords
- Cost-effectiveness analysis
- Cystic Fibrosis
- Health Economics
- Modelling
Profiles
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Tracey Sach
- Norwich Medical School - Honorary Professorial Fellow
- Health Economics - Member
Person: Honorary, Research Group Member
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David Turner
- Norwich Medical School - Associate Professor in Health Economics
- Population Health - Member
- Health Economics - Member
- Health Services and Primary Care - Member
- Norwich Clinical Trials Unit - Member
Person: Research Group Member, Research Centre Member, Academic, Teaching & Research
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Jennifer Whitty
- Norwich Medical School - Honorary Professor
- Norwich Institute for Healthy Aging - Member
- Health Economics - Member
- Health Services and Primary Care - Member
- Respiratory and Airways Group - Member
Person: Honorary, Research Group Member, Research Centre Member