Health economic modelling in Cystic Fibrosis: a systematic review
File(s)
Accepted version
Author(s)
Type
Journal Article
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.
Date Issued
2019-07-01
Date Acceptance
2019-01-21
Citation
Journal of Cystic Fibrosis, 2019, 18 (4), pp.452-460
ISSN
1569-1993
Publisher
Elsevier
Start Page
452
End Page
460
Journal / Book Title
Journal of Cystic Fibrosis
Volume
18
Issue
4
Copyright Statement
© 2019 European Cystic Fibrosis Society. Published by Elsevier B.V. All rights reserved. This manuscript is licensed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International Licence http://creativecommons.org/licenses/by-nc-nd/4.0/.
Sponsor
Cystic Fibrosis Trust
Identifier
https://www.ncbi.nlm.nih.gov/pubmed/30738801
PII: S1569-1993(19)30013-X
Grant Number
SRC 004
Subjects
Cost-effectiveness analysis
Cystic Fibrosis
Health Economics
Modelling
Publication Status
Published
Coverage Spatial
Netherlands
Date Publish Online
2019-02-07