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Population Screening for Hereditary Haemochromatosis in Australia: Construction and Validation of a State-Transition Cost-Effectiveness Model


de Graaff, B and Si, L and Neil, AL and Yee, KC and Sanderson, K and Gurrin, LC and Palmer, AJ, Population Screening for Hereditary Haemochromatosis in Australia: Construction and Validation of a State-Transition Cost-Effectiveness Model, PharmacoEconomics - Open, 1, (1) pp. 37-51. ISSN 2509-4254 (2017) [Refereed Article]

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Copyright The Author(s) 2016. Licensed under Creative Commons Attribution 4.0 International (CC BY 4.0)

DOI: doi:10.1007/s41669-016-0005-0


Introduction: HFE-associated haemochromatosis, the most common monogenic disorder amongst populations of northern European ancestry, is characterised by iron overload. Excess iron is stored in parenchymal tissues, leading to morbidity and mortality. Population screening programmes are likely to improve early diagnosis, thereby decreasing associated disease. Our aim was to develop and validate a health economics model of screening using utilities and costs from a haemochromatosis cohort.

Methods: A state-transition model was developed with Markov states based on disease severity. Australian males (aged 30 years) and females (aged 45 years) of northern European ancestry were the target populations. The screening strategy was the status quo approach in Australia; the model was run over a lifetime horizon. Costs were estimated from the government perspective and reported in 2015 Australian dollars ($A); costs and qualityadjusted life-years (QALYs) were discounted at 5% annually. Model validity was assessed using goodness-of- fit analyses. Second-order Monte-Carlo simulation was used to account for uncertainty in multiple parameters.

Results: For validity, the model reproduced mortality, life expectancy (LE) and prevalence rates in line with published data. LE for C282Y homozygote males and females were 49.9 and 40.2 years, respectively, slightly lower than population rates. Mean (95% confidence interval) QALYS were 15.7 (7.723.7) for males and 14.4 (6.722.1) for females. Mean discounted lifetime costs for C282Y homozygotes were $A22,737 (367085,793) for males and $A13,840 (133567,377) for females. Sensitivity analyses revealed discount rates and prevalence had the greatest impacts on outcomes.

Conclusion: We have developed a transparent, validated health economics model of C282Y homozygote haemochromatosis. The model will be useful to decision makers to identify cost-effective screening strategies.

Item Details

Item Type:Refereed Article
Keywords:health economics, modelling, screening, haemochromatosis
Research Division:Health Sciences
Research Group:Health services and systems
Research Field:Health services and systems not elsewhere classified
Objective Division:Health
Objective Group:Public health (excl. specific population health)
Objective Field:Behaviour and health
UTAS Author:de Graaff, B (Dr Barbara de Graaff)
UTAS Author:Si, L (Mr Lei Si)
UTAS Author:Neil, AL (Associate Professor Amanda Neil)
UTAS Author:Yee, KC (Dr Kwang Yee)
UTAS Author:Sanderson, K (Associate Professor Kristy Sanderson)
UTAS Author:Palmer, AJ (Professor Andrew Palmer)
ID Code:112633
Year Published:2017 (online first 2016)
Deposited By:Menzies Institute for Medical Research
Deposited On:2016-11-21
Last Modified:2020-01-30
Downloads:153 View Download Statistics

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