The cost-effectiveness of screening for hereditary hemochromatosis in Germany: a remodeling study
Abstract
Objective Genetic tests for hereditary hemochromatosis (HH) are currently included in the German ambulatory care reimbursement scheme but only for symptomatic individuals and the offspring of HH patients. This study synthesizes the most current evidence to examine whether screening in the broader population is cost-effective and to identify the best choice of initial and follow-up screening tests. Methods A probabilistic decision-analytic model was constructed to calculate cost per life year gained (LYG) for HH screening among male Caucasians aged 30. Three strategies were considered in both the general population and male offspring of HH patients: phenotypic (transferrin saturation, TS), genotypic (C282Y mutation), and sequential (genotype if TS is elevated) screening. Results The incremental cost-effectiveness of sequential screening among male offspring, sequential population-wide screening, and genotypic screening is 41000, 124000, and 161000 Eero/LYG, respectively. All other strategies were subject to simple or extended dominance. The results are subject to high uncertainty. The most influential parameters in the deterministic one-way sensitivity analysis are discounting of life years gained and the adherence of patients to preventive phlebotomy. Discussion The current German policy of only screening at-risk individuals is consistent with health economic decision making based on typically accepted thresholds. However, conducting the DNA test after the first elevated TS r…
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STMs used to evaluate screening strategies consider two types: one-time screening or repeated interval screening.
“STMs used to evaluate screening strategies [16,17] consider two types: one-time screening, (e.g., of newborns [18] or genetic screening [19 -21]) or repeated (interval) screening (e.g., for cancer [22][23][24][25] or HIV [26,27]).”
From State-Transition Modeling: A Report of the ISPOR-SMDM Modeling Good Research Practices Task Force-3 (2012) · cites this paper as [21]
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Screening for hereditary haemochromatosis within families and beyond
McCune, C. A., Ravine, D., Worwood, M., Jackson, H. A., Evans, H. M., & Hutton, D. (2003) · The Lancet · doi:10.1016/s0140-6736(03)14963-x · Cited by 21 · see citing works
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State-Transition Modeling: A Report of the ISPOR-SMDM Modeling Good Research Practices Task Force-3
Siebert, U., Alagoz, O., Bayoumi, A. M., Jahn, B., Owens, D. K., Cohen, D. J., & Kuntz, K. M. (2012) · Value in Health · doi:10.1016/j.jval.2012.06.014 · Cited by 495 · see citing works
State-Transition Modeling: A Report of the ISPOR-SMDM Modeling Good Research Practices Task Force–3
Siebert, U., Alagoz, O., Bayoumi, A. M., Jahn, B., Owens, D. K., Cohen, D. J., & Kuntz, K. M. (2012) · Medical Decision Making · doi:10.1177/0272989x12455463 · Cited by 348 · see citing works
Grosse, S. D., Gurrin, L. C., Bertalli, N. A., & Allen, K. J. (2018) · Genetics in Medicine · doi:10.1038/gim.2017.121 · Cited by 76 · see citing works
- Authors
- Rogowski, W. H.
- Affiliations
- Helmholtz Munich (DE)
- Corresponding author
- Rogowski, Wolf H.
- ORCID
- Rogowski, Wolf H.
- Venue
- Medical Decision Making
- Publisher
- SAGE Publications
- Published
- 2008-10-03
- Volume
- 29
- Issue
- 2
- Pages
- 224-238
- ISSN
- 0272-989X (ISSN-L)
- 1552-681X
- OpenAlex
- W2070059936
- Cited by
- 42
- References
- 1
- Citations by year
- 2025: 1 · 2024: 2 · 2022: 1 · 2020: 2 · 2018: 1 · 2017: 3 · 2016: 5 · 2015: 5 · 2014: 1 · 2013: 2 · 2012: 4
- FWCI
- 3.26
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Medical Decision Making (publishedVersion)
- PubMed (publishedVersion)
- RePEc: Research Papers in Economics (submittedVersion)
- Site cant be reached (submittedVersion)
- Topics
- Health Sciences › Medicine › Hematology › Iron Metabolism and Disorders
- Social Sciences › Economics, Econometrics and Finance › Economics and Econometrics › Health Systems, Economic Evaluations, Quality of Life
- Health Sciences › Medicine › Genetics › Hemoglobinopathies and Related Disorders
- Keywords
- hereditary hemochromatosis, population-wide screening, cost-effectiveness, C282Y mutation, transferrin saturation, sequential screening, cost per life-year gained, health policy, discount rate, sensitivity analysis
- MeSH terms
- Adult, Cost-Benefit Analysis, Decision Support Techniques, Genetic Testing, Genotype, Germany, Hemochromatosis, Humans, Male, Phenotype, Sensitivity and Specificity, Models, Econometric
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