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Cost-Effectiveness of Different Population Screening Strategies for Hereditary Haemochromatosis in Australia

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Abstract

Introduction

Amongst populations of northern European ancestry, HFE-associated haemochromatosis is a common genetic disorder characterised by iron overload. In the absence of treatment, excess iron is stored in parenchymal tissues, causing morbidity and mortality. Population screening programmes may increase early diagnosis and reduce associated disease. No contemporary health economic evaluation has been published for Australia. The objective of this study was to identify cost-effective screening strategies for haemochromatosis in the Australian setting.

Methods

A Markov model using probabilistic decision analysis was developed comparing four adult screening strategies: the status quo (cascade and incidental screening), genotyping with blood and buccal samples and transferrin saturation followed by genotyping (TfS). Target populations were males (30 years) and females (45 years) of northern European ancestry. Cost-effectiveness was estimated from the government perspective over a lifetime horizon.

Results

All strategies for males were cost-effective compared to the status quo. The incremental costs (standard deviation) associated with genotyping (blood) were AUD7 (56), TfS AUD15 (45) and genotyping (buccal) AUD63 (56), producing ICERs of AUD1673, 4103 and 15,233/quality-adjusted life-year (QALY) gained, respectively. For females, only the TfS strategy was cost-effective, producing an ICER of AUD10,195/QALY gained. Approximately 3% of C282Y homozygotes were estimated to be identified with the status quo approach, compared with 40% with the proposed screening strategies.

Conclusion

This model estimated that genotyping and TfS strategies are likely to be more cost-effective screening strategies than the status quo.

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Acknowledgements

The authors would like to acknowledge the support provided by the Centre for Health Economics Research and Evaluation at the University of Technology Sydney. In particular, we would like to thank Professor Jane Hall, Associate Professor Stephen Goodall and Dr. Rob Anderson (University of Exeter Medical School).

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Authors and Affiliations

  1. Menzies Institute for Medical Research, Medical Science 2 Building, University of Tasmania, 17 Liverpool St, Private Bag 23, Hobart, TAS, 7000, Australia

    Barbara de Graaff, Amanda Neil, Lei Si, Kristy Sanderson & Andrew J. Palmer

  2. School of Medicine, Medical Science 1 Building, University of Tasmania, 17 Liverpool St, Private Bag 68, Hobart, TAS, 7000, Australia

    Kwang Chien Yee

  3. Melbourne School of Population and Global Health, University of Melbourne, 207 Bouverie St, Carlton, VIC, 3053, Australia

    Lyle Gurrin

Authors
  1. Barbara de Graaff
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Corresponding author

Correspondence to Andrew J. Palmer.

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Funding

No financial support was provided for this project nor for the preparation of the manuscript. Barbara de Graaff was supported by a Ph.D. Australian Postgraduate Research Award scholarship provided by the Australian Government: this body did not have any role in the study.

Support

Support was provided by members of the Centre for Health Economics Research and Evaluation at the University of Technology Sydney [Professor Jane Hall, Associate Professor Stephen Goodall and Dr. Rob Anderson (University of Exeter)] by providing preliminary work on an economic model for haemochromatosis. None of the CHERE work is included in this manuscript.

Conflict of interest

Barbara de Graaff, Amanda Neil, Kristy Sanderson, Lei Si, Kwang Chien Yee, Lyle Gurrin and Andrew J. Palmer have no conflicts of interest to declare that are directly relevant to the content of this review.

Author contributions

Barbara de Graaff planned and constructed the model, conducted the model simulations and sensitivity analyses, and prepared the manuscript. Lei Si assisted with construction and validation of the model and assisted with preparation of the manuscript. Amanda Neil and Kristy Sanderson contributed to constructing the model, interpreting results and assisted with preparation of the manuscript. Kwang Chien Yee assisted with assessing the model for face validity and assisted with preparation of the manuscript. Lyle Gurrin assisted with the construction and validity of the model, and assisted with preparation of the manuscript. Andrew Palmer assisted with planning and constructing the model, conducting simulations and sensitivity analyses, and assisted with preparation of the manuscript.

Appendix

Appendix

See Table 6 and Fig. 2.

Table 6 One-way sensitivity analyses, total government costs
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de Graaff, B., Neil, A., Si, L. et al. Cost-Effectiveness of Different Population Screening Strategies for Hereditary Haemochromatosis in Australia. Appl Health Econ Health Policy 15, 521–534 (2017). https://doi.org/10.1007/s40258-016-0297-3

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