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Health Economic Evaluations of Hemochromatosis Screening and Treatment: A Systematic Review

Author

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  • Malvina Hoxha

    (Catholic University Our Lady of Good Counsel)

  • Visar Malaj

    (University of Tirana
    CERGE-EI Foundation Teaching Fellow)

  • Bruno Zappacosta

    (Catholic University Our Lady of Good Counsel)

Abstract

Background Hereditary hemochromatosis (HH) is an autosomal recessive disorder that leads to iron overload and multiorgan failure. Objectives The aim of this systematic review was to provide up-to-date evidence of all the current data on the costs and cost effectiveness of screening and treatment for HH. Methods We searched PubMed, Cochrane Library, National Health Service Economic Evaluation Database (NHSEED), Cost-Effectiveness Analysis Registry (CEA Registry), Health Technology Assessment Database (HTAD), Centre for Reviews and Dissemination (CRD), and Econlit until April 2023 with no date restrictions. Articles that reported cost-utility, cost-description, cost-minimization, cost-effectiveness, or cost-benefit analyses for any kind of management (drugs, screening, etc.) were included in the study. Patients with HH, their siblings, or individuals suspected of having HH were included in the study. All screening and treatment strategies were included. Two authors assessed the quality of evidence related to screening (either phenotype or genotype screening) and treatment (phlebotomy and electrophoresis). Narrative synthesis was used to analyse the similarities and differences between the respective studies. Results Thirty-nine papers were included in this study. The majority of the studies reported both the cost of phenotype screening, including transferrin saturation (TS), serum ferritin, and liver biopsy, and the cost of genotype screening (HFE screening, C282Y mutation). Few studies reported the cost for phlebotomy and erythrocytapheresis treatment. Data revealed that either phenotype or genotype screening were cost effective compared with no screening. Treatment studies concluded that erythrocytapheresis might be a cost-effective therapy compared with phlebotomy. Conclusions Economic studies on either the screening, or treatment strategy for HH patients should be performed in more countries. We suggest that cost-effectiveness studies on the role of deferasirox in HH should be carried out as an alternative therapy to phlebotomy.

Suggested Citation

  • Malvina Hoxha & Visar Malaj & Bruno Zappacosta, 2024. "Health Economic Evaluations of Hemochromatosis Screening and Treatment: A Systematic Review," PharmacoEconomics - Open, Springer, vol. 8(2), pages 147-170, March.
    • Handle: RePEc:spr:pharmo:v:8:y:2024:i:2:d:10.1007_s41669-023-00463-6
    DOI: 10.1007/s41669-023-00463-6
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    References listed on IDEAS

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    1. Drummond, Michael F. & Sculpher, Mark J. & Torrance, George W. & O'Brien, Bernie J. & Stoddart, Greg L., 2005. "Methods for the Economic Evaluation of Health Care Programmes," OUP Catalogue, Oxford University Press, edition 3, number 9780198529453, Decembrie.
    2. Barbara Graaff & Amanda Neil & Kristy Sanderson & Lei Si & Kwang Yee & Andrew Palmer, 2015. "A Systematic Review and Narrative Synthesis of Health Economic Studies Conducted for Hereditary Haemochromatosis," Applied Health Economics and Health Policy, Springer, vol. 13(5), pages 469-483, October.
    3. Barbara Graaff & Amanda Neil & Lei Si & Kwang Chien Yee & Kristy Sanderson & Lyle Gurrin & Andrew J. Palmer, 2017. "Cost-Effectiveness of Different Population Screening Strategies for Hereditary Haemochromatosis in Australia," Applied Health Economics and Health Policy, Springer, vol. 15(4), pages 521-534, August.
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