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- Title
Screening for hereditary hemochromatosis in siblings and children of affected patients. A cost-effectiveness analysis.
- Authors
El-Serag, Hashem B.; Inadomi, John M.; El-Serag, H B; Inadomi, J M; Kowdley, K V
- Abstract
<bold>Background: </bold>Screening for hereditary hemochromatosis is traditionally done by using serum iron studies. However, mutation analysis of the hemochromatosis-associated HFE gene has recently become available.<bold>Objective: </bold>To compare the cost-effectiveness of no screening with four screening strategies that incorporate HFE gene testing or serum iron studies.<bold>Design: </bold>Cost-effectiveness analysis.<bold>Data Sources: </bold>Published literature.<bold>Target Population: </bold>Siblings and children of an affected proband.<bold>Time Horizon: </bold>Lifetime from 10 years of age (children) or 45 years of age (siblings).<bold>Perspective: </bold>Societal.<bold>Intervention: </bold>1) Serum iron studies. 2) Gene testing of the proband. If the proband is homozygous (C82Y+/+), the spouse undergoes gene testing; if he or she is heterozygous (C82Y+/-), the children undergo gene testing. 3) Gene testing of the proband; if he or she is homozygous, relatives undergo gene testing. 4) Direct gene testing of relatives.<bold>Outcome Measures: </bold>Cost per life-year saved and incremental cost-effectiveness ratio.<bold>Results Of Base-case Analysis: </bold>In children, HFE gene testing of the proband was the most cost-effective strategy for screening one child (incremental cost-effectiveness ratio, $508 per life-year saved). HFE gene testing of the proband followed by testing of the spouse was the most cost-effective strategy for screening two or more children (incremental cost-effectiveness ratio, $3665 per life-year saved). In siblings, all screening strategies were dominant compared with no screening. Strategies using HFE gene testing were less costly than serum iron studies.<bold>Results Of Sensitivity Analysis: </bold>Despite varying the prevalence of mutations and regardless of the cost of the genetic test in one- and two-way sensitivity analyses, HFE gene testing remained cost-effective.<bold>Conclusions: </bold>HFE gene testing for the C282Y mutation is a cost-effective method of screening relatives of patients with hereditary hemochromatosis.
- Subjects
HEMOCHROMATOSIS diagnosis; HUMAN chromosome abnormality diagnosis
- Publication
Annals of Internal Medicine, 2000, Vol 132, Issue 4, p261
- ISSN
0003-4819
- Publication type
journal article
- DOI
10.7326/0003-4819-132-4-200002150-00003