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J Formos Med Assoc. 2015 Aug;114(8):722-8. doi: 10.1016/j.jfma.2013.06.017. Epub 2013 Aug 9.

Cost-effectiveness analysis of carrier and prenatal genetic testing for X-linked hemophilia.

Author information

1
Department of Pediatrics, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan, Taiwan.
2
Department of Public Health, College of Medicine, National Cheng Kung University, Tainan, Taiwan.
3
Department of Pediatrics, Chi-Mei Hospital, Tainan, Taiwan.
4
Department of Pediatrics, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan, Taiwan; Department of Pediatrics, Chi-Mei Hospital, Tainan, Taiwan. Electronic address: shiojean@gmail.com.

Abstract

BACKGROUND/PURPOSE:

Hemophilia involves a lifelong burden from the perspective of the patient and the entire healthcare system. Advances in genetic testing provide valuable information to hemophilia-affected families for family planning. The aim of this study was to analyze the cost-effectiveness of carrier and prenatal genetic testing in the health-economic framework in Taiwan.

METHODS:

A questionnaire was developed to assess the attitudes towards genetic testing for hemophilia. We modeled clinical outcomes of the proposed testing scheme by using the decision tree method. Incremental cost-effectiveness analysis was conducted, based on data from the National Health Insurance (NHI) database and a questionnaire survey.

RESULTS:

From the NHI database, 1111 hemophilic patients were identified and required an average medical expenditure of approximately New Taiwan (NT) $2.1 million per patient-year in 2009. By using the decision tree model, we estimated that 26 potential carriers need to be tested to prevent one case of hemophilia. At a screening rate of 79%, carrier and prenatal genetic testing would cost NT $85.9 million, which would be offset by an incremental saving of NT $203 million per year by preventing 96 cases of hemophilia. Assuming that the life expectancy for hemophilic patients is 70 years, genetic testing could further save NT $14.2 billion. Higher screening rates would increase the savings for healthcare resources.

CONCLUSION:

Carrier and prenatal genetic testing for hemophilia is a cost-effective investment in healthcare allocation. A case management system should be integrated in the current practice to facilitate patient care (e.g., collecting family pedigrees and providing genetic counseling).

KEYWORDS:

carrier testing; economic analysis; ethics; hemophilia; prenatal diagnosis

PMID:
23932837
DOI:
10.1016/j.jfma.2013.06.017
[Indexed for MEDLINE]
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