Carrier Screening for Cystic Fibrosis

Author:

Asch David A.1234,Hershey John C.2356,Dekay Michael L.35,Pauly Mark V.36,Patton James P.3,Jedrziewski M. Kathryn,Frei Frances7,Giardine Rose8,Kant Jeffrey A.9,Mennuti Michael T.8

Affiliation:

1. Veterans Affairs Medical Center, Philadelphia, Pennsylvania

2. Division of General Internal Medicine, University of Pennsylvania School of Medicine, Philadelphia

3. Leonard Davis Institute of Health Economics, University of Pennsylvania

4. Center for Bioethics, University of Pennsylvania School of Medicine

5. Department of Operations and Information Management, The Wharton School, University of Pennsylvania

6. Department of Health Care Systems, The Wharton School, University of Pennsylvania

7. Simon School of Business, University of Rochester

8. Department of Obstetrics and Gynecology, University of Pennsylvania School of Medicine

9. Department of Pathology and Laboratory Medicine, University of Pennsylvania School of Medicine

Abstract

Objectives. To evaluate the costs and clinical effects of 16 alternative strategies for cystic fibrosis (CF) carrier screening in the reproductive setting; and to test the sensitivity of the results to assumptions about cost and detection rate, stakeholder perspective, DNA test specificity, chance of nonpaternity, and couples' reproductive plans. Method. Cost-effectiveness analysis. Results. A sequential screening strategy had the lowest cost per CF birth avoided. In this strategy, the first partner was screened with a standard test that identifies 85% of carriers. The second partner was screened with an expanded test if the first partner's screen was positive. This strategy identified 75% of anticipated CF births at a cost of $367,000 each. This figure does not include the lifetime medical costs of caring for a patient with CF, and it assumes that couples who identify a pregnancy at risk will choose to have prenatal diagnosis and termination of affected pregnancies. The cost per CF birth identified is approximately half this figure when couples plan two children. Conclusions. The cost-effectiveness of CF carrier screening depends greatly on couples' reproductive plans. CF carrier screening is most cost-effective when it is performed sequentially, when the information is used for more than one pregnancy, and when the intention of the couple is to identify and terminate affected pregnancies. These conclusions are important for policy considerations regarding population-based screening for CF, and may also have important implications for screening for less common diseases.

Publisher

SAGE Publications

Subject

Health Policy

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