Skip to main content
The BMJ logoLink to The BMJ
. 1995 Dec 2;311(7018):1460–1464. doi: 10.1136/bmj.311.7018.1460

Cost effectiveness of antenatal screening for cystic fibrosis.

H S Cuckle 1, G A Richardson 1, T A Sheldon 1, P Quirke 1
PMCID: PMC2543729  PMID: 8520332

Abstract

OBJECTIVE--To estimate the cost effectiveness of different antenatal screening programmes for cystic fibrosis. SETTING--Antenatal clinics and general practices in the United Kingdom. DESIGN--Four components of the screening process were identified: information giving, DNA testing, genetic counselling, and prenatal diagnosis. The component costs were derived from the literature and from a pilot screening study in Yorkshire. The cost of a given screening programme was then obtained by summing the components according to the specific screening strategy adopted (sequential and couple), the proportion of carriers detected by the DNA test, and the uptake of screening. Baseline assumptions were made about the proportion with missing information on carrier status from previous pregnancies (20%), the proportion changing partners between pregnancies (20%), and the uptake of prenatal diagnosis (100%). Sensitivity analysis was performed by varying these assumptions. MAIN OUTCOME MEASURE--Cost per affected pregnancy detected. RESULTS--Under the baseline assumptions sequential screening costs between pounds 40,000 and pounds 90,000 per affected pregnancy detected, depending on the carrier detection rate and uptake. Couple screening was more expensive, ranging from pounds 46,000 to pounds 104,000. From the sensitivity analysis a 10% change in the assumed proportion with missing information from a previous pregnancy alters the cost by pounds 4000; a 10% change in the proportion with new partners has a similar effect but only for couple screening; and cost will change directly in proportion to the uptake of prenatal diagnosis. CONCLUSIONS--While economic analysis cannot determine screening policy, the paper provides the NHS with the information on cost effectiveness needed to inform decisions on the introduction of a screening service for cystic fibrosis.

Full text

PDF
1460

Selected References

These references are in PubMed. This may not be the complete list of references from this article.

  1. Asch D. A., Patton J. P., Hershey J. C., Mennuti M. T. Reporting the results of cystic fibrosis carrier screening. Am J Obstet Gynecol. 1993 Jan;168(1 Pt 1):1–6. doi: 10.1016/s0002-9378(12)90875-3. [DOI] [PubMed] [Google Scholar]
  2. Cheadle J., Myring J., al-Jader L., Meredith L. Mutation analysis of 184 cystic fibrosis families in Wales. J Med Genet. 1992 Sep;29(9):642–646. doi: 10.1136/jmg.29.9.642. [DOI] [PMC free article] [PubMed] [Google Scholar]
  3. Garber A. M., Fenerty J. P. Costs and benefits of prenatal screening for cystic fibrosis. Med Care. 1991 May;29(5):473–489. doi: 10.1097/00005650-199105000-00007. [DOI] [PubMed] [Google Scholar]
  4. Ginsberg G., Blau H., Kerem E., Springer C., Kerem B. S., Akstein E., Greenberg A., Kolumbos A., Abeliovich D., Gazit E. Cost-benefit analysis of a national screening programme for cystic fibrosis in an Israeli population. Health Econ. 1994 Jan-Feb;3(1):5–23. doi: 10.1002/hec.4730030104. [DOI] [PubMed] [Google Scholar]
  5. Harris H., Scotcher D., Hartley N., Wallace A., Craufurd D., Harris R. Cystic fibrosis carrier testing in early pregnancy by general practitioners. BMJ. 1993 Jun 12;306(6892):1580–1583. doi: 10.1136/bmj.306.6892.1580. [DOI] [PMC free article] [PubMed] [Google Scholar]
  6. Kerem B., Rommens J. M., Buchanan J. A., Markiewicz D., Cox T. K., Chakravarti A., Buchwald M., Tsui L. C. Identification of the cystic fibrosis gene: genetic analysis. Science. 1989 Sep 8;245(4922):1073–1080. doi: 10.1126/science.2570460. [DOI] [PubMed] [Google Scholar]
  7. Lieu T. A., Watson S. E., Washington A. E. The cost-effectiveness of prenatal carrier screening for cystic fibrosis. Obstet Gynecol. 1994 Dec;84(6):903–912. [PubMed] [Google Scholar]
  8. Livingstone J., Axton R. A., Gilfillan A., Mennie M., Compton M., Liston W. A., Calder A. A., Gordon A. J., Brock D. J. Antenatal screening for cystic fibrosis: a trial of the couple model. BMJ. 1994 Jun 4;308(6942):1459–1462. doi: 10.1136/bmj.308.6942.1459. [DOI] [PMC free article] [PubMed] [Google Scholar]
  9. Mennie M. E., Gilfillan A., Compton M., Curtis L., Liston W. A., Pullen I., Whyte D. A., Brock D. J. Prenatal screening for cystic fibrosis. Lancet. 1992 Jul 25;340(8813):214–216. doi: 10.1016/0140-6736(92)90476-j. [DOI] [PubMed] [Google Scholar]
  10. Miedzybrodzka Z. H., Dean J. C., Russell G., Friend J. A., Kelly K. F., Haites N. E. Prevalence of cystic fibrosis mutations in the Grampian region of Scotland. J Med Genet. 1993 Apr;30(4):316–317. doi: 10.1136/jmg.30.4.316. [DOI] [PMC free article] [PubMed] [Google Scholar]
  11. Miedzybrodzka Z. H., Hall M. H., Mollison J., Templeton A., Russell I. T., Dean J. C., Kelly K. F., Marteau T. M., Haites N. E. Antenatal screening for carriers of cystic fibrosis: randomised trial of stepwise v couple screening. BMJ. 1995 Feb 11;310(6976):353–357. doi: 10.1136/bmj.310.6976.353. [DOI] [PMC free article] [PubMed] [Google Scholar]
  12. Miedzybrodzka Z. H., Yin Z., Kelly K. F., Haites N. E. Evaluation of laboratory methods for cystic fibrosis carrier screening: reliability, sensitivity, specificity, and costs. J Med Genet. 1994 Jul;31(7):545–550. doi: 10.1136/jmg.31.7.545. [DOI] [PMC free article] [PubMed] [Google Scholar]
  13. Miedzybrodzka Z., Semper J., Shackley P., Abdalla M., Donaldson C. Stepwise or couple antenatal carrier screening for cystic fibrosis?: women's preferences and willingness to pay. J Med Genet. 1995 Apr;32(4):282–283. doi: 10.1136/jmg.32.4.282. [DOI] [PMC free article] [PubMed] [Google Scholar]
  14. Mooney G., Lange M. Ante-natal screening: what constitutes 'benefit'? Soc Sci Med. 1993 Oct;37(7):873–878. doi: 10.1016/0277-9536(93)90140-y. [DOI] [PubMed] [Google Scholar]
  15. Piggott M., Wilkinson P., Bennett J. Implementation of an antenatal serum screening programme for Down's syndrome in two districts (Brighton and Eastbourne). The Brighton and Eastbourne Down's Syndrome Screening Group. J Med Screen. 1994 Jan;1(1):45–49. doi: 10.1177/096914139400100109. [DOI] [PubMed] [Google Scholar]
  16. Riordan J. R., Rommens J. M., Kerem B., Alon N., Rozmahel R., Grzelczak Z., Zielenski J., Lok S., Plavsic N., Chou J. L. Identification of the cystic fibrosis gene: cloning and characterization of complementary DNA. Science. 1989 Sep 8;245(4922):1066–1073. doi: 10.1126/science.2475911. [DOI] [PubMed] [Google Scholar]
  17. Robson M., Abbott J., Webb K., Dodd M., Walsworth-Bell J. A cost description of an adult cystic fibrosis unit and cost analyses of different categories of patients. Thorax. 1992 Sep;47(9):684–689. doi: 10.1136/thx.47.9.684. [DOI] [PMC free article] [PubMed] [Google Scholar]
  18. Rommens J. M., Iannuzzi M. C., Kerem B., Drumm M. L., Melmer G., Dean M., Rozmahel R., Cole J. L., Kennedy D., Hidaka N. Identification of the cystic fibrosis gene: chromosome walking and jumping. Science. 1989 Sep 8;245(4922):1059–1065. doi: 10.1126/science.2772657. [DOI] [PubMed] [Google Scholar]
  19. Shackley P., McGuire A., Boyd P. A., Dennis J., Fitchett M., Kay J., Roche M., Wood P. An economic appraisal of alternative pre-natal screening programmes for Down's syndrome. J Public Health Med. 1993 Jun;15(2):175–184. [PubMed] [Google Scholar]
  20. Sheldon T. A., Simpson J. Appraisal of a new scheme for prenatal screening for Down's syndrome. BMJ. 1991 May 11;302(6785):1133–1136. doi: 10.1136/bmj.302.6785.1133. [DOI] [PMC free article] [PubMed] [Google Scholar]
  21. Shrimpton A. E., McIntosh I., Brock D. J. The incidence of different cystic fibrosis mutations in the Scottish population: effects on prenatal diagnosis and genetic counselling. J Med Genet. 1991 May;28(5):317–321. doi: 10.1136/jmg.28.5.317. [DOI] [PMC free article] [PubMed] [Google Scholar]
  22. Super M., Schwarz M. J., Malone G., Roberts T., Haworth A., Dermody G. Active cascade testing for carriers of cystic fibrosis gene. BMJ. 1994 Jun 4;308(6942):1462–1467. doi: 10.1136/bmj.308.6942.1462. [DOI] [PMC free article] [PubMed] [Google Scholar]
  23. Wald N. J. Couple screening for cystic fibrosis. Lancet. 1991 Nov 23;338(8778):1318–1319. doi: 10.1016/0140-6736(91)92605-2. [DOI] [PubMed] [Google Scholar]
  24. Wald N. J., George L. M., Wald N. M., Mackenzie I. Couple screening for cystic fibrosis. Lancet. 1993 Nov 20;342(8882):1307–1308. doi: 10.1016/0140-6736(93)92403-g. [DOI] [PubMed] [Google Scholar]

Articles from BMJ : British Medical Journal are provided here courtesy of BMJ Publishing Group

RESOURCES