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Original Paper

The Cost Effectiveness of Screening Newborns for Congenital Adrenal Hyperplasia

Yoo B.K.a · Grosse S.D.b

Author affiliations

aUniversity of Rochester, School of Medicine and Dentistry, Department of Community and Preventive Medicine, Division of Health Services Research and Policy, Rochester, N.Y.; bCenters for Disease Control and Prevention (CDC), National Center on Birth Defects and Developmental Disabilities, Atlanta, Ga., USA

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Public Health Genomics 2009;12:67–72

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Article / Publication Details

First-Page Preview
Abstract of Original Paper

Received: February 27, 2008
Accepted: June 18, 2008
Published online: September 14, 2008
Issue release date: November 2008

Number of Print Pages: 6
Number of Figures: 1
Number of Tables: 2

ISSN: 1662-4246 (Print)
eISSN: 1662-8063 (Online)

For additional information: https://www.karger.com/PHG

Abstract

Objective: To assess the cost effectiveness of newborn screening for congenital adrenal hyperplasia (CAH) in the U.S. newborn population. Methods: We constructed a decision model to estimate the incremental cost-effectiveness ratio (ICER) of CAH screening compared to a strategy of no screening. Two types of cost effectiveness analyses (CEA) were conducted to measure ICER as net cost per life year (LY): (1) traditional CEA with sensitivity and scenario analyses, and (2) probabilistic CEA. Results: ICERs for (1) base-case analysis in traditional CEA and (2) probabilistic CEA were USD 292,000 and USD 255,700 per LY saved in 2005 USD, respectively. ICERs were particularly sensitive to assumptions regarding the mortality rate for the salt wasting type of CAH, in a range from 2 to 9%. The ICERs for best-case and worst-case scenarios were USD 30,900 and USD 2.9 million per LY saved, respectively. Conclusions: Using common benchmarks for cost effectiveness, our results indicate that CAH screening would be unlikely to be considered cost effective unless assumptions favorable to screening were adopted, although it could meet economic criteria used to assess U.S. regulatory policies. A limitation is that the analysis excludes outcomes such as correct assignment of gender and quality of life.

© 2008 S. Karger AG, Basel


References

  1. New MI: An update of congenital adrenal hyperplasia. Ann N Y Acad Sci 2004;1038:14–43.
  2. National Newborn Screening and Genetics Resource Center: National Newborn Screening Report – 2000. Austin, TX, The National Newborn Screening and Genetics Resource Center, February 2003.
  3. van der Kamp HJ, Wit JM: Neonatal screening for congenital adrenal hyperplasia. Eur J Endocrinol 2004;151(suppl 3):U71–U75.
  4. American College of Medical Genetics Newborn Screening Expert Group: Newborn screening: toward a uniform screening panel and system – executive summary. Pediatrics 2006;117:S296–S307.
  5. National Newborn Screening and Genetics Resource Center: U.S. National Screening Status Report. http://genes-r-us.uthscsa.edu/nbsdisorders.pdf (accessed January 8, 2008).
  6. Loeber JG: Neonatal screening in Europe; the situation in 2004. J Inherit Metab Dis 2007;30:430–438.
  7. Grosse SD, Van Vliet G: How many deaths can be prevented by newborn screening for congenital adrenal hyperplasia? Horm Res 2007;67:284–291.
  8. Minutti CZ, Lacey JM, Magera MJ, Hahn SH, McCann M, Schulze A, Cheillan D, Dorche C, Chace DH, Lymp JF, Zimmerman D, Rinaldo P, Matern D: Steroid profiling by tandem mass spectrometry improves the positive predictive value of newborn screening for congenital adrenal hyperplasia. J Clin Endocrinol Metab 2004;89:3687–3693.
  9. Carroll AE, Downs SM: Comprehensive cost-utility analysis of newborn screening strategies. Pediatrics 2006;117: S287–S295.
  10. Centers for Disease Control and Prevention (CDC) National Center for Health Statistics (NCHS). http://www.cdc.gov/nchs/fastats/lifexpec.htm (accessed December 6, 2007).
  11. Therrell BL Jr, Berenbaum SA, Manter-Kapanke V, Simmank J, Korman K, Prentice L, Gonzalez J, Gunn S: Results of screening 1.9 million Texas newborns for 21-hydroxylase-deficient congenital adrenal hyperplasia. Pediatrics 1998;101:583–590.
  12. Brosnan CA, Brosnan P, Therrell BL, Slater CH, Swint JM, Annegers JF, Riley WJ: A comparative cost analysis of newborn screening for classic congenital adrenal hyperplasia in Texas. Public Health Rep 1998;113:170–178.
  13. National Newborn Screening Information System: Reports for Congenital Adrenal Hyperplasia (CAH). http://www2.uthscsa.edu/nnsis/ (accessed December 22, 2007).
  14. Thil’en A, Nordenstrom A, Hagenfeldt L, von Dobeln U, Guthenberg C, Larsson A: Benefits of neonatal screening for congenital adrenal hyperplasia (21-hydroxylase deficiency) in Sweden. Pediatrics 1998;101:E11.
  15. Kaye CI, Committee on Genetics, Accurso F, La Franchi S, Lane PA, Hope N, Sonya P, Schaefer GB, Lloyd-Puryear MA: Newborn screening fact sheets. Pediatrics 2006;118:e934–e963.
  16. Kovács J, Votava F, Heinze G, Sólyom J, Lebl J, Pribilincová Z, Frisch H, Battelino T, Waldhauser F, Middle European Workshop on Paediatric Endocrinology-Congenital Adrenal Hyperplasia Study Group: Lessons from 30 years of clinical diagnosis and treatment of congenital adrenal hyperplasia in five middle European countries. J Clin Endocrinol Metab 2001;86:2958–2964.
  17. Van der Kamp HJ, Noordam K, Elvers B, Van Baarle M, Otten BJ, Verkerk PH: Newborn screening for congenital adrenal hyperplasia in the Netherlands. Pediatrics 2001;108:1320–1324.
  18. Thilen A, Larsson A: Congenital adrenal hyperplasia in Sweden 1969–1986. Prevalence, symptoms and age at diagnosis. Acta Paediatr Scand 1990;79:168–175.
  19. Brosnan PG, Brosnan CA, Kemp SF, Domek DB, Jelley DH, Blackett PR, Riley WJ: Effect of newborn screening for congenital adrenal hyperplasia. Arch Pediatr Adolesc Med 1999;153:1272–1278.
  20. Strnadova KA, Votava F, Lebl J, Mühl A, Item C, Bodamer OA, Torresani T, Bouska I, Waldhauser F, Sperl W: Prevalence of congenital adrenal hyperplasia among sudden infant death in the Czech Republic and Austria. Eur J Pediatr 2007;166:1–4.
  21. Grosse SD, Van Vliet G: Outcomes in CAH: Need for evidence-based estimates. Horm Res 2007;68:203.
  22. Luce BR, Manning WG, Siegel JE, Lipscomb J: Estimating costs in cost-effectiveness analysis; in Gold MR, Siegel JE, Russell LB, Weinstein MC (eds): Cost-Effectiveness in Health and Medicine. New York, Oxford University Press, 1996, pp 176–213.
  23. Meltzer D: Accounting for future costs in medical cost-effectiveness analysis. J Health Econ 1997;16:33–64.
  24. Neumann PJ, Sandberg EA, Bell CM, Stone PW, Chapman RH: Are pharmaceuticals cost-effective? A review of the evidence. Health Aff (Millwood) 2000;19:92–109.
  25. Grosse SD: Assessing cost effectiveness in health care: the history of the USD 50,000 per QALY threshold. Exp Rev Pharmacoecon Outcomes Res 2008;8:165–178.
  26. Maciosek MV, Coffield AB, Edwards NM, Flottemesch TJ, Goodman MJ, Solberg LI: Priorities among effective clinical preventive services: results of a systematic review and analysis. Am J Prev Med 2006;31:52–61.
  27. Ubel PA, Hirth RA, Chernew ME, Fendrick AM: What is the price of life and why doesn’t it increase at the rate of inflation? Arch Intern Med 2003;163:1637–1641.
  28. Aldy JE, Viscusi WK: Age differences in the value of statistical life: revealed preference evidence. Rev Environ Econ Policy 2007;1:241–260.
  29. Jaaskelainen J, Voutilainen R: Long-term outcome of classical 21-hydroxylase deficiency: diagnosis, complications and quality of life. Acta Paediatr 2000;89:183–187.

Article / Publication Details

First-Page Preview
Abstract of Original Paper

Received: February 27, 2008
Accepted: June 18, 2008
Published online: September 14, 2008
Issue release date: November 2008

Number of Print Pages: 6
Number of Figures: 1
Number of Tables: 2

ISSN: 1662-4246 (Print)
eISSN: 1662-8063 (Online)

For additional information: https://www.karger.com/PHG


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