PEDIATRICS Vol. 107 No. 5 May 2001, pp. 1238

Analyzing the Cost of Neonatal Screening for Congenital Adrenal Hyperplasia

To the Editor.

The technical report on congenital adrenal hyperplasia (CAH) provided an informative and concise summary of several controversial issues.¹ It did not discuss cost except to incorrectly assert that reduction of false-positives significantly improves cost-effectiveness (the cost of rescreening false-positives is actually a small part of program costs). We feel that a detailed discussion of both cost and effect are essential to any position paper on screening because all screens have strong advocates, but resources used on one screen will be unavailable for another screen or health intervention. Economists call this opportunity cost.² There are standard methods for describing, measuring, and valuing costs, and the need for applying a systematic approach to an economic analysis is widely accepted.^3,4

We examined the opportunity cost of a neonatal screening program for CAH by comparing the cost and effect of newborn screening for CAH versus no screening in Texas, a state with 2 mandated screens.⁵ The costs of specimen testing, follow-up, and diagnostic evaluation during 1994 were included. We calculated the incremental cost (the extra dollar cost of the newborn screening program) and the incremental effect (the extra number of newborns diagnosed early because of the newborn screening program) when compared with a no-screening alternative. The analysis indicated that the total cost to diagnose 7 infants clinically (the no-screening alternative) was $79 187 ($11 312 per infant) in 1994 US dollars. The incremental cost of diagnosing 6 infants detected on the first screen was $691 013 ($115 169 per infant). The second screen identified 2 more infants with simple virilizing CAH at an incremental cost of $485 730 ($242 865 per infant). The incremental cost for the 2-screen program for 8 infants not yet recognized clinically was $1 176 743 ($147 093 per infant). Rescreening neonates with false-positive tests added little to the total, and decreasing the number of false-positives would not substantially reduce cost.

In a subsequent retrospective cohort study, we compared the Texas screened population with neonates in Arkansas and Oklahoma who had not been screened during a 5-year period.⁶ The incidences of classic CAH in the unscreened (5.75 per 100 000) and screened (6.26 per 100 000) were not significantly different. Screening did result in significantly earlier diagnoses (P = .01) and shorter hospital stays in males with salt-wasting CAH. Based on the cost data described above, we estimated that the cost of adding a single screen for CAH to an existing newborn screening program will be $257 735 per 100 000 newborns (1994 US dollars). Setting up a second screen de novo will be $918 839 per 100 000 newborns, mostly because of expenses related to specimen collection.

With an infinite amount of resources, health departments could fund every effective program regardless of the cost and the extent of benefit to society. However, we share a medical commons and so must choose between competing alternatives.⁷ The purpose of determining costs and outcomes of health care is not to dictate policy but rather to provide information that facilitates debate and informs decision-making.

Christine A. Brosnan
Patrick G. Brosnan
J. Michael Swint
^* School of Nursing
 School of Medicine
and ^§ School of Public Health
University of Texas Houston Health Science Center
Houston, TX 77030

REFERENCES

1. American Academy of Pediatrics Section on Endocrinology and Committee on Genetics Technical report: congenital adrenal hyperplasia. Pediatrics. 2000; 106:1511-1518 [Abstract/Free Full Text]
2. Russell LB, Siegel JE, Daniels N, Gold MR, Luce BR, Mandelblatt JS. Cost-effectiveness analysis as a guide to resource allocation in health: roles and limitations. In: Gold M, Siegel J, Russell L, Weinstein M, eds. Cost-Effectiveness in Health and Medicine. New York, NY: Oxford University Press; 1996
3. Drummond MF, O'Brien BJ, Stoddart GL, Torrance GW. Methods for the Economic Evaluation of Health Care Programmes. New York, NY: Oxford University Press; 1997
4. Weinstein MC, Siegel JE, Gold MR, Kamlet MS, Russell LB Recommendations of the panel on cost-effectiveness in health and medicine. JAMA. 1996; 276:1253-1258 [Abstract/Free Full Text]
5. Brosnan CA, Brosnan P, Therrell BL, A comparative cost analysis of newborn screening for classic congenital adrenal hyperplasia in Texas. Public Health Rep. 1998; 113:170-178 [Medline]
6. Brosnan PG, Brosnan CA, Kemp SF, Effect of newborn screening for congenital adrenal hyperplasia. Arch Pediatr Adolesc Med. 1999; 153:1272-1278 [Abstract/Free Full Text]
7. Hiatt H Protecting the medical commons: who is responsible? N Engl J Med. 1975; 293:235-241 [Abstract]

In Reply.

We agree with Brosnan et al that cost is an important consideration in the establishment of any screening program. However, we feel that to base the economic analysis only on the incremental cost of diagnosing affected infants in a screening program over the cost of clinical diagnosis in a nonscreening situation does not reflect the true implications of a screening program.

It is a well-recognized fact that the ability to clinically diagnose CAH in the newborn period is poor, especially in males.^1,2 In a substantial proportion of infants with 21-OH-deficient CAH, the first manifestation of the disease may be a life-threatening salt-wasting crisis occurring in the first weeks of life. In addition, virilization of female infants can be severe enough to result in incorrect gender assignment.^2,3 Newborn screening is aimed at identifying infants at risk for the development of life-threatening adrenal crisis and to prevent the incorrect male sex assignment of affected female infants with ambiguous genitalia.^4-7 A number of studiesincluding Brosnan et al's comparison of screened Texas newborns and nonscreened infants from 2 other states with no screening program mentioned in their letterhave demonstrated that screening results in significantly earlier diagnosis.^6-10 Further, it has been observed that the higher prevalence of CAH for girls than for boys that existed before screening has disappeared, indicating that those boys who might have died in the neonatal period are now surviving.⁶ It is evident that the potential savings of averting death and other complications that may profoundly affect physical and psychological well-being, albeit difficult to estimate, must also be taken into account when calculating the cost-benefits of these programs.

Jaime L. Frias
Lenore S. Levine
Sharon E. Oberfield
Sonya Pang
Janet Silverstein
for the AAP Ad Hoc Writing Committee

REFERENCES

1. Pang S, Clark A Congenital adrenal hyperplasia due to 21-hydroxylase deficiency: newborn screening and its relationship to the diagnosis and treatment of the disorder. Screening. 1993; 2:105-139
2. Allen DB, Hoffman GL, Fitzpatrick P, Improved precision of newborn screening for congenital adrenal hyperplasia using weight-adjusted criteria for 17-hydroxiprogesterone levels. J Pediatr. 1997; 130:128-133 [CrossRef][Medline]
3. Levine LS Congenital adrenal hyperplasia. Pediatr Rev. 2000; 21:139-170 [Free Full Text]
4. American Academy of Pediatrics, Committee on Genetics Newborn screening fact sheets. Pediatrics. 1996; 98:473-501 [Abstract/Free Full Text]
5. Pang S, Wallace M, Hofman L, Worldwide experience in newborn screening for congenital adrenal hyperplasia due to 21-hydroxylase deficiency. Pediatrics. 1996; 81:866-874 [Abstract/Free Full Text]
6. Thilén A, Nordenström A, Hegenfeldt L, von Döbeln U, Guthenberg C, Larsson A. Benefits of neonatal screening for congenital adrenal hyperplasia (21-hydroxylase deficiency) in Sweden. Pediatrics. 1998;101(4). URL: http://www.pediatrics.org/cgi/content/full/101/4/ell
7. Therrell BL, Berenbaum SA, Manter-Kapanke V, Results of screening 1.9 million Texas newborns for 21-hydroxylase-deficient congenital adrenal hyperplasia. Pediatrics. 1998; 101:583-590 [Abstract/Free Full Text]
8. Witchel SF, Nayak S, Suda-Hartman M, Newborn screening for 21-hydroxylase deficiency: results of CYP21 molecular genetic analysis. J Pediatr. 1997; 131:328-331 [CrossRef][Medline]
9. Brosnan PG, Brosnan CA, Kemp SF, Effects of newborn screening for congenital adrenal hyperplasia. Arch Pediatr Adolesc Med. 1999; 153:1272-1278
10. Brosnan CA, Brosnan PG Methodological issues in newborn screening evaluation with special reference to congenital adrenal hyperplasia. J Pediatr Endocrinol Metab. 2000; 13:1555-1562 [Medline]