In the article by Yoo and Grosse, entitled “The Cost Effectiveness of Screening Newborns for Congenital Adrenal Hyperplasia” [Public Health Genomics 2009;12:67–72], the cost-effectiveness results for newborn screening for congenital adrenal hyperplasia (CAH) do not accurately reflect the assumptions stated in Table 1 of the article. Mr. Orban Holdgate informed Dr. Grosse that the original cost-effectiveness model incorrectly applied the 80% reduction in mortality among infants with the salt-wasting (SW) form of CAH with screening to just a subset of infants with SW-CAH.
When the deterministic cost-effectiveness model was corrected for that error, the number of deaths from SW-CAH in the screening scenario was 3.2 times less and the number of averted deaths was 2.22 times greater. Consequently, the incremental cost-effectiveness ratio (ICER) reported in the article, USD 292,000 per life-year (LY) saved, was greatly overstated. A corrected estimate by Mr. Holdgate of the base-case ICER, assuming all assumptions reported in the original article, is USD 128,000 per LY saved, in 2005. All ICERs reported in the original Table 2 for the various sensitivity analyses should be similarly adjusted downwards. The results for the probabilistic cost-effectiveness analysis should be disregarded; Dr. Grosse was not able to replicate that analysis.
In qualitative terms, the original conclusion of Yoo and Grosse is not affected: newborn screening for CAH would not be considered cost-effective using a threshold value of USD 50,000 per LY saved. However, it might be considered cost-effective if a higher threshold value were used.
The correct Table 2 reads as follows:
Table 2.
CEA for newborn screening for CAH: year 2005, discount rate 3%
| Range | ICER (USD/life-year saved) |
|
|---|---|---|
| Traditional CEA | ||
| Base-case analysis | 128,200 | |
| Best-case analysis | 15,700 | |
| Worst-case analysis | 706,600 | |
| One-way sensitivity analysis of base-case analysis | ||
| (1) Cost per screening infant without follow-up | USD 2.3 | 80,900 |
| USD 6.0 | 175,600 | |
| (2) Cost of follow-up and confirmatory test per screen positive | USD 130 | 117,300 |
| USD 637 | 182,000 | |
| (3) Screen false-positive rate | 0.1% | 105,800 |
| 1.0% | 156,300 | |
| (4) Incidence of CAH | 1 in 25,000 | 182,300 |
| 1 in 12,000 | 84,700 | |
| (5) SW mortality without screening | 2.0% | 269,300 |
| 9.0% | 59,800 | |
| (6) Reduction in SW mortality with screening | 74% | 138,600 |
| 86% | 119,300 | |
CEA, cost-effectiveness analysis; CAH, congenital adrenal hyperplasia; SW, salt wasting; ICER, incremental cost-effectiveness ratio.