Universal versus targeted screening of infants for sickle cell disease: A cost-effectiveness analysis☆,☆☆,★
Section snippets
Structure of the Decision Model
Three newborn screening strategies for SCD were compared: (1) no screening, (2) targeted screening of African Americans, and (3) universal screening. Infants screened in the targeted screening or universal screening strategies had a confirmatory test if the screening test result was positive. Patients with SCD identified through screening (“SCD identified”) were treated with prophylactic penicillin and referred for parental education and comprehensive care. Patients with SCD who were not
Base-Case Analysis: Health Outcomes and Costs
Table III shows the number of infants screened, cases of SCD identified, total deaths, and total costs for each of the 3 strategies applied to a hypothetical cohort of 1,000,000 infants.
Strategy Infants screened per 1,000,000 Total costs discounted Total cases identified Total deaths No screening 0 $418,812 0 13 Targeted screening of African Americans 296,000* 1,398,388 316 7 Universal screening 990,000† 2,690,307 404 5 *Assumes 80% of African Americans and 20% of non-African Americans screened at
DISCUSSION
Our analysis compared health outcomes, costs, and incremental cost-effectiveness of universal screening for SCD with screening targeted to African Americans. We found that targeted screening compared with no screening was always cost-effective. Universal screening compared with targeted screening always identifies more infants with disease and prevents more deaths but incurs greater dollar cost.
Results of the sensitivity analysis showed the powerful influence of 2 factors, the proportion of
Acknowledgements
We thank Mr James McKinna for providing the screening cost data, Ms Marva Houston for providing follow-up educational and counseling costs, and Dr Robert Mauro for insightful suggestions.
References (34)
- et al.
Clinical events in the first decade in a cohort of infants with SCD
Blood
(1995) - et al.
Bacteremia in sickle hemoglobinopathies
J Pediatr
(1986) Anaphylaxis
Curr Prob Pediatr
(1992)- et al.
Clinical presentation of homozygous SCD
J Pediatr
(1985) - et al.
Bacteremia in children with sickle hemoglobin C disease and sickle beta+-thalassemia: Is prophylactic penicillin necessary?
J Pediatr
(1995) - et al.
Neonatal screening for SCD: a cost-effectiveness analysis
J Pediatr
(1991) - et al.
A cost-effectiveness evaluation of newborn hemoglobinopathy screening from the perspective of state health care systems
Early Hum Dev
(1996) - et al.
Prophylaxis with oral penicillin in children with sickle cell anemia: a randomized trial
N Engl J Med
(1986) Newborn screening for SCD and other hemoglobinopathies
JAMA
(1987)- et al.
Newborn screening fact sheets: sickle cell diseases: SS, SC, and S β-thalassemia
Pediatrics
(1996)
SCD: screening, diagnosis, management, and counseling in newborns and infants
The Council of Regional Networks for Genetic Services (CORN), National Newborn Screening Report-1992
Newborn screening for hemoglobinopathies in Colorado: the first ten years
Am J Dis Child
Newborn screening for SCD: 4 years of experience from California’s newborn screening program
J Pediatr Hematol Oncol
Bacterial meningitis and septicemia in SCD
Am J Dis Child
Clinicopathologic characteristics of septicemia in SCD
Am J Dis Child
Pneumococcal septicemia in children with sickle cell anemia: changing trend of survival
JAMA
Cited by (67)
Inherited disorders of hemoglobin: A review of old and new diagnostic methods
2024, Blood Cells, Molecules, and DiseasesGlobal migration and the changing distribution of sickle haemoglobin: A quantitative study of temporal trends between 1960 and 2000
2014, The Lancet Global HealthCitation Excerpt :Finally, whether the trends observed over the last half-century will continue in the coming years is unclear,60 and the implementation of specific health interventions should be carefully considered. Studies have been published about the cost-effectiveness of targeted versus universal screening programmes,43,61 and this effectiveness will depend on the individual context of each country. Nevertheless, increased awareness about this disorder and its wide distribution can only benefit patients and carriers.
Global epidemiology of Sickle haemoglobin in neonates: A contemporary geostatistical model-based map and population estimates
2013, The LancetCitation Excerpt :Additionally, the population growth rate is still relatively high in many countries with high frequencies of HbS so that, without prenatal diagnosis and avoidance strategies,12 existing allele frequencies will lead to a progressive increase in the absolute number of SS neonates for many generations. In high-income countries, screening programmes are now commonly needed to diagnose HbS in mixed populations to provide counselling and to prevent serious clinical complications.13 These factors strongly suggest that the number of AS and SS cases and associated care costs will substantially increase over the coming decades.12,14–16
A Markov model to analyze cost-effectiveness of screening for severe combined immunodeficiency (SCID)
2011, Molecular Genetics and MetabolismCitation Excerpt :Screening appeared cost-effective for test costs up to $50, if all other variables are constant. Our estimates are similar to ICER between no screening and universal screening for other newborn metabolic diseases (ICER of $5,800 to $42,000/life years) or sickle cell anemia (ICER of $13,000/life years saved) [28,29]. Incidence of disease and specificity of the screening test greatly influence cost-effectiveness.
A Newborn Screening Program for Sickle Cell Disease in Murcia (Spain)
2023, International Journal of Neonatal Screening
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Dr Panepinto is now a member of The Department of Pediatric Hematology/Oncology, Brown University School of Medicine, Providence, Rhode Island.
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Reprint requests: Peter A. Lane, MD, University of Colorado Health Sciences Center, Campus Box C222, 4200 East Ninth Ave, Denver, CO 80262.
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0022-3476/2000/$12.00 + 0 9/21/103569