Skip to main content
Archives of Disease in Childhood. Fetal and Neonatal Edition logoLink to Archives of Disease in Childhood. Fetal and Neonatal Edition
. 1998 Nov;79(3):F161–F167. doi: 10.1136/fn.79.3.f161

Costing model for neonatal screening and diagnosis of haemoglobinopathies

E Cronin, C Normand, J Henthorn, M Hickman, S Davies
PMCID: PMC1720852  PMID: 10194984

Abstract

AIM—To compare the costs and cost effectiveness of universal and targeted screening for the haemoglobinopathies; to compare the cost of two laboratory methods; and to estimate the cost effectiveness of programmes at different levels of prevalence and mix of haemoglobinopathy traits.
METHODS—A retrospective review of laboratory and follow up records to establish workload and costs, and estimation of costs in a range of circumstances was made in a haematology department and sickle cell and thalassaemia centre, providing antenatal and neonatal screening programmes in Inner London. The costs for 47 948 babies, screened during 1994, of whom 25 had clinically significant haemoglobinopathies and 704 had haemoglobinopathy traits, were retrospectively assessed.
RESULTS—The average cost per baby tested (isoelectric focusing and high power liquid chromatography) was £3.51/£3.83 respectively; the cost per case of sickle cell disease identified (IEF/HPLC) was £6738/ £7355; the cost per trait identified (IEF/HPLC) was £234/£255; the cost per extra case of SCD and trait identified by universal programme varied.
CONCLUSIONS—IEF and HPLC are very similar in terms of average cost per test. At 16 traits/1000 and 0.5 SCD/1000 there was no significant identification cost difference between universal and targeted programmes. Below this prevalence, a targeted programme is cheaper but likely to miss cases of SCD. If targeted programmes were 90-99% effective, universal programmes would cease to be good value except at very high prevalence. Greater use of prenatal diagnosis, resulting in termination, and therefore fewer affected births, reduces the cost effectiveness of universal screening. Screening services should aim to cover a screened population which will generate a workload over 25 000 births a year, and preferably over 40000.



Full Text

The Full Text of this article is available as a PDF (144.8 KB).

Selected References

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

  1. Adjaye N., Bain B. J., Steer P. Prediction and diagnosis of sickling disorders in neonates. Arch Dis Child. 1989 Jan;64(1 Spec No):39–43. doi: 10.1136/adc.64.1_spec_no.39. [DOI] [PMC free article] [PubMed] [Google Scholar]
  2. Barton C. J., Watson A. Neonatal screening for haemoglobinopathies. BMJ. 1988 Jul 16;297(6642):200–200. doi: 10.1136/bmj.297.6642.200. [DOI] [PMC free article] [PubMed] [Google Scholar]
  3. Bergman A. B. Is universal neonatal hemoglobinopathy screening cost-effective? Arch Pediatr Adolesc Med. 1995 Apr;149(4):466–467. doi: 10.1001/archpedi.1995.02170160120022. [DOI] [PubMed] [Google Scholar]
  4. Delahunty T. Convenient screening for hemoglobin variants by using the Diamat HPLC system. Clin Chem. 1990 Jun;36(6):903–905. [PubMed] [Google Scholar]
  5. Eastman J. W., Wong R., Liao C. L., Morales D. R. Automated HPLC screening of newborns for sickle cell anemia and other hemoglobinopathies. Clin Chem. 1996 May;42(5):704–710. [PubMed] [Google Scholar]
  6. Emond A. M., Collis R., Darvill D., Higgs D. R., Maude G. H., Serjeant G. R. Acute splenic sequestration in homozygous sickle cell disease: natural history and management. J Pediatr. 1985 Aug;107(2):201–206. doi: 10.1016/s0022-3476(85)80125-6. [DOI] [PubMed] [Google Scholar]
  7. Gaston M. H., Verter J. I., Woods G., Pegelow C., Kelleher J., Presbury G., Zarkowsky H., Vichinsky E., Iyer R., Lobel J. S. Prophylaxis with oral penicillin in children with sickle cell anemia. A randomized trial. N Engl J Med. 1986 Jun 19;314(25):1593–1599. doi: 10.1056/NEJM198606193142501. [DOI] [PubMed] [Google Scholar]
  8. Githens J. H., Lane P. A., McCurdy R. S., Houston M. L., McKinna J. D., Cole D. M. Newborn screening for hemoglobinopathies in Colorado. The first 10 years. Am J Dis Child. 1990 Apr;144(4):466–470. doi: 10.1001/archpedi.1990.02150280088018. [DOI] [PubMed] [Google Scholar]
  9. Griffiths P. D., Mann J. R., Darbyshire P. J., Green A. Evaluation of eight and a half years of neonatal screening for haemoglobinopathies in Birmingham. Br Med J (Clin Res Ed) 1988 Jun 4;296(6636):1583–1585. doi: 10.1136/bmj.296.6636.1583. [DOI] [PMC free article] [PubMed] [Google Scholar]
  10. Harris M. S., Eckman J. R. Georgia's experience with newborn screening: 1981 to 1985. Pediatrics. 1989 May;83(5 Pt 2):858–860. [PubMed] [Google Scholar]
  11. Horn M. E., Dick M. C., Frost B., Davis L. R., Bellingham A. J., Stroud C. E., Studd J. W. Neonatal screening for sickle cell diseases in Camberwell: results and recommendations of a two year pilot study. Br Med J (Clin Res Ed) 1986 Mar 15;292(6522):737–740. doi: 10.1136/bmj.292.6522.737. [DOI] [PMC free article] [PubMed] [Google Scholar]
  12. Huisman T. H. Usefulness of cation exchange high performance liquid chromatography as a testing procedure. Pediatrics. 1989 May;83(5 Pt 2):849–851. [PubMed] [Google Scholar]
  13. Hurst D. Newborn screening for sickle cell disease and other hemoglobinopathies. Northern California's experience. Pediatrics. 1989 May;83(5 Pt 2):868–871. [PubMed] [Google Scholar]
  14. Kinney T. R., Sawtschenko M., Whorton M., Shearin J., Stine C., Hofman L., Safko R., Vitaglione T., Kaufman R. E. Newborn screening for sickle cell disease and other hemoglobinopathies. Techniques' comparison and report of the North Carolina experience. Pediatrics. 1989 May;83(5 Pt 2):843–848. [PubMed] [Google Scholar]
  15. Kleman K. M., Vichinsky E., Lubin B. H. Experience with newborn screening using isoelectric focusing. Pediatrics. 1989 May;83(5 Pt 2):852–854. [PubMed] [Google Scholar]
  16. Lee A., Thomas P., Cupidore L., Serjeant B., Serjeant G. Improved survival in homozygous sickle cell disease: lessons from a cohort study. BMJ. 1995 Dec 16;311(7020):1600–1602. doi: 10.1136/bmj.311.7020.1600. [DOI] [PMC free article] [PubMed] [Google Scholar]
  17. Rowley P. T., Huntzinger D. J. Newborn sickle cell screening. Benefits and burdens realized. Am J Dis Child. 1983 Apr;137(4):341–345. doi: 10.1001/archpedi.1983.02140300023007. [DOI] [PubMed] [Google Scholar]
  18. Sprinkle R. H., Hynes D. M., Konrad T. R. Is universal neonatal hemoglobinopathy screening cost-effective? Arch Pediatr Adolesc Med. 1994 May;148(5):461–469. doi: 10.1001/archpedi.1994.02170050019004. [DOI] [PubMed] [Google Scholar]
  19. Tsevat J., Wong J. B., Pauker S. G., Steinberg M. H. Neonatal screening for sickle cell disease: a cost-effectiveness analysis. J Pediatr. 1991 Apr;118(4 Pt 1):546–554. doi: 10.1016/s0022-3476(05)83375-x. [DOI] [PubMed] [Google Scholar]

Articles from Archives of Disease in Childhood. Fetal and Neonatal Edition are provided here courtesy of BMJ Publishing Group

RESOURCES