Hostname: page-component-745bb68f8f-s22k5 Total loading time: 0 Render date: 2025-01-25T22:46:44.077Z Has data issue: false hasContentIssue false
  • English
  • Français

Comparing the clinical and economic effects of clinical examination, pulse oximetry, and echocardiography in newborn screening for congenital heart defects: A probabilistic cost-effectiveness model and value of information analysis

Published online by Cambridge University Press:  01 April 2007

Ingolf Griebsch ,
Rachel L. Knowles ,
Jacqueline Brown ,
Catherine Bull ,
Christopher Wren  and
Carol A. Dezateux
Ingolf Griebsch
Affiliation:
University of Bristol
Rachel L. Knowles
Affiliation:
UCL Institute of Child Health
Jacqueline Brown
Affiliation:
University of Bristol
Catherine Bull
Affiliation:
Great Ormond Street Hospital for Children
Christopher Wren
Affiliation:
Freemann Hospital
Carol A. Dezateux
Affiliation:
UCL Institute of Child Health
Get access Rights & Permissions [Opens in a new window]

Abstract

Objectives: Congenital heart defects (CHD) are an important cause of death and morbidity in early childhood, but the effectiveness of alternative newborn screening strategies in preventing the collapse or death—before diagnosis—of infants with treatable but life-threatening defects is uncertain. We assessed their effectiveness and efficiency to inform policy and research priorities.

Methods: We compared the effectiveness of clinical examination alone and clinical examination with either pulse oximetry or screening echocardiography in making a timely diagnosis of life-threatening CHD or in diagnosing clinically significant CHD. We contrasted their cost-effectiveness, using a decision-analytic model based on 100,000 live births, and assessed future research priorities using value of information analysis.

Results: Clinical examination alone, pulse oximetry, and screening echocardiography achieved 34.0, 70.6, and 71.3 timely diagnoses per 100,000 live births, respectively. This finding represents an additional cost per additional timely diagnosis of £4,894 and £4,496,666 for pulse oximetry and for screening echocardiography. The equivalent costs for clinically significant CHD are £1,489 and £36,013, respectively. Key determinants of cost-effectiveness are detection rates and screening test costs. The false-positive rate is very high with screening echocardiography (5.4 percent), but lower with pulse oximetry (1.3 percent) or clinical examination alone (.5 percent).

Conclusions: Adding pulse oximetry to clinical examination is likely to be a cost-effective newborn screening strategy for CHD, but further research is required before this policy can be recommended. Screening echocardiography is unlikely to be cost-effective, unless the detection of all clinically significant CHD is considered beneficial and a 5 percent false-positive rate acceptable.

Keywords

Newborn screeningCongenital heart defectsCost-effectivenessDecision-analytic modelValue of information analysis
Type
GENERAL ESSAYS
Information
International Journal of Technology Assessment in Health Care , Volume 23 , Issue 2 , April 2007 , pp. 192 - 204
Copyright
Copyright © Cambridge University Press 2007

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)

References

REFERENCES

1.Acharya, G, Sitras, V, Maltau, JM et al. , Major congenital heart disease in Northern Norway: Shortcomings of pre- and postnatal diagnosis. Acta Obstet Gynecol Scand. 2004; 83: 1124–1129.CrossRefGoogle ScholarPubMed
2.Ainsworth, S, Wyllie, JP, Wren, C. Prevalence and clinical significance of cardiac murmurs in neonates. Arch Dis Child Fetal Neonatal Ed. 1999; 80: F43–F45.CrossRefGoogle ScholarPubMed
3.Alberman, E. The National Down Syndrome Cytogenetic Register (NDSCR). J Med Screen. 2002; 9: 97–98.CrossRefGoogle ScholarPubMed
4.Arlettaz, R, Archer, N, Wilkinson, AR. Natural history of innocent heart murmurs in newborn babies: Controlled echocardiographic study. Arch Dis Child Fetal Neonatal Ed. 1998; 78: F166–F170.CrossRefGoogle ScholarPubMed
5.Bakr, AF, Habib, HS. Combining pulse oximetry and clinical examination in screening for congenital heart disease. Pediatr Cardiol. 2005; 26: 832–835.CrossRefGoogle ScholarPubMed
6.Boneva, RS, Botto, LD, Moore, CA et al. , Mortality associated with congenital heart defects in the United States: Trends and racial disparities, 1979-1997. Circulation. 2001; 103: 2376–2381.CrossRefGoogle ScholarPubMed
7.Bove, EL, Bull, C, Stark, J et al. , Congenital heart disease in the neonate: Results of surgical treatment. Arch Dis Child. 1983; 58: 137–141.CrossRefGoogle ScholarPubMed
8.Bricker, L, Garcia, J, Henderson, J et al. , Ultrasound screening in pregnancy: A systematic review of the clinical effectiveness, cost-effectiveness and women's views. Health Technol Assess. 2000; 4: i–193.CrossRefGoogle ScholarPubMed
9.Briggs, AH. Handling uncertainty in economic evaluation. In: Drummond, MFMcGuire, A, eds. Economic evaluation in health care. Oxford, New York: Oxford University Press; 2001: 172–214.Google Scholar
10.Brown, KL, Ridout, DA, Goldman, AP, Hoskote, A, Penny, DJ. Risk factors for long intensive care unit stay after cardiopulmonary bypass in children. Crit Care Med. 2003; 31: 28–33.CrossRefGoogle ScholarPubMed
11.Bull, C. Current and potential impact of fetal diagnosis on prevalence and spectrum of serious congenital heart disease at term in the UK. British Paediatric Cardiac Association. Lancet. 1999; 354: 1242–1247.CrossRefGoogle ScholarPubMed
12.Chilcott, J, Brennan, A, Booth, A, Karnon, J, Tappenden, P. The role of modelling in prioritising and planning clinical trials. Health Technol Assess. 2003; 7: 1–125.CrossRefGoogle ScholarPubMed
13.Clancy, RR, McGaurn, SA, Wernovsky, G et al. , Preoperative risk-of-death prediction model in heart surgery with deep hypothermic circulatory arrest in the neonate. J Thorac Cardiovasc Surg. 2000; 119: 347–357.CrossRefGoogle ScholarPubMed
14.Claxton, K. The irrelevance of inference: A decision-making approach to the stochastic evaluation of health care technologies. J Health Econ. 1999; 18: 341–364.CrossRefGoogle Scholar
15.Claxton, K, Neumann, PJ, Araki, S, Weinstein, MC. Bayesian value-of-information analysis. An application to a policy model of Alzheimer's disease. Int J Technol Assess Health Care. 2001; 17: 38–55.CrossRefGoogle ScholarPubMed
16.Claxton, K, Cohen, JT, Neumann, PJ. When is evidence sufficient? Health Aff (Millwood). 2005; 24: 93–101.CrossRefGoogle ScholarPubMed
17.Clemen, RT, Winkler, RL. Combining probability distributions from experts in risk analysis. Risk Anal. 1999; 19: 187–203.CrossRefGoogle Scholar
18.de Wahl, GA, Mellander, M, Sunnegardh, J, Sandberg, K, Ostman-Smith, I. Screening for duct-dependant congenital heart disease with pulse oximetry: A critical evaluation of strategies to maximize sensitivity. Acta Paediatr. 2005; 94: 1590–1596.CrossRefGoogle Scholar
19.Eurocat Surveillance Data: Table B3 1996-1999. Available at: www.biomedicalweb.biz/eurocat. Accessed 2004.Google Scholar
20.Fenwick, E, Claxton, K, Sculpher, M. Representing uncertainty: The role of cost-effectiveness acceptability curves. Health Econ. 2001; 10: 779–787.CrossRefGoogle ScholarPubMed
21.Glazener, CM, Ramsay, CR, Campbell, MK et al. , Neonatal examination and screening trial (NEST): A randomised, controlled, switchback trial of alternative policies for low risk infants. BMJ. 1999; 318: 627–631.CrossRefGoogle Scholar
22.Griebsch, I, Coast, J, Brown, J. Quality-adjusted life-years lack quality in pediatric care: A critical review of published cost-utility studies in child health. Pediatrics. 2005; 115: e600–e614.CrossRefGoogle ScholarPubMed
23.Hall, DM. The role of the routine neonatal examination. BMJ. 1999; 318: 619–620.CrossRefGoogle ScholarPubMed
24.Hoffman, JI, Kaplan, S. The incidence of congenital heart disease. [Review] [124 refs]. J Am Coll Cardiol. 2002; 39: 1890–1900.CrossRefGoogle Scholar
25.Hoke, TR, Donohue, PK, Bawa, PK et al. , Oxygen saturation as a screening test for critical congenital heart disease: A preliminary study. Pediatr Cardiol. 2002; 23: 403–409.CrossRefGoogle ScholarPubMed
26.Hunink, MG, Glasziou, PP, Siegel, JE et al. , Decision making in health and medicine. Cambridge: Cambridge University Press; 2001.Google Scholar
27.Knowles, R, Griebsch, I, Dezateux, C et al. , Newborn screening for congenital heart defects: A systematic review and cost-effectiveness analysis. Health Technol Assess. 2005; 9: 1–168.CrossRefGoogle ScholarPubMed
28.Koppel, RI, Druschel, CM, Carter, T et al. , Effectiveness of pulse oximetry screening for congenital heart disease in asymptomatic newborns. Pediatrics. 2003; 111: 451–455.CrossRefGoogle ScholarPubMed
29.Kwon, C. FPM. The magnitude and challenge of false-positive newborn screening test results. Arch Pediatr Adolesc Med. 2000; 154: 714–718.CrossRefGoogle Scholar
30.Netten, A, Rees, T, Harrison, G. Unit costs of health and social care 2001. The University of Kent, Canterbury: Personal Social Services Research Unit (PSSRU).Google Scholar
31.Office for National Statistics. 2003 Death registrations in England and Wales 2002. Health Stat Q. 18: 57–64.Google Scholar
32.Petrou, S. Methodological issues raised by preference-based approaches to measuring the health status of children. Health Econ. 2003; 12: 697–702.CrossRefGoogle ScholarPubMed
33.Reich, JD, Miller, S, Brogdon, B et al. , The use of pulse oximetry to detect congenital heart disease. J Pediatr. 2003; 142: 268–272.CrossRefGoogle ScholarPubMed
34.Richmond, S, Reay, G, Abu, HM. Routine pulse oximetry in the asymptomatic newborn. Arch Dis Child Fetal Neonatal Ed. 2002; 87: F83–F88.CrossRefGoogle ScholarPubMed
35.Roberts, TE. Economic evaluation and randomised controlled trial of extracorporeal membrane oxygenation: UK collaborative trial. The Extracorporeal Membrane Oxygenation Economics Working Group. BMJ. 1998; 317: 911–915.CrossRefGoogle ScholarPubMed
36.Rosano, A, Botto, LD, Botting, B, Mastroiacovo, P. Infant mortality and congenital anomalies from 1950 to 1994: An international perspective. J Epidemiol Community Health. 2000; 54: 660–666.CrossRefGoogle ScholarPubMed
37.Sands, A, Craig, B, Mulholland, C et al. , Echocardiographic screening for congenital heart disease: A randomized study. J Perinat Med. 2002; 30: 307–312.CrossRefGoogle ScholarPubMed
38.Spiegelhalter, DJ, Harris, NL, Bull, K, Franklin, RCG. Empirical evaluation of prior beliefs about frequencies: Methodology and a case study in congenital heart disease. J Am Stat Assoc. 1994; 89: 435–443.CrossRefGoogle Scholar
39.Stinnett, AA, Mullahy, J. Net health benefits: A new framework for the analysis of uncertainty in cost-effectiveness analysis. Med Decis Making. 1998; 18: S68–S80.CrossRefGoogle ScholarPubMed
40.Tilford, JM. Cost-effectiveness analysis and emergency medical services for children: Issues and applications. Ambul Pediatr. 2002; 2 (Suppl): 330–336.2.0.CO;2>CrossRefGoogle ScholarPubMed
41.Tulloh, RM, Tansey, SP, Parashar, K et al. , Echocardiographic screening in neonates undergoing surgery for selected gastrointestinal malformations. Arch Dis Child Fetal Neonatal Ed. 1994; 70: F206–F208.CrossRefGoogle ScholarPubMed
42.Wren, C, O'Sullivan, JJ. Survival with congenital heart disease and need for follow up in adult life. Heart. 2001; 85: 438–443.CrossRefGoogle ScholarPubMed
43.Wren, C, Richmond, S, Donaldson, L. Presentation of congenital heart disease in infancy: Implications for routine examination. Arch Dis Child Fetal Neonatal Ed. 1999; 80: F49–F53.CrossRefGoogle ScholarPubMed
Supplementary material: PDF

Griebsch supplementary

Griebsch supplementary

Download Griebsch supplementary(PDF)
PDF 35 KB