title: 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.
abstract: OBJECTIVE
  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 pounds and 4,496,666 pounds for pulse oximetry and for screening echocardiography. The equivalent costs for clinically significant CHD are 1,489 pounds and 36,013 pounds, 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.
authors.affiliation: Department of Social Medicine, University of Bristol, UK. Ingolf.Griebsch@merck.de
authors.forename: Ingolf
authors.initials: I
authors.lastname: Griebsch
authors.forename: Rachel L
authors.initials: RL
authors.lastname: Knowles
authors.forename: Jacqueline
authors.initials: J
authors.lastname: Brown
authors.forename: Catherine
authors.initials: C
authors.lastname: Bull
authors.forename: Christopher
authors.initials: C
authors.lastname: Wren
authors.forename: Carol A
authors.initials: CA
authors.lastname: Dezateux
mesh_terms: D003362:Cost-Benefit Analysis; D004452:Echocardiography; D006330:Heart Defects, Congenital / Q000175:diagnosis*; D006801:Humans; D007231:Infant, Newborn; D015997:Neonatal Screening / Q000191:economics* / Q000379:methods; D010092:Oximetry / Q000379:methods*; D013222:State Medicine; D006113:United Kingdom