title: Children with suspected craniosynostosis: a cost-effectiveness analysis of diagnostic strategies.
abstract: OBJECTIVE
  Our purpose was to evaluate the clinical and economic impact of three evaluation strategies in children at different risks of craniosynostosis.
  
  
  METHODS
  A decision-analytic and cost-effectiveness model was constructed to compare three evaluation in strategies in children with suspected synostosis: no imaging, radiography (if abnormal, followed by three-dimensional CT [3D CT]), and 3D CT. Three risk groups were analyzed on the basis of the prevalence (pretest probability) of disease: low (completly healthy children; prevalence, 34/100,000), intermediate (healthy children with head deformity; prevalence, 1/115), and high risk (children with syndromic craniofacial disorders [i.e., Crouzon's syndrome or Apert's syndrome]; prevalence, 9-10/10). Test performance (sensitivity and specificity) of the evaluation strategies was obtained from the literature. Costs (not charge) estimates were obtained from the hospital cost-accounting database and from the Medicaid fee schedule.
  
  
  RESULTS
  In the low-risk group, the radiographic and 3D CT strategies resulted in a cost per quality-adjusted life year (QALY) gained of more than $560,000. In the intermediate-risk group, the radiographic strategy resulted in a cost per QALY gained of $54,600. Three-dimensional CT was more effective than the two other strategies but at a higher cost-hence, with a cost per QALY gained of $374,200. In the high-risk group, 3D CT was the most effective strategy with a cost per QALY gained of $33,800. Less experienced radiologists and poor-quality studies increased the evaluation cost per QALY gained for all of the risk groups because of decreased effectiveness.
  
  
  CONCLUSIONS
  Radiologic screening of completely healthy children (low risk) for synostosis is not warranted because of the high cost per QALY gained of the radiographic and 3D CT strategies. In healthy children with head deformity (intermediate risk), the radiographic strategy had a reasonable cost per QALY gained. Three-dimensional CT was more effective but had a high cost per QALY gained. In children with syndromic craniofacial disorders (high risk), 3D CT was the most effective strategy and had a reasonable cost per QALY gained. Selection of children with suspected craniosynostosis based on their risk group and use of the most appropriate evaluation strategy could maximize clinical and economic outcomes for these patients.
authors.affiliation: Department of Radiology, Health Outcomes, Policy and Economics (HOPE) Center, Brain Institute, Miami Children's Hospital, 3100 S.W. 62 Ave., Miami, FL 33155, USA.
authors.forename: L Santiago
authors.initials: LS
authors.lastname: Medina
authors.forename: Randy R
authors.initials: RR
authors.lastname: Richardson
authors.forename: Kerry
authors.initials: K
authors.lastname: Crone
mesh_terms: D002648:Child; D003362:Cost-Benefit Analysis / Q000191:economics; D003398:Craniosynostoses / Q000175:diagnosis* / Q000191:economics* / Q000209:etiology; D003661:Decision Support Techniques; D006801:Humans; D021621:Imaging, Three-Dimensional / Q000191:economics*; D017063:Outcome Assessment, Health Care; D019057:Quality-Adjusted Life Years; D011859:Radiography / Q000191:economics*; D012307:Risk Factors; D012680:Sensitivity and Specificity; D014057:Tomography, X-Ray Computed / Q000191:economics*