Related Experiment Video
Updated: Sep 30, 2026
Midface Hypoplasia and Cranial Base Morphology in Syndromic Craniosynostosis: A Comparative Analysis Study Using a Predictive Regression Model
Published on: November 4, 2025
Children with suspected craniosynostosis: a cost-effectiveness analysis of diagnostic strategies
L Santiago Medina1, Randy R Richardson, Kerry Crone
1Department of Radiology, Health Outcomes, Policy and Economics (HOPE) Center, Brain Institute, Miami Children's Hospital, 3100 S.W. 62 Ave., Miami, FL 33155, USA.
Insights
For children with suspected craniosynostosis, diagnostic strategy depends on risk. Radiography is cost-effective for intermediate-risk cases, while 3D CT is best for high-risk syndromic disorders.
Area of Science:
- Pediatric Radiology
- Health Economics
- Medical Decision Making
Background:
- Craniosynostosis evaluation involves complex diagnostic decisions.
- Risk stratification is crucial for appropriate imaging strategies.
- Clinical and economic outcomes vary significantly with evaluation methods.
Purpose of the Study:
- To compare the clinical and economic impact of three craniosynostosis evaluation strategies.
- To analyze these strategies across different pediatric risk groups.
- To determine cost-effectiveness for optimizing patient care.
Main Methods:
- A decision-analytic and cost-effectiveness model was developed.
- Three strategies were compared: no imaging, radiography with potential 3D CT, and direct 3D CT.
- Analysis included low (healthy), intermediate (head deformity), and high (syndromic) risk groups.
Main Results:
- Radiographic and 3D CT strategies were not cost-effective for low-risk children (>$560,000/QALY).
- For intermediate-risk children, radiography cost $54,600/QALY, while 3D CT cost $374,200/QALY.
- In high-risk children, 3D CT was most effective at $33,800/QALY.
Conclusions:
- Radiologic screening is not recommended for low-risk children due to high costs.
- Radiography offers reasonable cost-effectiveness for intermediate-risk children.
- 3D CT is the most effective and cost-effective strategy for high-risk children with syndromic craniofacial disorders.
Objective:
Our purpose was to evaluate the clinical and economic impact of three evaluation strategies in children at different risks of craniosynostosis.
Materials And 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.
Conclusion:
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.
