A Photo Score For Aesthetic Outcome In Sagittal Synostosis: An ERN CRANIO Collaboration
1Dutch Craniofacial Center, Erasmus MC-Sophia Children's Hospital, University Medical Centre Rotterdam, Department of Plastic and Reconstructive and Hand surgery, Rotterdam, the Netherlands.
The Journal of Craniofacial Surgery
|September 13, 2023
Summary
A new photo score for sagittal synostosis was developed to assess craniofacial anomalies. While individual feature agreement was fair-moderate, the overall phenotype score showed substantial agreement among experts.
Area of Science:
- Craniofacial Surgery
- Pediatric Plastic Surgery
- Medical Imaging Analysis
Background:
- European Reference Network (ERN) CRANIO aims to standardize care for rare craniofacial anomalies.
- Objective data collection is crucial for multicenter comparison of treatment outcomes in conditions like craniosynostosis.
- A reliable, suture-specific photo score is needed to objectively assess phenotypical severity and treatment outcomes.
Purpose of the Study:
- To develop a reproducible and reliable suture-specific photo score for sagittal synostosis.
- To enable cross-center comparison of phenotypical severity and aesthetic outcomes.
- To evaluate the interrater reliability of the developed photo score.
Main Methods:
- Retrospective study of nonsyndromic sagittal synostosis patients (<19 years) from 6 ERN CRANIO centers.
- Inclusion of preoperative and postoperative photo sets (bird's eye, lateral, anterior-posterior views).
- Development and consensus on a photo score including frontal bossing, skull elongation, biparietal narrowness, etc., with scoring from normal to severe. Interrater agreement assessed using Fleiss' Kappa and ICC.
Main Results:
- 36 patients with scaphocephaly were scored by 20 raters.
- Interrater agreement for individual items ranged from fair (Kappa 0.38 for biparietal narrowness) to moderate (Kappa 0.56 for frontal bossing).
- Substantial agreement (ICC 0.69) was achieved for the sum score of individual photo score items.
Conclusions:
- The developed photo score is the first large-scale, multicenter tool to assess phenotypical characteristics of sagittal synostosis.
- While individual phenotypical characteristics showed suboptimal agreement, the summed score demonstrated substantial agreement, indicating consensus on overall severity.
- This tool supports standardized, cross-center evaluation of sagittal synostosis severity and treatment outcomes.
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