ArticleThe Journal of craniofacial surgery2026
A 2D Photo Score for Phenotypic Severity in Unilateral Coronal Synostosis Patients.
Article in The Journal of craniofacial surgery, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
backgroundUnilateral coronal synostosis (UCS) presents with a wide range of phenotypic severity, yet commonly used outcome measures are heterogeneous and often subjective, resource-intensive, or non-suture-specific. Reliable grading of phenotypic severity is crucial for evaluating aesthetic outcomes and for benchmarking treatment results across centers. The aim of this study was to develop a simple, suture-specific, 2D photo-based scoring system for UCS and to assess its reliability across multiple expert centers.
methodsA retrospective multicenter study was conducted using standardized photos of patients with nonsyndromic UCS from 6 European expert centers. A UCS-specific photo score was developed through expert consensus and included 5 items: forehead shape, temporal hollowing, orbital dystopia, and nasal deviation, together with an overall phenotype item. All items were scored on a 4-point ordinal scale. A cumulative severity score was calculated from the 4 feature-specific items, excluding the overall phenotype. Twenty craniofacial plastic surgeons and pediatric neurosurgeons independently scored 60 photo sets.
resultsAgreement for individual items was moderate (κ=0.48-0.56), with lower reliability for the overall phenotype item (κ=0.41). The agreement was highest for the cumulative score of feature-specific items (intraclass correlation coefficient: 0.64). Reliability was consistent between 2D photos and 3D-renderings.
conclusionThis study introduces the first multicenter, suture-specific photo score for UCS. Combining multiple UCS-specific features into a cumulative score substantially improves reliability, supporting its use as a practical tool for standardized phenotypic severity assessment and multicenter benchmarking.
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