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Interrater Reliability for Classifying Craniofacial Microsomia Severity: A Call for Objective Evaluation
Elsa M Ronde1,2,3,4, Jitske W Nolte2,4, Alfred G Becking2,3,4
1Department of Plastic, Reconstructive and Hand Surgery, Amsterdam UMC, University of Amsterdam, Amsterdam, the Netherlands.
Abstract:
ObjectiveThe severity of craniofacial microsomia (CFM) is generally classified using the Orbit, Mandible, Ear, Soft tissue, Nerve (OMENS) classification score. The global assessment of the Phenotypic Assessment Tool for Craniofacial Microsomia (PAT-CFM), is a pictorial modification of the OMENS classification. The aim of this study was to assess the interrater reliability of the PAT-CFM global assessment score.DesignIn this prospective cohort study, three clinicians completed the global assessment form of the PAT-CFM. The mandible was classified based on orthopantomogram- and/or computed tomography images.ParticipantsConsecutive patients with CFM or microtia.Main Outcome MeasureInterrater agreement was calculated using the weighted Krippendorff alpha (α), with 95% confidence intervals (CI).ResultsIn total, 53 patients were included (106 hemifaces). The reliabilities of the main classification components ranged from high for the mandible (α = 0.904 [95% CI 0.860-0.948]) and ear (α = 0.958 [95% CI 0.934-0.983]) subscales, to tentative for the orbital summary score (α = 0.682 [0.542-0.821]), and nerve summary score (α = 0.782 [0.666-0.900]) subscales.ConclusionsThe reliability of the ear and radiographic mandible scales of the PAT-CFM global classification were high, while the orbit, facial nerve and soft tissue subscales may have limited reliability. Research focusing on radiographic severity scores for hypoplasia of the orbits and soft tissues, as well as objective measures for overall facial hypoplasia using non-ionizing forms of imaging for early classification, are warranted.
Insights
The Phenotypic Assessment Tool for Craniofacial Microsomia (PAT-CFM) shows high reliability for classifying ear and mandible severity in craniofacial microsomia (CFM). However, orbit, nerve, and soft tissue assessments may be less reliable.
Area of Science:
- Medical Imaging
- Plastic Surgery
- Genetics
Background:
- Craniofacial microsomia (CFM) severity is typically assessed using the Orbit, Mandible, Ear, Soft tissue, Nerve (OMENS) score.
- The Phenotypic Assessment Tool for Craniofacial Microsomia (PAT-CFM) offers a pictorial modification of the OMENS classification for global assessment.
Purpose of the Study:
- To evaluate the interrater reliability of the PAT-CFM global assessment score for craniofacial microsomia.
Main Methods:
- A prospective cohort study involving three clinicians assessing PAT-CFM global forms.
- Mandible classification utilized orthopantomogram and/or computed tomography imaging.
- Interrater agreement was quantified using weighted Krippendorff alpha (α) with 95% confidence intervals (CI).
Main Results:
- The study included 53 patients (106 hemifaces).
- High reliability was observed for mandible (α = 0.904) and ear (α = 0.958) subscales.
- Tentative reliability was found for orbit (α = 0.682) and nerve (α = 0.782) subscales.
Conclusions:
- The ear and radiographic mandible scales of the PAT-CFM demonstrate high interrater reliability.
- The orbit, facial nerve, and soft tissue subscales may have limited reliability.
- Further research is needed for objective, non-ionizing imaging methods for early classification of orbital and soft tissue hypoplasia.

