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Quantification of osseous facial dysmorphology in untreated unilateral coronal synostosis
Insights
Untreated unilateral coronal synostosis causes measurable midface differences. This study quantifies these facial morphology changes in living individuals, highlighting the need for natural history data.
Area of Science:
- Craniofacial surgery
- Pediatric plastic surgery
- Medical imaging
Background:
- Unilateral coronal synostosis (UCS) leads to midface dysmorphology, alongside cranial and orbital deformities.
- Limited data exists on the natural history of untreated UCS due to early surgical intervention in infancy.
- This study addresses the data gap by analyzing midface morphology in living individuals with untreated UCS.
Purpose of the Study:
- To quantitatively assess the natural midface morphology in individuals with untreated unilateral coronal synostosis.
- To establish a baseline for comparison with treated patients and normal skulls.
- To demonstrate the feasibility of international collaboration for studying rare craniofacial anomalies.
Main Methods:
- Utilized high-resolution computed tomography (CT) scans from 11 living individuals with untreated UCS across three international centers.
- Constructed 3D image volumes and calculated 14 pairs of distances between 11 osseous landmarks on ipsilateral and contralateral hemifaces.
- Expressed measurements as ipsilateral:contralateral ratios and compared them with data from dried skulls.
Main Results:
- Significant differences (greater than 5%) were observed in 12 of 14 measured midface distances between the ipsilateral and contralateral sides.
- Most measurements were decreased on the ipsilateral side (affected by synostosis) compared to the contralateral side.
- Established discrete, measurable differences in facial morphology between untreated UCS and normal skulls.
Conclusions:
- Untreated unilateral coronal synostosis results in distinct and quantifiable midface morphological differences.
- International collaboration is effective for gathering sufficient data on rare craniofacial anomalies.
- Quantitative natural history data is crucial for assessing the outcomes of surgical treatments for craniofacial anomalies.
Abstract:
Unilateral coronal synostosis results in dysmorphology of the midface in addition to well-characterized cranial and orbital deformities. Because most American infants with this problem have undergone cranio-orbital surgery within their first year of life for the past 25 years, a paucity of data exist regarding the natural history of untreated unilateral coronal synostosis. In an attempt to remedy this void, an international search was conducted to identify computed tomography data sets of living individuals with untreated unilateral coronal synostosis; data were obtained from two European centers and one center in the United States. Results limited to the study of the midface are presented here. Digital data from high-resolution head computed tomography scans of 11 living, white individuals with untreated unilateral coronal synostosis were obtained from three craniofacial centers (in Denmark, The Netherlands, and the United States). Image volumes were constructed from each scan using ANALYZE biomedical imaging software. Fourteen pairs of three-dimensional distances were calculated on the ipsilateral (the side of the synostosis) and the contralateral (the side opposite to the synostosis) hemifaces using 11 osseous landmarks. The resulting measurements were expressed as a ratio of the ipsilateral: contralateral sides. Descriptive statistics were derived for the untreated unilateral coronal synostosis population and compared with analogous measurements performed on dried skulls. Age at computed tomography ranged from 1.1 to 21.1 years (mean, 6.6 years; median, 4.1 years). Twelve of the 14 measured distances differed by greater than 5 percent on the ipsilateral side, and all but one of these measurements were decreased on the ipsilateral side when compared with contralateral values. The results of this study support the following conclusions: (1) There are discrete and measurable differences in the facial morphology between patients with untreated unilateral coronal synostosis and normal skulls. (2) Intercenter and international collaboration can provide a sufficient number of individuals with rare craniofacial anomalies to quantitatively determine group characteristics. (3) Quantitative documentation of rare anomaly natural history is necessary for quantitative outcome assessment of treated patients.
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