Analysis of skeletal mandibular abnormalities associated with cervicofacial lymphatic malformations

Abstract

Objectives/hypothesis: Many children with cervicofacial lymphatic malformations have facial skeletal abnormalities. This study qualitatively and quantitatively evaluated these bony mandibular abnormalities.

Study design: Retrospective chart review.

Methods: Patients with craniofacial lymphatic malformations presenting to a vascular anomalies center during a 2-year period were included. An age-matched control population was found on the Picture Archiving and Communication System database. Three-dimensional reconstructions were created from computed tomography and magnetic resonance imaging. Qualitative observations and quantitative measurements were taken of the gonial angle, mandibular anterior dentoalveolar height, and anterior condylar displacement.

Results: There were 23 controls. A total of 21 patients with "beard" distribution malformations were studied; 10 had unilateral and 11 had bilateral disease.Qualitatively, a few patterns emerged: outward ramal flaring, anterior displacement of the mandible, relative ipsilateral facial "hypertrophy," and anterior positioning of the maxilla and orbit. The open-bite deformity was a common finding leading to malocclusion and oral incompetence. Quantitatively, in 67 nondiseased sides, the average gonial angle was 131 degrees (standard deviation [SD] = 6.8), whereas in 32 diseased sides, the average angle was 152 degrees (SD = 14.0, P < 3.8E-09). With half-beard malformations, the diseased side averaged 153.9 and the nondiseased side averaged 140.8 (P = .008). The average condylar displacement was 8.5 mm versus 5.9 mm (diseased vs. nondiseased), and the average mandibular dentoalveolar height to face ratio was 0.37 (control = 0.34).

Conclusions: Cervicofacial lymphatic malformations in the "beard" distribution are associated with significant bony abnormalities leading to both functional and aesthetic sequelae. Our study is the first quantitative analysis of these changes. This is the first step in planning for dentofacial orthopedics, orthodontics, and orthognathic surgery.