Temporomandibular joint anomalies in pediatric craniofacial Gorham-Stout disease

Purpose: Gorham-Stout disease (GS) is a rare and little understood bone disease that leads to the progressive replacement of the affected bone by fibrous tissues. The mandible is the most commonly affected craniofacial bone, but there is no report to date of temporomandibular joint (TMJ) lesions in this condition. We aimed to characterize the involvement of the TMJ in this uncommon bone disorder. Material and methods: We retrospectively included all patients managed for craniofacial forms of GS in our pediatric institution over a period of 12 years. We collected clinical data on TMJ function and radiological data from computed tomography and magnetic resonance imaging. Results: Four pediatric patients were managed for craniofacial forms of GS between 2005 and 2017. All patients presented with various radiological lesions of the TMJs, including osteolytic lacunae of the condylar head, condylar head flattening and thinning of the glenoid cavity. Only one patient presented with functional TMJ impairment. Conclusion: The TMJ appears to be radiology affected in pediatric craniofacial forms of GS. Nevertheless, TMJ lesions, even when radiologically severe, rarely impair TMJ function. TMJ structure and function should be systematically assessed in craniofacial forms of GS, and, in the case of joint lesions, a regular follow-up of TMJ function should be considered. (c) 2018 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.