Radiographic extent of maxillofacial Gorham's disease and its impact on recurrence: A systematic review

Objective This systematic review assessed the radiographic extent of maxillofacial Gorham's disease and its impact on recurrence. Study Design PubMed and Cochrane databases were searched. The key words were (Gorham Disease OR Vanishing Bone Disease OR Gorham Stout Syndrome) AND (Jaw OR Maxilla OR Mandible OR Maxillofacial). Results Forty-one cases from 39 articles were included. Based on radiographic extent, the lesions were classified as large or small. Most cases occurred in the mandible (24), followed by a combination of maxilla and/or mandible and other bones (12), both mandible and maxilla (3), and maxilla only (2). Only mandibular cases were analyzed for radiographic extent. Of the 24 mandibular lesions, 16 were large and 8 were small. Nine of the large lesions occurred in the young age group with 2 recurrences, followed by 5 in the middle-aged group with 1 recurrence. Small lesions were nearly equally distributed over the age groups, with 2 recurrences in the middle-aged group and 1 recurrence in the adult age group. Age, sex, and lesion size had no significant effect on recurrence. Conclusions Radiographic extent of the lesion does not impact recurrence, suggesting a possible existence of aggressive and nonaggressive variants of maxillofacial Gorham's disease. This systematic review assessed the radiographic extent of maxillofacial Gorham's disease and its impact on recurrence. PubMed and Cochrane databases were searched. The key words were (Gorham Disease OR Vanishing Bone Disease OR Gorham Stout Syndrome) AND (Jaw OR Maxilla OR Mandible OR Maxillofacial). Forty-one cases from 39 articles were included. Based on radiographic extent, the lesions were classified as large or small. Most cases occurred in the mandible (24), followed by a combination of maxilla and/or mandible and other bones (12), both mandible and maxilla (3), and maxilla only (2). Only mandibular cases were analyzed for radiographic extent. Of the 24 mandibular lesions, 16 were large and 8 were small. Nine of the large lesions occurred in the young age group with 2 recurrences, followed by 5 in the middle-aged group with 1 recurrence. Small lesions were nearly equally distributed over the age groups, with 2 recurrences in the middle-aged group and 1 recurrence in the adult age group. Age, sex, and lesion size had no significant effect on recurrence. Radiographic extent of the lesion does not impact recurrence, suggesting a possible existence of aggressive and nonaggressive variants of maxillofacial Gorham's disease.