A case of Gorham's disease in the maxilla of a 56-year-old male patient is described. The clinical presentation, radiographic and histopathological features and treatment are presented. A discussion of the current understanding of this rare disease, based on review of the literature, is offered.

Immunohistochemical demonstration of NCAM, HCAM, and ICAM-1 is not related to perineural invasion or tumor behavior. PECAM-1 expression was related to vascular invasion and poor patient prognosis in three cases.

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