Abstract

<h3>Background</h3> A 65-year-old woman presented to the oral maxillofacial department with a nonhealing oral antral fistula. Radiographic examination revealed opacification of the left maxillary antrum and an ill-defined radiolucency in the maxilla. The patient continued to develop further osteolytic lesions in the skull and mandible, leading to anesthesia of the third division of the trigeminal nerve. <h3>Objective</h3> To discuss the clinicopathological correlation, differential diagnosis, and pathway to achieving a definitive diagnosis in this challenging case. <h3>Results</h3> Clinical symptoms included a history of fatigue, painful joints, and chest pain. Multiple biopsies taken from the osteolytic lesions of the mandible, maxilla, and a supraclavicular lymph node revealed naked noncaseating granulomas. Computed tomography (CT) of the head and neck revealed an irregular, diffuse, mottled appearance of the bone in the left maxillary alveolus, right mandible, and left temporal bone. Angiotensin-converting enzyme and serum calcium levels were normal. CT scans showed hilar lymphadenopathy, which was not visible on plain film radiography. Exclusion of other causes of granulomatous inflammation eventually led to a diagnosis of sarcoidosis. The patient has been discussed at a multiple disciplinary team meeting and referred to a specialist team. <h3>Conclusions</h3> Sarcoidosis is a chronic multisystem granulomatous disease characterized histologically by naked noncaseating granulomas. It has an unknown etiology and no pathognomonic diagnostic test, making definitive diagnosis difficult. This patient presented with rare and aggressive features appearing initially to be from the maxillary antrum, eventually affecting the jaws and skull with only subtle symptoms of the more cardinal features of the disease.

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