Sarcoidosis affecting the maxilla: report of 2 cases

Abstract

Sarcoidosis is a granulomatous disease that is likely a hypersensitivity reaction to an environmental agent, characterized by the presence of noncaseating granulomas typically affecting the hilar lymph nodes and the lungs. The disease affects adults in their third to fifth decades and extramediastinal involvement affects mainly the lymph nodes, liver, skin, and eyes. Within the oral cavity, sarcoidosis affects the minor salivary glands in 25% to 50% of patients. Osseous involvement has been documented in 3% to 13% of patients but only 26 cases have been reported in the jawbones.

Here we present 2 new cases of sarcoidosis affecting the jaw bones. The first case was that of a 65-year-old female patient with a known history of sarcoidosis with respiratory symptoms managed with budesonide inhaler who presented with an expansile radiolucent lesion of the anterior maxilla in the area of the right maxillary canine and incisors. The second case is of a 55-year-old female patient with stable pulmonary sarcoidosis not currently undergoing therapy who presented with a radiolucency of the right maxilla with the right maxillary canine “floating in space.” The differential diagnoses included odontogenic cysts and tumors, Langerhans cell histiocytosis, and central giant cell granuloma.

Biopsies from both patients revealed non-necrotizing granulomatous inflammation consistent with the patients’ known history of sarcoidosis. Histochemical studies to rule out an infectious cause for the granulomas were negative. Of the 26 previously reported cases of sarcoidosis involving the jaw bones, the mean age at diagnosis was 37.4 years (range 16-59 years) and there was a female predilection (1.9:1) Involvement of jaw bones was observed primarily in tooth-bearing areas with two cases affecting the mandibular condyles. The mandible and maxilla were equally affected in 38.5% of cases and all presented as poorly defined radiolucencies. In four cases, there was generalized involvement of both the mandible and the maxilla. In 11 cases (42.3%), oral involvement preceded identification of systemic disease. Management of patients ranged from surgical to nonsurgical intervention including the use of steroid treatment. Patients who received surgical treatment exhibited improvement of the jaw lesions, often exhibiting osseous regeneration on follow-up visits.