Intra-oral Sarcoidosis Preceding Lofgren’s Syndrome

Objective: A case of Lofgren’s syndrome with a primary lesion of intraoral sarcoidosis is described.

Methods: An incisional biopsy of a “fibroma” in the right posterior mucobuccal fold of the lower jaw of a 39-year old male who presented to the dentist for examination of this lesion, was carried out and subjected to histological examination. Macroscopically, the specimen was a 0.6 gm tissue with dimensions 1.7 x 1.2 x 0.7 cm3. It had a partial nodular center and appeared transparent on the incision surface. Two weeks later, the patient suffered from ankle pain, malaise, and painful erythematous nodes at the lower legs. Moreover, the patient complained of shortness of breath. Previous medical history was non-contributory. Chest radiographs, CBC, dermatological, and ophthalmological examinations, sonograms of peripheral lymph nodes, liver, spleen, pancreas, and kidneys, as well as pulmonary function tests were carried out.

Results: Chest radiography revealed hilar lymphadenopathy. This condition was considered to be consistent with sarcoidosis, lymphogranulomatosis, or carcinoma. The patient’s full blood count was within normal range. The VDRL test for syphilis was non-reactive. Pulmonary function tests revealed a mild restrictive component. Histologically, there were numerous small granulomas composed mainly of epithelioid cells with ample cytoplasm and scattered by Langhan’s giant cells and lymphocytes. Necrosis was either absent or limited to a small central fibrinoid focus. There was no microscopic evidence of acid-fast bacilli in the Ziehl-Neelsen staining or fungi in the PAS staining.

Conclusion: A distinctive feature in this case of Lofgren’s syndrome is the initial histologic evidence of intraoral sarcoidosis before the presence of further clinical manifestations.