Unusual presentation of Sjögren syndrome.

This case report describes a patient with Sjögren syndrome (SS) whose sole presenting feature was bilateral and painful submandibular gland enlargement. Extensive workup for alternate etiologies was negative and while serologies specific for SS were unremarkable, the diagnosis was eventually suggested by excisional biopsy. This case highlights the difficulty in making an early diagnosis of SS and demonstrates the important role of excisional biopsy in that diagnosis.