A woman in her 30s with a history of bruxism and long-term use of a bite guard presented with pain centering on left submandibular area for 2 years. She was initially prescribed a muscle relaxant without notable improvement. Physical examination revealed a mild firmness in the left submandibular area. The mucosa of the oral cavity and floor of mouth was normal. Cranial nerve function, including tongue mobility and sensation, was also normal. Contrast-enhanced computed tomography (CT) demonstrated a 1.7 × 1.5 × 1.4-cm mass, adjacent to the left submandibular gland. Scattered, nonenlarged lymph nodes were observed (Figure , A). An ultrasonography-guided fine needle aspiration biopsy was nondiagnostic owing to an inability to obtain sufficient tissue. The patient underwent surgical exploration with left submandibular gland excision and resection of the left submandibular mass (Figure , B). Histopathologic findings showed spindle cell neoplasm with morphologic features and no malignant neoplasm identified (Figure , C). Results from immunohistochemical analysis were positive for S100 (Figure , D) and negative for smooth muscle actin, CD34, and pankeratin.