A case of adenoid cystic carcinoma originating in the anterior lingual salivary gland with contralateral cervical lymph node metastasis eight years after primary tumor resection

Abstract

Adenoid cystic carcinoma （AdCC） is a rare salivary gland tumor that occurs in various head and neck sites, often diagnosed at an advanced stage. It grows slowly but infiltrates surrounding nerves, leading to frequent local recurrence and distant metastasis, resulting in a poor prognosis. AdCC comprises approximately 10％ of salivary gland tumors and predominantly occurs in minor salivary glands, most commonly in the palate, whereas its occurrence in the anterior lingual salivary gland is extremely rare. Here, we present a case of AdCC thought to originate from the anterior lingual gland, which developed contralateral cervical lymph node metastasis approximately 8 years after surgery. A 79-year-old male presented with an elastic, hard, ulcerative mass extending from the right tongue tip to the ventral surface of the tongue. Incisional biopsy confirmed AdCC. A wide safety margin was set, and the sublingual gland, oral floor tissues, and part of the mylohyoid muscle were resected, followed by a skin graft from the abdomen. The surgical margins were negative. During follow-up, 7 years and 10 months after surgery, contralateral submandibular lymph node metastasis was observed. No primary site recurrence occurred. A left supra-omohyoid neck dissection was performed for local control. At 1 year and 7 months post-surgery, no complications or recurrence had occurred. Although no postoperative complications or local recurrence have been detected, continued careful long-term follow-up is required.