Adenoid cystic carcinoma (ACC) is one of the most common malignant salivary gland tumors, and is the major type of malignant tumor in the minor salivary glands. Among salivary gland tumors, the most frequent site of ACC is the palate, followed by the tongue, buccal mucosa, lip, and floor of the mouth. Among the malignant tumors originating from the minor salivary glands of the tongue, more than 85% involve the base of the tongue. Further, ACC arising from the anterior third of the tongue, particularly from an anterior lingual salivary gland, is extremely rare. Herein, we report a case of ACC originating from the anterior lingual salivary gland. A 72-year-old man presented with an elastic, immobile mass accompanied by pain on the left side of the tongue. Following incisional biopsy, the mass was diagnosed as ACC. Partial glossectomy was performed with sufficient safety margin. The anterior third of the tongue was resected along with the sublingual salivary gland, tissues of the oral floor and the genioglossus muscle. Histological examination revealed typical ACC with the predominant cribriform pattern. Although no tumor cells were detected in the margins, some tumor cells showed perineural invasion. At the 20-month follow-up, no evidence of local recurrence, distant metastasis, or dysfunction was observed. However, ACC has a relentless clinical course and frequently a fatal outcome, owing to its invasive local growth and a high incidence of distant metastasis. Therefore, rigorous long-term follow-up is essential.
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