A 55-year-old woman sought out the stomatology clinic, complaining of a nodule under the tongue with three years of evolution. Clinical examination revealed a mobile nodule of firm consistency, covered by intact mucosa of normal color, measuring about 2 cm, located on the left mouth floor. In view of the hypotheses of benign or glandular mesenchymal neoplasia, an excisional biopsy was performed. During surgery, the posterior part of the lesion was attached to the musculature. Histopathological examination showed malignant glandular neoplasia, characterized by islands of basaloid cells containing multiple pseudocystic cylindrical spaces filled with basophilic material. The surgical margins were compromised of the neoplasm. The diagnosis was adenoid cystic carcinoma. The patient underwent surgery with a safety margin and radiotherapy. After treatment of the mucositis caused by radiotherapy, the patient was rehabilitated with removable dental prostheses and is undergoing clinical follow-up with no signs of recurrence after 18 months. A 55-year-old woman sought out the stomatology clinic, complaining of a nodule under the tongue with three years of evolution. Clinical examination revealed a mobile nodule of firm consistency, covered by intact mucosa of normal color, measuring about 2 cm, located on the left mouth floor. In view of the hypotheses of benign or glandular mesenchymal neoplasia, an excisional biopsy was performed. During surgery, the posterior part of the lesion was attached to the musculature. Histopathological examination showed malignant glandular neoplasia, characterized by islands of basaloid cells containing multiple pseudocystic cylindrical spaces filled with basophilic material. The surgical margins were compromised of the neoplasm. The diagnosis was adenoid cystic carcinoma. The patient underwent surgery with a safety margin and radiotherapy. After treatment of the mucositis caused by radiotherapy, the patient was rehabilitated with removable dental prostheses and is undergoing clinical follow-up with no signs of recurrence after 18 months.