Title: Extraglandular Submandibular Adenoid Cystic Carcinoma Involving the Lingual Nerve: A Case Report
Authors: A. Bouhlala, Y. Chabraoui, M. Tayane, S. Aourarh, T.O. El Assead, I. Benchafai,
Volume: 10
Issue: 6
Pages: 52-54
Publication Date: 2026/06/28
Abstract:
Malignant tumors of the submandibular salivary gland are uncommon and often difficult to differentiate clinically from benign lesions. Adenoid cystic carcinoma (ACC) is characterized by slow growth and a strong tendency for perineural invasion. We report a rare case of extraglandular ACC arising from accessory salivary glands in the submandibular region, independent of the submandibular gland. A 38-year-old woman presented with a painful left laterocervical mass associated with hypoesthesia in the distribution of the lingual nerve. Magnetic resonance imaging revealed an irregularly contoured mass in the left submandibular region. Surgical exploration identified a polylobulated tumor encasing the lingual nerve, necessitating tumor excision with nerve sacrifice and submandibulectomy. Histopathological examination confirmed adenoid cystic carcinoma with perineural and vascular invasion, while the submandibular gland was free of tumor. The patient received adjuvant radiotherapy. This case highlights an unusual extraglandular origin of ACC and emphasizes the importance of multidisciplinary management.