Primary intestinal-type adenocarcinoma of the oral tongue: Case report and review of histologic origin and oncologic management.

Abstract

Primary intestinal-type adenocarcinoma (ITAC) of the oral tongue is an extremely uncommon malignancy with only 3 cases reported in the literature. This high-grade malignancy originates from metaplasia of minor salivary glands. A 40-year-old man presented with a gradually enlarging midline oral tongue mass, odynophagia, and dysphagia. Management included a median lingual glossectomy, bilateral neck dissections, and adjuvant chemoradiation with 5-fluorouracil (5-FU). Additional chemotherapy with folinic acid, fluorouracil, and oxaliplatin (FOLFOX) was given to mimic therapy in colonic adenocarcinomas. Thirteen months after surgery and adjuvant chemoradiotherapy, there is no evidence of locoregional or distant disease. His diet and speech have normalized after reconstruction without free tissue transfer. We report the fourth case of oral tongue ITAC, and present the first histologic evidence of metaplasia of oral cavity salivary epithelium. We also discuss adjuvant therapy recommendations given the lack of clarity for treatment of this rare disease.