Summary

Papillary cystadenoma is an unusual benign cystic neoplasm with cystic spaces of diverse sizes and intraluminal papillary projections histologically, which is commonly seen in the lips, cheek and palate. We report a case of papillary cystadenoma in a 40-year-old man on the anterior one-third of the tongue with classical histological features. The paper highlights the rarity of the site of occurrence with emphasis on differential diagnosis and the need for considering papillary cystadenoma when benign cystic lesions are encountered in the anterior one-third of the tongue.