Papillary cystadenocarcinoma (CAC) a rare malignant salivary gland tumor which commonly arises in the major glands, mainly the parotid, and less frequently in the minor salivary glands. Localization in the sublingual salivary gland is very rare. The authors present a case of papillary CAC originating from the right sublingual gland in a 78-year-old female. A review of the literature revealed only 3 previously published detailed cases (in addition to 2 cases as part of larger series) of papillary CAC of the sublingual gland. The salient demographic, clinical, and histopathologic findings of papillary CAC, as well as its surgical treatment, are discussed. Key words:Papillary cystadenocarcinoma, sublingual gland, cystic structures ABSTRACT Article history: