Immunohistochemical and ultrastructural findings in a rare case of papillary cystadenocarcinoma arising from the left sublingual gland of a 55-year-old Japanese man are reported. Histologically, the tumor tissue was found to be composed of various-sized cystic cavities in which papillary epithelial projections with thin fibrovascular cores were observed. The papillary projections consisted of a single layer to several layers of high columnar epithelial cells. Invasion to the surrounding fibrous tissue and into the lymphatics was observed, thus suggesting an aggressive potential in the present case. The possibility of the involvement of myoepithelial cells could be excluded based on the immunohistochemical and ultrastructural findings. The immunohistochemical and ultrastructural findings also suggested that this type of salivary gland tumor, at least the present case, may arise from striated or excretory ducts. There was positive immunostaining for tumor markers CA19-9 and CA125. However, the biological role of these carbohydrate antigens in salivary gland tumors is unclear at present. Further investigations are, therefore, called for to solve this issue.