Cheilitis glandularis (CG) is an uncommon disease that usually affects the lower lip of adults. It is characterized by enlargement and eversion of the lip in association with excretory duct dilatation. The presence of minor salivary gland hyperplasia is controversial. Three types of CG have been described in the literature; the classification is based on the common clinical and histopathologic findings (ie, simple, superficial, and deep). This report is of an unusual case of CG simplex for which the initial histopathologic diagnosis was papillary cystadenoma, a neoplastic process. The lesion was completely excised, and final microscopic review of a larger specimen revealed chronic sclerosing sialadenitis, dilated salivary secretory ducts with oncocytic change and periductal inflammation, and foci of adenomatous hyperplasia consisting of enlarged ducts exhibiting squamous epithelial metaplasia and hyperplasia with papillary architecture. The latter proliferative pattern is an unusual finding in what is otherwise clinically diagnosed as CG.