Mucoepidermoid carcinoma arising in a background of sialadenoma papilliferum: a case report

Abstract

Sialadenoma papilliferum is a rare tumor, primarily of minor salivary gland origin, first described by Abrams and Finck (Cancer 24:1057-63, 1969). It is both an exophytic and endophytic papillary lesion histologically resembling syringocystadenoma papilliferum of sweat gland. The tumor is considered benign although rare recurrent cases have been reported. Three cases of malignant transformation of sialadenoma papilliferum have been described in the literature. We report a high grade mucoepidermoid carcinoma arising in a background of sialadenoma papilliferum, at the base of the tongue, an unusual location for minor salivary gland neoplasms. Eleven months after excision and nodal dissection, there is no evidence of recurrence or metastasis.

Keywords: Mucoepidermoid carcinoma; Salivary gland; Sialadenoma papilliferum.

Fig. 1

a Sialadenoma papilliferum. Exophytic portion. 20×; hemotoxylin-eosin stain. Boxed area is shown in higher magnification in illustration b. b Excretory salivary duct in direct connection with squamous epithelium. Arrow indicates the connection between squamous surface epithelium and salivary excretory duct columnar epithelium. 200×; hemotoxylin-eosin stain. c Mucoepidermoid carcinoma with intermixed components of squamoid, mucous and intermediate cells. 200×; hemotoxylin-eosin stain

Fig. 2

a Mucoepidermoid carcinoma arising in a background of sialadenoma papilliferum. Arrow, superficial component of sialadenoma papilliferum. Arrowhead, deep portion of the lesion with mucoepidermoid carcinoma intermixed with sialadenoma papilliferum. 10×, hemotoxylin-eosin stain. b Mucoepidermoid carcinoma. Predominantly squamous component with high grade nuclear features. 400×; hemotoxylin-eosin stain. c Mucicarmine stain for mucoepidermoid carcinoma, demonstrating both intraglandular and intracellular mucin. 400×; Mucicarmine stain (Sigma-Aldrich, St. Louis, MO)