Sialadenoma Papilliferum: Analysis of Seven New Cases and Review of the Literature

Abstract

Sialadenoma papilliferum (SP) is a rare benign salivary gland neoplasm that comprises from 0.4 to 1.2% of all salivary gland tumors. The tumor is so named because of its microscopic resemblance to the syringocystadenoma papilliferum, an uncommon benign tumor of sweat gland origin. The purpose of this paper is to report the clinical and microscopic features of seven new cases of SP and combine them with cases previously reported in the English language literature to further define this unusual lesion. Combining our cases with acceptable cases from the literature, the palate (especially the hard palate) was the most common site, with 80% of the cases occurring in this location. Other locations in decreasing order were buccal mucosa, upper lip, retromolar pad, and parotid gland. Age at diagnosis ranged from 18 to 87 years with a peak in the 6th decade and an average age of 56.4 years. Microscopically, the lesions demonstrated a papillary surface morphology, and the papillary projections varied from long and pointed to short and blunted. The supporting connective tissue contained a variable number of convoluted ductal structures, which often fused with the overlying surface epithelium. The ductal structures exhibited papillary infoldings and were lined by a double layer of epithelium consisting of basal cell layer and a luminal layer of cuboidal-to-columnar ductal cells. Immunohistochemical reactivity with p63 and p40 indicated that the basal cell layer was comprised predominantly of neoplastic myoepithelial cells. The luminal cells were immunoreactive with epithelial membrane antigen characteristic of ductal cell differentiation. Conservative surgical treatment was accomplished in most cases and appears to be adequate treatment as only two recurrences were documented. Several case reports of purported malignant transformation in SP have been reported in the literature, but in our opinion, there is insufficient evidence in the publications to unequivocally determine whether any of the malignancies truly originated within a pre-existing SP.

Keywords: Papilliferum; Salivary gland neoplasm; Sialadenoma papilliferum; Syringocystadenoma.

Fig. 1

Anatomic location of SP (combined UKCD and literature cases)

Fig. 2

Age Distribution of SP (combined UKCD and literature cases)

Fig. 3

Case 5. SP showing surface papillary projections overlying a proliferation of convoluted ductal structures. (H&E stain, orig mag ×40)

Fig. 4

Case 4. SP showing cleft formation (asterisk) at the point of fusion of the surface epithelium with the underlying ductal structures. (H&E stain, orig mag ×200)

Fig. 5

Case 4. SP showing ductal structures exhibiting papillary infoldings. The ductal structures are lined by a flattened basal cell layer and a cuboidal-to-columnar luminal layer. (H&E stain, orig mag ×200)

Fig. 6

Case 5. Ductal structures of SP exhibiting oncocytic metaplasia (a) and mucous goblet cells (b). (H&E stain, orig mag ×400 [a], orig mag ×200 [b])

Fig. 7

Cases 2, 5. SP demonstrating immunoreactivity to p40 in the basal cells of the ductal structures (a) and epithelial membrane antigen (EMA) in the luminal cells (b). (p40 immunostain, orig mag ×400, EMA immunostain, orig mag ×600)

Fig. 8

Low-power photomicrographs illustrating the lesions in the microscopic differential diagnosis of SP. a exophytic ductal papilloma, b intraductal papilloma, c inverted ductal papilloma, d papillary cystadenoma. (All images are H&E stained, orig mag ×40)