Intraductal Papilloma: Atypical Presentation

Abstract

Ductal papillomas have unique papillary features arising from the salivary gland duct system. They comprise three rare benign adenomas, namely, inverted ductal papilloma, sialadenoma papilliferum, and intraductal papilloma. Intraductal papilloma is an extremely rare benign salivary gland tumor that occurs most commonly in the minor salivary glands. Here, we are presenting a case report of intraductal papilloma in an 18-year-old patient.

A Rare Case of Intraductal Papilloma Arising from Minor Salivary Gland in the Floor of the Mouth

“…Therefore, the present case is the third intraductal papilloma report that developed in the sublingual region, involving a minor salivary gland. Although with good prognosis and low recurrence rates, it is important to distinguish intraductal papillomas of other ductal neoplasms of salivary ductal origin, as papillary cystadenoma, cystadenocarcinoma, sialadenoma papilliferum, and inverted ductal papilloma [ 1 , 2 , 6 ].…”

“…In contrast with intraductal papilloma, cystadenoma and cystadenocarcinoma show a multicystic well-circumscribed lesion lined by papillary epithelium with variable size [ 2 , 6 ]. However, the cystadenocarcinoma is covered by a papillary epithelium with remarkable nuclear and structural atypia, some mitosis, and invasion of the surrounding tissue or infiltration of the adjacent salivary gland [ 6 ].…”

“…Additionally, sialadenoma papilliferum is characterized by a combination of exophytic projection of the surface squamous epithelium and inward papillary proliferation of duct epithelial cells [ 1 , 2 , 6 , 7 ]. The underlying glandular component forms papillary extensions surfaced by columnar or cuboidal cells or both, which is contiguous with underlying arborizing ductal structures and cystic spaces.…”

“…Microscopically, intraductal papillomas, as described in Figure 1, are usually well-circumscribed unilocular lesion 2 Case Reports in Pathology exhibiting true intracystic papillary projections of ductal epithelium composed of small cuboidal cells arranged around a central lumen [2,3,5,6]. The lumen is partially or completely filled by branching papillary elements consisting of a fibrovascular core, and the epithelium that lines the cystic-like cavity is the same type of epithelium of papillary fronds, causing the mucus retention [2][3][4][5]7]. The presence of a varying degree of oncocytic metaplasia, as observed in the present case, is common in duct cysts arising from minor salivary glands [6].…”

“…The incidence of oral tumors arising from the salivary ductal glands, such as intraductal papillomas, is difficult to determine because different terminology has been used for the denomination of the same lesion [1]. Specifically, oral intraductal papilloma in minor salivary glands is a benign solitary tumor characterized by intracystic papillary growth and duct-like structures that affect predominantly the lip and buccal mucosa of the 50-year-old or older patients [1][2][3][4][5][6]. The occurrence of intraductal papilloma on the floor of the mouth is extremely rare, and there are only a few cases reported in the English literature [4,5].…”