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

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          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.

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          Author and article information

          Contributors
          craig.fowler@uky.edu
          Journal
          Head Neck Pathol
          Head Neck Pathol
          Head and Neck Pathology
          Springer US (New York )
          1936-055X
          1936-0568
          8 September 2017
          June 2018
          : 12
          : 2
          : 193-201
          Affiliations
          ISNI 0000 0004 1936 8438, GRID grid.266539.d, Division of Oral and Maxillofacial Pathology, , University of Kentucky College of Dentistry, ; Lexington, KY USA
          Article
          PMC5953874 PMC5953874 5953874 852
          10.1007/s12105-017-0852-8
          5953874
          28887760
          d75c1a9d-57ca-4192-b11d-271729304cc6
          © Springer Science+Business Media, LLC 2017
          History
          : 20 January 2017
          : 2 September 2017
          Categories
          Original Paper
          Custom metadata
          © Springer Science+Business Media, LLC, part of Springer Nature 2018

          Papilliferum,Syringocystadenoma,Salivary gland neoplasm,Sialadenoma papilliferum

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