Intraoral Pigmented Low-Grade Adenocarcinoma, Not Otherwise Specified: Case Report and Immunohistochemical Study

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Intraoral Pigmented Low-Grade Adenocarcinoma, Not Otherwise Specified: Case Report and Immunohistochemical Study Jessica Luana dos Santos1 · Albina Messias de Almeida Milani Altemani2 · Alexandre Elias Trivellato3 · Cássio Edvard Sverzut3 · Luciana Yamamoto Almeida1 · Lucas Ribeiro Teixeira1 · Alfredo Ribeiro‑Silva1 · Jorge Esquiche León4 Received: 2 August 2017 / Accepted: 8 December 2017 © Springer Science+Business Media, LLC, part of Springer Nature 2017

Abstract Salivary adenocarcinoma, not otherwise specified (AdCaNOS) is a rare malignant tumor with potential diagnostic challenge, which mainly affects the parotid glands; however, the minor salivary glands can also be involved by AdCaNOS. This paper reports a case of a 45-year-old Afro-descendant woman complaining of a slow-growing mass with 6 months of evolution in the left superior vestibular fornix. Microscopic examination revealed an infiltrative epithelial neoplasm composed of predominantly solid growth pattern, arranged in a lobular configuration, admixed with glandular or ductal structures. Perineural invasion was evident. The tumor cells were polygonal or oval showing focally mild nuclear pleomorphism, and eosinophilic or clear cytoplasm. Notably, some areas exhibited intracytoplasmic pigment granules mainly in non-luminal cells, as well as sebaceous-like cells, discrete hyaline material deposition and foci of infiltration of residual salivary gland parenchyma. Tumor cells were negative for PAS, mucicarmine and Alcian blue stains. By immunohistochemistry, the tumor cells were diffuse and strongly positive for pan-cytokeratin (CK) AE1/AE3, 34betaE12 CK, vimentin, p63 and S100. CK7 and EMA strongly highlighted the ductal structures. Solid areas also showed diffuse and moderate expression of CD56. Podoplanin (D2-40), GFAP and Calponin, followed by DOG-1, were focally positive; whereas CK20, α-SMA, h-Caldesmon, CD57, ERBB2/HER2 and p53 were negative. Ki-67 was