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

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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 < 2%. Consecutive serial tissue sections using CD57 confirmed the perineural invasion. Positivity for HMB-45 and MART-1/Melan-A, as well as Fontana-Masson stain (and potassium permanganate bleaching-sensitive), identified the pigment granules as melanin. To the best of our knowledge, this is the first case of intraoral low-grade AdCaNOS with intracytoplasmic melanin granules.

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Funding

Jorge Esquiche León has received research Grants from State of São Paulo Research Foundation (2011/52090-8 and 2016/11419-0).

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Authors and Affiliations

  1. Oral Pathology, Department of Pathology and Forensic Medicine, Ribeirão Preto Medical School (FMRP/USP), University of São Paulo, Avenida do Café, S/N, Ribeirão Preto, São Paulo, 14040-904, Brazil

    Jessica Luana dos Santos, Luciana Yamamoto Almeida, Lucas Ribeiro Teixeira & Alfredo Ribeiro-Silva

  2. Pathology Division, Department of Pathology, Faculty of Medical Sciences, State University of Campinas, Rua Tessália Vieira de Camargo, 126, Campinas, São Paulo, 13083-887, Brazil

    Albina Messias de Almeida Milani Altemani

  3. Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Surgery and Periodontology, Ribeirão Preto Dental School (FORP/USP), University of São Paulo, Avenida do Café, S/N, Ribeirão Preto, São Paulo, 14040-904, Brazil

    Alexandre Elias Trivellato & Cássio Edvard Sverzut

  4. Oral Pathology, Department of Stomatology, Public Oral Health, and Forensic Dentistry, Ribeirão Preto Dental School (FORP/USP), University of São Paulo, Avenida do Café, S/N, Ribeirão Preto, São Paulo, 14040-904, Brazil

    Jorge Esquiche León

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  1. Jessica Luana dos Santos
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Correspondence to Jorge Esquiche León.

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dos Santos, J.L., de Almeida Milani Altemani, A.M., Trivellato, A.E. et al. Intraoral Pigmented Low-Grade Adenocarcinoma, Not Otherwise Specified: Case Report and Immunohistochemical Study. Head and Neck Pathol 12, 610–618 (2018). https://doi.org/10.1007/s12105-017-0875-1

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