Abstract
Sclerosing polycystic adenosis (SPA) is a rare condition of salivary glands. The most common site is the parotid gland (80 % of cases). SPA shows no gender predilection and occurs over a wide age spectrum (9–84 years). SPA is mostly unifocal, but may rarely be multifocal. Histologically, SPA are sharply circumscribed mostly unencapsulated lesions composed of acinar and ductal components with variable cytomorphological characteristics, including foamy, vacuolated, apocrine, mucous, clear/ballooned, squamous, columnar and oncocyte-like cells. Characteristic for SPA is the presence of large acinar cells with abundant eosinophilic cytoplasmic granules. The stroma is densely collagenized, frequently harbouring a variably intense chronic inflammatory infiltrate and may contain fat. Rarely the stroma is myxoid. Some degree of intraductal epithelial proliferations have been reported in at least 50 % of cases. The proportion of cases with epithelial proliferations that fulfill criteria for high-grade ductal carcinoma in-situ is <10 %. Immunohistochemically, both ductal and acinar cells are positive for broad spectrum cytokeratins. There is variable immunoreactivity for epithelial membrane antigen and S-100 protein. CEA, p53 and HER2 is reportedly negative. Gross cystic disease fluid protein-15 is strongly expressed in the acinar component. There is consistent but variable expression of estrogen and progesterone receptors. The proliferative index (Ki-67) is low (1–2 %) in the benign (acinar and ductal) components. Using HUMARA methodology (non-random inactivation of X-chromosomes), six cases with atypical epithelial proliferations have been shown to be clonal processes. Recurrences have been reported in up to 19 % of cases.
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Smith BC, Ellis GL, Slater LJ, et al. Sclerosing polycystic adenosis of major salivary glands. A clinicopathologic analysis of nine cases. Am J Surg Pathol. 1996;20:161–70.
Bharadwaj G, Nawroz I, O’Regan B. Sclerosing polycystic adenosis of the parotid gland. Br J Oral Maxillofac Surg. 2007;45:74–6.
Etit D, Pilch BZ, Osgood R, et al. Fine-needle aspiration biopsy findings in sclerosing polycystic adenosis of the parotid gland. Diagn Cytopathol. 2007;35:444–7.
Fulciniti F, Losito NS, Ionna F, et al. Sclerosing polycystic adenosis of the parotid gland: report of one case diagnosed by fine-needle cytology with in situ malignant transformation. Diagn Cytopathol. 2010;38:368–73.
Gnepp DR, Wang LJ, Brandwein-Gensler M, et al. Sclerosing polycystic adenosis of the salivary gland: a report of 16 cases. Am J Surg Pathol. 2006;30:154–64.
Gupta R, Jain R, Singh S, et al. Sclerosing polycystic adenosis of parotid gland: a cytological diagnostic dilemma. Cytopathology. 2009;20:130–2.
Gurgel CA, Freitas VS, Ramos EA, et al. Sclerosing polycystic adenosis of the minor salivary gland: case report. Braz J Otorhinolaryngol. 2010;76:272.
Imamura Y, Morishita T, Kawakami M, et al. Sclerosing polycystic adenosis of the left parotid gland: report of a case with fine needle aspiration cytology. Acta Cytol. 2004;48:569–73.
Meer S, Altini M. Sclerosing polycystic adenosis of the buccal mucosa. Head Neck Pathol. 2008;2:31–5.
Noonan VL, Kalmar JR, Allen CM, et al. Sclerosing polycystic adenosis of minor salivary glands: report of three cases and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007;104:516–20.
Perottino F, Barnoud R, Ambrun A, et al. Sclerosing polycystic adenosis of the parotid gland: diagnosis and management. Eur Ann Otorhinolaryngol Head Neck Dis. 2010;127:20–2.
Skalova A, Gnepp DR, Simpson RH, et al. Clonal nature of sclerosing polycystic adenosis of salivary glands demonstrated by using the polymorphism of the human androgen receptor (HUMARA) locus as a marker. Am J Surg Pathol. 2006;30:939–44.
Skalova A, Michal M, Simpson RH, et al. Sclerosing polycystic adenosis of parotid gland with dysplasia and ductal carcinoma in situ. Report of three cases with immunohistochemical and ultrastructural examination. Virchows Arch. 2002;440:29–35.
Mackle T, Mulligan AM, Dervan PA, et al. Sclerosing polycystic sialadenopathy: a rare cause of recurrent tumor of the parotid gland. Arch Otolaryngol Head Neck Surg. 2004;130:357–60.
Tokyol C, Aktepe F, Hasturk GS, et al. Sclerosing polycystic adenosis of the parotid gland presenting with a Warthin tumor. Kulak Burun Bogaz Ihtis Derg. 2012;22:288–92.
Kim BC, Yang DH, Kim J, et al. Sclerosing polycystic adenosis of the parotid gland. J Craniofac Surg. 2012;23:e451–2.
Kloppenborg RP, Sepmeijer JW, Sie-Go DM, et al. Sclerosing polycystic adenosis: a case report. B-Ent. 2006;2:189–92.
Eliot CA, Smith AB, Foss RD. Sclerosing polycystic adenosis. Head Neck Pathol. 2012;6:247–9.
Petersson F, Tan PH, Hwang JS. Sclerosing polycystic adenosis of the parotid gland: report of a bifocal, paucicystic variant with ductal carcinoma in situ and pronounced stromal distortion mimicking invasive carcinoma. Head Neck Pathol. 2011;5:188–92.
Beato Martinez A, Moreno Juara A, Candia Fernandez A. Sclerosing polycystic adenosis of the submandibular gland. Acta Otorrinolaringol Esp.
Swelam WM. The pathogenic role of Epstein-Barr virus (EBV) in sclerosing polycystic adenosis. Pathol Res Pract. 2010;206:565–71.
Boecker W, Moll R, Dervan P, et al. Usual ductal hyperplasia of the breast is a committed stem (progenitor) cell lesion distinct from atypical ductal hyperplasia and ductal carcinoma in situ. J Pathol. 2002;198:458–67.
Lacroix-Triki M, Mery E, Voigt JJ, et al. Value of cytokeratin 5/6 immunostaining using D5/16 B4 antibody in the spectrum of proliferative intraepithelial lesions of the breast. A comparative study with 34betaE12 antibody. Virchows Arch. 2003;442:548–54.
Otterbach F, Bankfalvi A, Bergner S, et al. Cytokeratin 5/6 immunohistochemistry assists the differential diagnosis of atypical proliferations of the breast. Histopathology. 2000;37:232–40.
Gnepp DR. Sclerosing polycystic adenosis of the salivary gland: a lesion that may be associated with dysplasia and carcinoma in situ. Adv Anat Pathol. 2003;10:218–22.
Endoh Y, Tamura G, Kato N, et al. Apocrine adenosis of the breast: clonal evidence of neoplasia. Histopathology. 2001;38:221–4.
Kazakov DV, Bisceglia M, Sima R, et al. Adenosis tumor of anogenital mammary-like glands: a case report and demonstration of clonality by HUMARA assay. J Cutan Pathol. 2006;33:43–6.
Acknowledgments
The author wishes to thank Professor Ken Berean, Department of Pathology, Surrey Memorial Hospital, University of British Columbia, Canada, for kindly providing some of the material that was used for the photomicrographs.
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Petersson, F. Sclerosing Polycystic Adenosis of Salivary Glands: A Review with Some Emphasis on Intraductal Epithelial Proliferations. Head and Neck Pathol 7 (Suppl 1), 97–106 (2013). https://doi.org/10.1007/s12105-013-0465-9
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DOI: https://doi.org/10.1007/s12105-013-0465-9