Synchronous ipsilateral sebaceous lymphadenoma and acinic cell adenocarcinoma of the parotid gland Marta Mayorga, MD, Nuria Fernández, MD, and J. Fernando Val-Bernal, MD, Santander, Spain UNIVERSITY OF CANTABRIA
The synchronous or metachronous occurrence of 2 tumors of the salivary glands in a patient is rare. These tumors are usually benign and of the same histologic type. Here we report a 78-year-old woman who developed a sebaceous lymphadenoma and an acinic cell adenocarcinoma simultaneously in the left parotid gland. To our knowledge, this combination of neoplasms has not been reported before. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999;88:593-6)
Primary epithelial tumors involving major salivary glands occur most frequently in the parotid gland. The majority of these tumors are unifocal, unilateral lesions and histologically single neoplasms. The coexistence of tumors of different histologic types is an extraordinary event in the salivary glands. Synchronous ipsilateral or bilateral parotid tumors of the same histologic type are found occasionally.1 The occurrence of 2 coexisting parotid tumors of different histologic types is rare.2-14 The purpose of this report is to describe a patient with a simultaneous and unilateral sebaceous lymphadenoma and acinic cell adenocarcinoma of the parotid gland. As far as we know, this combination of tumors has not been described.
CASE REPORT A 78-year-old woman was first seen with a painless, slowly enlarging swelling of the right parotid region, which had increased in size over 2 months. Physical examination revealed a nontender, firm, nodular mass estimated to measure 3 × 3 cm. No facial nerve palsy, cervical lymphadenopathy, or oral lesions were noted. The results of routine laboratory tests were within the normal range. Fine needle aspiration biopsy disclosed clusters of large cells with abundant, finely granular cytoplasm, indistinct borders, and large, hyperchromatic, round to ovoid, centrally placed nuclei. After the diagnosis of acinic cell adenocarcinoma was made, a superficial parotidectomy was performed. The postoperative course was uneventful. The patient complained of gustatory sweating, flushing, and a feeling of warmth in the area of sensory distribution of the auriculotemporal nerve 13 months after surgery.
Department of Anatomical Pathology, Marqués de Valdecilla University Hospital, Medical Faculty, University of Cantabria, Santander, Spain. Received for publication Apr 8, 1999; returned for revision May 26, 1999; accepted for publication Aug 2, 1999. Copyright © 1999 by Mosby, Inc. 1079-2104/99/$8.00 + 0 7/14/101852
Fig 1. Gross specimen showing 2 separate tumors found in the parenchyma of right parotid gland. The larger tumor is sebaceous lymphadenoma and the smaller, cystic lesion, an acinic cell adenocarcinoma. The sebaceous lymphadenoma has spongy appearance.
METHODS The resected surgical specimen was fixed in 10% buffered formalin, and representative samples were embedded in paraffin. Sections were stained with hematoxylin-eosin and periodic acid-Schiff reaction (PAS) stain with and without diastase digestion. Frozen 593
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B Fig 2. Sebaceous lymphadenoma. A, Sebaceous differentiation within epithelial islands and lymphoid stroma are evident (hematoxylin-eosin, original magnification ×25). B, Tumor epithelium, including sebaceous component, shows reactivity for epithelial membrane antigen. Sebaceous cells display bubbly pattern (hematoxylin-eosin, original magnification ×25).
tissue sections were stained with oil red O. Selected paraffin blocks were prepared for immunohistochemical staining by a streptavidin-biotin-peroxidase technique by using diaminobenzidine as the chromogen and Harris hematoxylin as the counterstain. The antibodies used and their clonality, manufacturers, and dilutions were as follows: epithelial membrane antigen (EMA, clone E29, Dako A/S, Glostrup, Denmark, 1:50), α1-antitrypsin (A1AT, polyclonal, Dako A/S, 1:500) and α1-antichymotrypsin (A1ACT, polyclonal, Dako A/S, 1:500).
PATHOLOGY The surgical specimen measured 7 × 6 × 3.5 cm. It consisted of glandular tissue surrounded by some fat tissue. The cut sections revealed a well-circumscribed, yellow-white, multicystic and soft nodule measuring 2 × 1.8 cm. Close to this nodule and separated by about 1 cm of parotid glandular tissue was a sharply demarcated
cystic mass containing brown semisolid debris. This mass measured 1.5 cm in diameter (Fig 1). Histologic examination disclosed 2 distinct tumors: a larger sebaceous lymphadenoma and a smaller acinic cell adenocarcinoma. The tumors were separated from each other by glandular tissue containing acini and ducts. The sebaceous lymphadenoma consisted of islands, ducts, and small-to-medium–sized cysts composed of columnar or cuboidal oncocytic cells. Sebaceous differentiation in focal areas was observed in the lining of the cysts and within the islands. The stroma between the epithelial elements was lymphoid, and lymphoid follicles were evident (Fig 2, A). The sebaceous cells showed abundant lipid in frozen sections stained with oil red O. EMA (Fig 2, B) immunostaining revealed a loculated pattern of the lesional cells. The acinic cell adenocarcinoma showed a papillarycystic pattern. It consisted of a large cystic space lined by epithelium of variable thickness and branching
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Fig 3. Acinic cell adenocarcinoma. A, Papillary-cystic growth pattern (hematoxylin-eosin, original magnification ×64). B, Microcystic area showing granular and acinar vacuolated cells (hematoxylin-eosin, original magnification ×100).
projections of epithelium occupying a portion of the cyst’s lumen (Fig 3, A). The cells were cuboidal to polygonal with granular, eosinophilic, or vacuolated cytoplasm and uniform, round, eccentric nuclei (Fig 3, B). The PAS stain highlighted the cytoplasmic granules, which were resistant to diastase digestion. Mitotic activity was 4 mitoses per 10 high-power fields (where 1 high-power field = 0.1225 mm2). Significant nuclear atypia, necrotic foci, and vascular invasion were not seen. The neoplastic cells showed reactivity for A1AT and A1ACT. Surgical margin was uninvolved in both tumors.
DISCUSSION The fact that this patient had 2 tumors was not recognized at the time of clinical evaluation. Only the smaller, malignant neoplasm was detected. Gross pathologic examination showed 2 separate, texturally different neoplasms. The presence of the second (benign) tumor came as a surprise.
In the literature, some tumors are described as arising in other neoplasms, that is, a malignant mixed tumor arising in a benign mixed tumor, as opposed to distinct, coexisting tumors. Warthin’s tumor, pleomorphic adenoma, oncocytoma, acinic cell adenocarcinoma, and basal cell adenoma are the tumors most commonly presenting as synchronous unilateral or bilateral parotid neoplasms of the same histologic type.15 In contrast, synchronous bilateral parotid gland tumors of different histologic types are found only rarely. The combination of Warthin’s tumor with pleomorphic adenoma is the most common situation, representing 42% of such cases.16 The synchronous, unilateral occurrence of sebaceous lymphadenoma and acinic cell adenocarcinoma of the parotid gland has not been reported, to our knowledge. Sebaceous lymphadenoma is a rare, benign tumor that is composed of nests and islands of epithelium, including sebaceous elements, surrounded by lymphoid stroma. The lipophilic dye, oil red O, helps to identify the seba-
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ceous cells in frozen tumor sections. The presence of milk fat globule-associated proteins in neoplastic sebaceous cells can be demonstrated by the monoclonal antibody E29. These sebaceous cells characteristically exhibit a bubbly pattern of EMA reactivity. The sebaceous lymphadenoma represents 0.1% of all salivary gland adenomas, and 92% of the cases arise in the parotid gland.17 A case of this neoplasm has been described as being associated with Warthin’s tumor.18 Acinic cell carcinoma comprises about 6% of all salivary gland tumors and 17% of primary malignant salivary gland neoplasms. Up to 3% are bilateral, although some bilateral cases that have been reported may represent the clear cell variant of oncocytoma. Women are affected more commonly than men by a ratio of 3:2.17 Histologic diagnosis is based on the acinar differentiation of the tumor cells. Serous differentiation is demonstrated by the presence of typical cytoplasmic granules that are highlighted with the PAS stain. According to Spiro et al,19 the papillary cystic pattern of the acinic cell adenocarcinoma should be recognized because it has such a poor prognosis. However, other investigators dispute this assertion.20,21 In our case, the tumor was encapsulated and showed no vascular invasion. The excision of the acinic cell adenocarcinoma was complete, with an adequate margin of noninvolved tissue. Accordingly, the prognosis of our patient is expected to be favorable. In conclusion, this appears to be the first documented case of synchronous unilateral sebaceous lymphadenoma and acinic cell adenocarcinoma in the parotid gland.
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Reprint requests: J. Fernando Val-Bernal, MD, PhD Departamento de Anatomía Patológica Hospital Universitario Marqués de Valdecilla Avda Valdecilla, 1 39008 Santander Spain