Journal of Cytology
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CASE REPORT  
Year : 2014  |  Volume : 31  |  Issue : 4  |  Page : 221-223
Oncocytic papillary cystadenoma of major salivary glands: Three rare cases with diverse cytologic features


Department of Pathology, Soonchunhyang University College of Medicine, Bucheon, Korea

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Date of Web Publication10-Feb-2015
 

   Abstract 

Oncocytic papillary cystadenoma (OPC) in the major salivary glands is extremely rare. We report three cases of these cystadenomas arising in major salivary glands, with varied cytologic features. Case 1: A 38-year-old man presented with a right parotid gland mass that showed papillary clusters of oncocytic cells on cytologic examination. Case 2: An 84-year-old man presented with a left parotid gland mass. Cytology revealed an acellular smear. Case 3: A 57-year-old man presented with a mass in the right submandibular gland. Cytology revealed irregular sheets of epithelial cells. Histologic diagnoses of OPC were made for all three cases.

Keywords: Major salivary gland; oncocytic papillary cystadenoma; parotid; submandibular

How to cite this article:
Chin S, Kim HK, Kwak JJ. Oncocytic papillary cystadenoma of major salivary glands: Three rare cases with diverse cytologic features. J Cytol 2014;31:221-3

How to cite this URL:
Chin S, Kim HK, Kwak JJ. Oncocytic papillary cystadenoma of major salivary glands: Three rare cases with diverse cytologic features. J Cytol [serial online] 2014 [cited 2020 Feb 25];31:221-3. Available from: http://www.jcytol.org/text.asp?2014/31/4/221/151140



   Introduction Top


Papillary cystadenoma of the salivary glands is an uncommon benign neoplasm, [1] which usually occurs in the minor salivary glands, [2],[3],[4] and very rarely in the major salivary glands. Extensive oncocytic change may be seen, and these lesions are classified as oncocytic variants. We report three cases of oncocytic papillary cystadenoma (OPC) arising in the major salivary glands.


   Case Reports Top


Case 1

Case 1 was a 38-year-old man who had had a mass on the right neck for 1-month. Palpation revealed a 3-cm firm mass in the right parotid gland region. Fine needle aspiration (FNA) was performed, and the cytologic smear showed irregular sheets and papillary clusters of epithelial cells [[Figure 1]a], with rich granular cytoplasm, bland-appearing nuclei, and conspicuous nucleoli [[Figure 1]b]. A Warthin tumor was suspected based on these findings. Computed tomography (CT) revealed a cystic mass with a slight enhancement in the right parotid gland, and the patient underwent right superficial parotidectomy. Gross examination showed a well-defined 2.5-cm unilocular cystic mass with multiple papillary projections. Microscopically, the epithelial lining comprised cuboidal to low-columnar cells [[Figure 1]c], with abundant granular, eosinophilic cytoplasm, and vesicular nuclei with prominent nucleoli [[Figure 1]d].
Figure 1: (a) Cytologic smear revealing many oncocytic cells in papillary c d clusters (Pap, x100), (b) with abundant, granular cytoplasm and prominent nucleoli (Pap, x400), (c) histological analysis revealed a multi locular cystic tumor fi lled with papillary projections (H and E, x40), (d) the papillae were lined by oncocytic cells with eosinophilic, granular cytoplasm), (H and E, ×200)

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Case 2

Case 2 was an 84-year-old man who had had a slow-growing mass on the left neck for 1-year. Palpation revealed a firm, tender mass in the left parotid gland area, 4 cm in size. FNA was performed, and the cytologic smear revealed a few scattered foamy macrophages and some inspissated proteinaceous material without epithelial cells, suggesting a cystic neoplasm [[Figure 2]a and b]. A CT scan revealed a 3.1 cm × 2.7 cm septated cystic mass in the superficial lobe of the left parotid gland. The patient underwent left superficial parotidectomy, revealing a multilocular cystic tumor with papillary projections. Microscopically, the tumor appeared as a well-defined cystic mass, with multiple papillary fronds lined by tall columnar cells [[Figure 2]c]. The cells had rich granular, eosinophilic cytoplasm, and bland-appearing nuclei with conspicuous nucleoli [[Figure 2]d].
Figure 2: (a and b) Cytology revealed inspissated proteinaceous material without epithelial cells (a: Pap, x100; b: Pap, x400), and (c) histology revealed a cystic mass with papillary projections in the lumen (H and E stain, ×40); (d) the tumor cells were columnar with rich eosinophilic granular cytoplasm (H and E stain, ×200)

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Case 3

Case 3 was a 57-year-old man who presented with a swelling on the right upper neck, which had slowly increased in size over 1-year. On palpation, a slightly tender 2-cm mass was found below the right submandibular angle. FNA was performed, and the cytologic smear showed irregular fragments of epithelial cells [[Figure 3]a]. The cells had moderate amounts of cytoplasm and bland-looking nuclei with inconspicuous nucleoli [[Figure 3]b]. Cytologic examination suggested a benign epithelial tumor, and a CT scan revealed a 3-cm cystic mass containing an inner solid portion. The patient underwent right submandibular gland resection. A 2.4-cm well-circumscribed cystic mass filled with papillary projections was found in the resected submandibular gland. Microscopy revealed a unilocular cyst with multiple papillary fronds lined by columnar epithelial cells, which had abundant eosinophilic cytoplasm and prominent nucleoli [[Figure 3]c and d]. All three cases were diagnosed as OPC.
Figure 3: (a) Cytologic smear revealed irregular fragments of epithelial cells (Pap, x100), (b) the cells had moderate amount cytoplasm and inconspicuous nucleoli (Pap, x400), (c) histology revealed a cystic mass fi lled with papillary fronds (H and E, ×40), (d) which were lined by columnar, oncocytic cells with prominent nucleoli (H and E, x200)

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   Discussion Top


Cystadenoma of the salivary glands is a rare benign tumor, usually presenting as a slow-growing mass. The tumor usually arises in the minor salivary glands (0.9-2% of all minor salivary gland neoplasms) but is extremely rare in the major salivary gland. A review reported only three cases of the tumor arising in major salivary glands, [2] and Tsurumi et al. [5] found that all four reported cases of salivary gland cystadenoma, between 1985 and 2003, had arisen in the minor salivary glands.

Cystadenoma can be subdivided into mucinous and papillary types. The latter has well-defined unilocular or multilocular cysts, with intraluminal papillary projections. [4],[6] The lining epithelial cells are cuboidal or columnar, and usually two layers in thickness. Oncocytic differentiation occurs in 1% of tumors and can be extensive. [7]

Oncocytic papillary cystadenoma is often confused with Warthin tumor, because of its histologic similarity; papillary structures, oncocytic cells with rich eosinophilic cytoplasm, and prominent nucleoli. However, Warthin tumors can be distinguished by their lymphoid-rich stroma. OPC may also be misdiagnosed as intraductal papilloma as the latter often has cystically dilated ducts with papillary epithelial projections. However, the papillary cores of intraductal papilloma are more complex and prominent than OPC. Another differential diagnosis is oncocytoma, which is characterized by polyhedral oncocytic cells. Cystic change may be present in this tumor, but the papillary pattern, typical of OPC, is not a feature of oncocytoma.

Cytologic findings of OPC have rarely been reported. Zhang et al.[8] reported diverse cytologic features of two cases of the tumor, one of which showed a cellular smear of oncocytic cells with prominent nucleoli and many histiocytes, and the other showed abundant necrotic debris, and large sheets of polygonal cells with vague papillary features and inconspicuous nucleoli. Gupta et al.[9] described somewhat different cytologic findings from two FNAs taken from the same OPC, with either atypical squamous cells in a necrotic background, consistent with squamous cell carcinoma, or many vacuolated and signet ring cells in a mucoid background. No oncocytic cells or papillary features were seen in either aspirate. Nasuti et al.[10] described a case of OPC in which the aspirate was insufficient for diagnosis because of paucicellular smear. Our cases also revealed varied cytologic features. The first and third case revealed a cellular smear. The first case showed distinct papillary clusters of oncocytic cells and the third case had irregular sheets of epithelial cells without identifiable oncocytic change. These contrast with the second case, in which only proteinous material without epithelial cells was found. It is assumed that the acellular or paucicellular smear of papillary cystadenoma is a result of the dilution of cystic fluid.

In summary, OPC is extremely rare in major salivary glands and tends to show varied cytologic features. Although cytologic diagnosis is challenging, the tumor should be included in the differential diagnosis when oncocytic cells or papillary features are present in cystic tumors of salivary glands.



 
   References Top

1.
Toida M, Shimokawa K, Makita H, Kato K, Kobayashi A, Kusunoki Y, et al. Intraoral minor salivary gland tumors: A clinicopathological study of 82 cases. Int J Oral Maxillofac Surg 2005;34:528-32.  Back to cited text no. 1
    
2.
Guccion JG, Redman RS, Calhoun NR, Saini N. Papillary cystadenoma of the palate: A case report and ultrastructural study. J Oral Maxillofac Surg 1997;55:759-64.  Back to cited text no. 2
    
3.
Matsuzaka K, Kokubu E, Takeda E, Tanaka Y, Shimono M, Inoue T. Papillary cystadenoma arising from the upper lip: A case report. Bull Tokyo Dent Coll 2003;44:213-6.  Back to cited text no. 3
    
4.
Lim CS, Ngu I, Collins AP, McKellar GM. Papillary cystadenoma of a minor salivary gland: Report of a case involving cytological analysis and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:e28-33.  Back to cited text no. 4
    
5.
Tsurumi K, Kamiya H, Yokoi M, Kameyama Y. Papillary oncocytic cystadenoma of palatal minor salivary gland: A case report. J Oral Maxillofac Surg 2003;61:631-3.  Back to cited text no. 5
    
6.
Gallego L, Junquera L, Fresno MF, de Vicente JC. Papillary cystadenoma and cystadenocarcinoma of salivary glands: Two unusual entities. Med Oral Patol Oral Cir Bucal 2008;13:E460-3.  Back to cited text no. 6
    
7.
Ellis G, Auclair P. Tumor of the salivary gland. AFIP Atlas of Tumor Pathology. 4 th ed. Washington, DC: ARP Press; 2008.  Back to cited text no. 7
    
8.
Zhang S, Bao R, Abreo F. Papillary oncocytic cystadenoma of the parotid glands: a report of 2 cases with varied cytologic features. Acta Cytol 2009;53:445-8.  Back to cited text no. 8
    
9.
Gupta S, Sodhani P, Jain S, Khurana N, Agarwal PN. Oncocytic papillary cystadenoma of parotid gland: A diagnostic challenge on fine-needle aspiration cytology. Diagn Cytopathol 2011;39:627-30.  Back to cited text no. 9
[PUBMED]    
10.
Nasuti JF, Gupta PK, Fleisher SR, LiVolsi VA. Nontyrosine crystalloids in salivary gland lesions: Report of seven cases with fine-needle aspiration cytology and follow-up surgical pathology. Diagn Cytopathol 2000;22:167-71.  Back to cited text no. 10
    

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Correspondence Address:
Jeong Ja Kwak
Department of Pathology, Soonchunhyang University College of Medicine, 170 Jomaru-ro, Wonmi-gu, Bucheon 420-767
Korea
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Source of Support: This work was supported in part by the Soonchunhyang University Research Fund, Conflict of Interest: None


DOI: 10.4103/0970-9371.151140

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