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CASE REPORT
Year : 2012  |  Volume : 1  |  Issue : 3  |  Page : 179-181

Cystadenoma: A perplexing entity with subtle literature


Department of Oral Pathology, St. Joseph Dental College & Hospital, Duggirala, Eluru, Andhra Pradesh, India

Date of Web Publication15-Oct-2012

Correspondence Address:
Anuradha Ananthaneni
D/No - 10/200, Anuradha ENT Hospital, Eluru Road, Gudivada, Krishna District - 521 301, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2277-8632.102448

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  Abstract 

Cystadenoma is an uncommon, benign cystic epithelial neoplasm containing epithelium-lined papillary projections into the cystic spaces. A 45-year-old female presented with a swelling on the left side of the roof of her mouth. Clinically, the lesion was nontender, soft, and fluctuant. A fine-needle aspiration biopsy was suggestive of a minor salivary gland neoplasm. An excision biopsy showed characteristic features of cystadenoma. This paper highlights its cytological and histopathological features with differential diagnosis. Cystadenoma of the palate is rare with very few earlier reports.

Keywords: Adenoma, cystic, salivary gland


How to cite this article:
Ananthaneni A, Kashyap B, Prasad VR, Srinivas V. Cystadenoma: A perplexing entity with subtle literature. J NTR Univ Health Sci 2012;1:179-81

How to cite this URL:
Ananthaneni A, Kashyap B, Prasad VR, Srinivas V. Cystadenoma: A perplexing entity with subtle literature. J NTR Univ Health Sci [serial online] 2012 [cited 2019 Nov 18];1:179-81. Available from: http://www.jdrntruhs.org/text.asp?2012/1/3/179/102448


  Introduction Top


Salivary gland neoplasms represent less than 1% of all tumors, and 3-5% of all head and neck neoplasms. Minor salivary gland tumors are infrequent, accounting for 10-15% of all salivary neoplasms. Cystadenoma of the salivary glands is a rare benign neoplasm in which the epithelial proliferation is characterized by the formation of multiple cystic structures. It represents 0.7-8.1% of all benign salivary gland tumors, 7% of all minor salivary gland tumors, and 3.1% of major salivary gland benign tumors. Almost 58% of these tumors occur in the parotid gland, 6.6% occur in the submandibular gland, 0.5% in the sublingual gland, 10.7% on the lips, 8.2% on the cheeks, 7.1% in the palate, and 9.1% in other intraoral sites. They are most common in the eighth decade of life with a mean age of occurrence of 55 years. There is a female predilection in the ratio of 2:1. Distribution in the minor salivary gland is more often on the lips, cheeks, and the palate. Clinically, they are asymptomatic and produce smooth slowly enlarging nodules usually <1 cm that may be compressible. Conservative surgical removal is the opted modality of treatment. [1],[2],[3],[4],[5],[6],[7],[8] Here, we report a case of cystadenoma of the palate, in a 45-year-old female with special emphasis on the cytological and histopathological features with differential diagnosis.


  Case Report Top


A 45-year-old female reported to the outpatient department, complaining of swelling in the upper left back tooth region since one year. History revealed extraction of teeth 26 and 27 one year back due to caries. Past medical history was noncontributory. The intraoral examination revealed a solitary swelling of size 1.5-2 cm in the left side of the hard palate, extending anterioposteriorly from the palatal aspect of teeth 25-28. Mediolaterally, the swelling extended 1 cm from midline to the mucogingival junction of the posterior teeth. The surface mucosa was intact. On palpation, the swelling was soft, fluctuant, nontender, and not fixed to the underlying tissues. No cervical lymphadenopathy was observed. Based on the history and clinical features, it was provisionally diagnosed as a benign salivary gland neoplasm. Routine blood investigations were within normal limits and there was no radiographic change. Fine-needle aspiration cytology yielded thick, yellow, puslike fluid. The smears showed mucoid material with occasional epithelial cells containing a regular round or oval nucleus with scant cytoplasm suggestive of a minor salivary gland tumor.

Complete surgical excision was done under local anesthesia with an uneventful postoperative recovery, and the resected tissue was submitted for histopathological diagnosis. Grossly, the specimen was black in color and soft in consistency. Microscopically, it showed multiple cystic areas adjacent to normal-appearing mucous salivary glands. The cystic spaces were lined partly by a bilayered columnar epithelium and partly by one to two layers of flattened cells in some areas with bland uniform nuclei. Focally, the epithelium showed thickening with papillary projections containing a central core of connective tissue. Mucinous changes were evident near the epithelial thickening. No cellular atypia was observed. Focal collection of chronic inflammatory cells was also evident. A histopathological diagnosis of papillary cystadenoma was established after excluding some of the lesions with analogous histopathological presentation [Figure 1], [Figure 2], [Figure 3].
Figure 1: Multiple cystic areas with papillary projections containing a central core of connective tissue

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Figure 2: Cystic spaces lined partly by a bilayered columnar epithelium and partly by one to two layers of flattened cells

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Figure 3: Focal epithelial thickenings

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  Discussion Top


Salivary gland cystadenoma is a rare benign tumor. An appraisal of the literature showed a number of incompatible interpretations and inconsistencies in terminology regarding these tumors. Many investigators include these tumors under the general heading of monomorphic adenoma or consider them to be reactive rather than neoplastic. Thackray and Lucas do not regard cystadenoma as a separate entity as they consider it as a primarily cystic tumor in which formation of the cyst is incidental. Cystadenoma is divided into papillary and mucinous types by the World Health Organization (WHO). The WHO defines papillary cystadenoma as a tumor that closely resembles Warthin's tumor but without the lymphoid elements, constituting multiple papillary projections and a greater variety of epithelial lining cells. If mucous cells predominate in the cell population of the lining epithelial cells, the tumor is termed as mucinous cystadenoma. [6],[7]

Majority of the palatal cases of cystadenoma are in the soft palate with a male predilection. Papillary cystadenomas of salivary gland origin have also been described in the larynx, lacrimal glands, body of the mandible, and nasopharynx. Furthermore, analogous lesions have been reported in other anatomic sites such as the epididymis, ovary, and kidney. Cystadenoma of minor salivary glands presents no discrete clinical features and closely resembles a mucocele. [7],[8],[9],[10]

Lim et al. were the foremost to provide a cytological description of smears prepared from fine-needle aspiration of papillary cystadenoma. [8] In this case, the cytology could direct us only to an interim diagnosis which was not definitive. The ultimate histopathological diagnosis of this rare minor salivary gland neoplasm required the elimination of similar lesions such as simple cyst, ductal ectasia, canalicular adenoma, intraductal papilloma, Warthin's tumor, low-grade mucoepidermoid carcinoma, and polycystic disease. Ductal ectasia and simple cyst rarely show prominent epithelial thickening and well-formed papillary projections, which are significant in our case. In addition, duct ectasia usually involves several ductal segments and is associated with fibrosis, chronic inflammation, and atrophy of the adjacent gland. Canalicular adenomas demonstrate large cystic areas with papillary proliferations but most commonly they present as single-layered cords or columnar or cuboidal epithelial cells with deeply basophilic nuclei with prominent vascularity in a loose connective tissue stroma. Intraductal papillomas are unicystic with intraluminal proliferations. These incongruous features ruled out canalicular adenoma and intraductal papilloma.

Absence of lymphoid aggregates with a germinal center and double-row tall columnar and low cuboidal cell lining of the epithelium ruled out Warthin's tumor as a diagnosis. Low-grade mucoepidermoid carcinoma presents, in addition to cystic structures, noncystic epithelial proliferations. The papillary growth is irregular and complex, as observed in intraductal papilloma. Moreover, the cell population in low-grade mucoepidermoid carcinoma exhibits a combination of epidermoid, mucosal, and, to a lesser extent, intermediate and basaloid cells which were not apparent in the present case. Polycystic disease is extremely rare and involves the entire gland more diffusely than does the well-circumscribed cystadenoma. Cystadenocarcinoma was eliminated based on the pattern of solid growth in focal areas, mode of invasion, cellular atypia in some cases, permeation or destruction of the glandular parenchyma, and breakdown of the glandular lobe architecture, as well as infiltration of adipose, muscle, or bone tissues.

A final diagnosis of cystadenoma was arrived at based on the insight of the pathologist; otherwise, these lesions are often interpreted as a hyperplasic process of the salivary ducts which may be the cause for the low incidence in the specialty literature. Nevertheless, clinically and histologically, the tumor displays a benign behavior with rare recurrences reported, which may be accredited to incomplete surgical excision.

 
  References Top

1.Speight PM, Barrett AW. Salivary gland tumours. Oral Dis 2002;8:229-40.  Back to cited text no. 1
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2.Yih WY, Kratochvil FJ, Stewart JC. Intraoral minor salivary gland neoplasms: Review of 213 cases. J Oral Maxillofac Surg 2005;63:805-10.  Back to cited text no. 2
[PUBMED]    
3.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. 3
[PUBMED]    
4.Ellis GL, Auclair PL. Atlas of tumour pathology. In: Tumours of the Salivary Glands. 3 rd series, fascicle 17. Washington: Armed Forces Institute of Pathology; 1996. p. 289-96.  Back to cited text no. 4
    
5.Rajendran R, Sivapathasundharam B. Tumors of the Salivary glands. In: Shafer's Textbook of Oral Pathology. 6 th ed., Ch. 3. India: Elsevier Pvt. Ltd.; 2009. p. 230.  Back to cited text no. 5
    
6.Alexis JB, Dembrow V. Papillary cystadenoma of a minor salivary gland. J Oral Maxillofac Surg 1995;53:70-2.  Back to cited text no. 6
[PUBMED]    
7.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. 7
[PUBMED]    
8.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. 8
    
9.Chaudhry AP, Vickers RA, Gorlin RJ. Intraoral minor salivary gland tumors. An analysis of 1,414 cases. Oral Surg Oral Med Oral Pathol 1961;14:1194-226.  Back to cited text no. 9
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10.Kerpel SM, Freedman PD, Lumerman H. The papillary cystadenoma of minor salivary gland origin. Oral Surg Oral Med Oral Pathol 1978;46:820-6.  Back to cited text no. 10
[PUBMED]    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


This article has been cited by
1 A Rare Case Report of Mucinous Cystadenoma with Immunohistochemical Analysis and Review of Literature
Charu Girotra,Mukul N. Padhye,Pratibha Mahajan,Aishwarya Nair
Journal of Maxillofacial and Oral Surgery. 2014;
[Pubmed] | [DOI]



 

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