Dermatology Online Journal

Papillary cystadenoma of the minor salivary gland of the lower lip
Daniel Araki Ribeiro¹, Maria Renata Sales Nogueira Costa², and Gerson Francisco de Assis³
Dermatology Online Journal 10 (1): 14

1. Núcleo de Avaliação Toxicogenética e Cancerígena - TOXICAN– Departamento de Patologia – Faculdade de Medicina de Botucatu – UNESP 2. Departamento de Estomatologia – Disciplina de Patologia – Faculdade de Odontologia de Bauru – USP 3. Departamento de Ciências Biológicas – Disciplina de Histologia – Faculdade de Odontologia de Bauru – USP. ak92@hotmail.com

---

Abstract

A case report of the papillary cystadenoma from minor salivary gland in lower lip of a 54-year-old man is described.

---

Introduction

The papillary cystadenoma (PC) is defined as a cystic adenoma in which the cystic space is filled with papillary projections [1], arising from undifferentiated epithelium of the intercalated ducts of the glands[3] that include the minor salivary glands, the parotid glands, larynx, nasopharynx and the lachrymal gland [2]. The most frequent locations for this tumor in the oral cavity are: hard palate, cheek, and posterior region of the tongue [2]. This lesion has a tendency to recur if not appropriately excised [3]. There are case reports of papillary cystadenoma of the oral region [1, 3, 4, 5, 6, 7, 8], however, PC arising from a minor salivary gland is rare [9, 10].

Case Report

A 54-year-old man was seen in July 2000 with an asymptomatic raised, smooth lesion of the lower oral mucosa. The lesion was well marginated and had an erythematous base. The appearance was similar to a mucocele, the working clinical diagnosis. It was excised under anesthesia and the biopsy fragment was submitted in 10 percent buffered formalin. Multiple 5-mm sections of were prepared and stained with hematoxilin-eosin (H&E).

Figure 1	Figure 2
Clinical appearance of PC in lower lip (Fig. 1). PC showing minor mucous salivary gland, fibrous connective tissue, and cystic epithelium. (H&E original magnification × 25)

Anatomopathologic findings

The macroscopic examination revealed fragments of smooth tissue, firm consistence, irregular surface, chestnut color with white areas, measuring, all the fragments, 0.8 × 0.6 × 0.4 cm. The specimen was submitted in 10 percent buffered formalin and multiple 5-mm sections were prepared and stained with H&E. The microscopic examination showed a minor mucous salivary gland beneath of fibrous connective tissue throughout peripheral region, papillary projections into the lumen and disorganized, columnar-pseudostratified epithelium (Fig. 2). There was a mucoid material within the cystic cavity, with few areas of epithelial metaplasia and intraductal papillary projections (Fig. 3). Muscle tissue was abundant outside the fibrous connective tissue. The cystic-epithelial stratum consisted of cells containing small amounts of mucinous material, characteristic of oncocytic cells (Fig 4). The microscopic diagnosis was PC.

Figure 3	Figure 4
PC displaying cystic cavity with mucous material, intraductal papillary projections, and an underlying minor mucous salivary gland (Fig 3. H&E original magnification × 10). Cystic epithelium in high magnification showing oncocytic cells (Fig. 4, H&E original magnification × 100).

Discussion

The World Health Organization (WHO) [11] described PC as "a tumor that closely resembles Warthin tumor but without the lymphoid elements, constituting multiple papillary projections and a greater variety of epithelial lining cells." In this sense, we believed that salivary glands tumors are difficult to diagnose or interpret because there are many possible patterns of presentation. In addition, PC of the minor salivary gland is rare [9, 10]. Moreover, many of the case reports of PC are old [2, 4, 5, 12]. Hopefully this report will serve as an update and provide new information about PC of the minor salivary gland.

A literature review of the clinical, histologic, and biologic features of PC shows no consensus regarding either age of onset or gender preference [1, 2, 3, 4, 5, 9]. In our case, the tumor occurred in lower oral mucosa of a 54-year-old man, whose tumor size was for about 0.20 cm³ and the therapy chosen was surgical excision [7]. Our patient has been followed for 2 years and has not had a recurrence.

On microscopic examination, PC has presents circumscribed fibrous tissue [1, 3, 6], multiple papillary projections into their lumina, and lining cells of cuboidal to columnar epithelium [3]. Oncocytic cells were present with no cellular atypia and no mitotic figures [6]. This cellular oncocytosis appears to be age related and is accelerated by smoking. Our patient is a smoker and we advised him to stop using any tobacco products after PC excision.

Based on both macroscopic and microscopic findings in this case report, we assume that PC is a benign neoplastic tumor.

References

1. Kameyama Y, Okada Y, Takehana S, Mizohata M, Nishio S, Enomoto M: Papillary cystadenoma, Int J Oral Surg. 14:556-9,1985.

2. Kerpel SM, Fredman PD, Lumerman H, The papillary cystadenoma of minor salivary gland origin. Oral Surg. 46:820-6,1978.

3. Alexis J, Dewbrow V, Papillary cystadenoma of a minor salivary gland. J Oral Maxillofac Surg. 53:70-2,1995.

4. Akin RK, Kreller AJ, Walters PJ, Papillary cystadenoma of the lower lip -report of a case. J Oral Surg. 31:858-60,1973.

5. Cathoun NR, Cerine FC, Mathews MJ, Papillary cystadenoma of the upper lip. Oral Surg. 21:782-5,1966.

6. Guccion JG, Redman RS, Calhoun NR, Saini N, Papillary cystadenoma of the palate - a case report and ultrastructural study. J Oral Maxillofac. Surg. 55:59-64,1997.

7. Waldron CA, El-Mofty SK, Gnepp DR, Tumors of the intraoral minor salivary glands - a demographic and histologic study of 426 cases. Oral Surg. 66:323,1988.

8. Whittaker JS, Turner EP, Papillary tumours of the minor salivary glands: J Clin Pathol. 29:795-805,1976.

9. Tsurumi K, Kamiya H, Yokoi M, Kameyama Y, Papillary oncocytic cystadenoma of palatal minor salivary gland: a case report. J Oral Maxillofac Surg. 61:631-3,2003.

10. Mahler V, Schell H, Papillary cystadenoma- a rare tumor of the minor salivary glands. Eur J Dermatol. 5:387-9,1999.

11. Seifert G, World Health Organization. Histological typing of salivary gland tumours. Springer-Verlag, 1991:16-7.

12. Parnes EJ, Pappilary cystadenoma. Oral Surg. 21:783-5,1966.

© 2004 Dermatology Online Journal