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CASE REPORT  
Year : 2013  |  Volume : 56  |  Issue : 2  |  Page : 163-165
Salivary duct cyst in the upper lip: Case report and review of the literature


1 Department of Clinical Dentistry, School of Dentistry, Walter Cantídio University Hospital, Federal University of Ceará, Fortaleza, Brazil
2 Division of Oral and Maxillofacial Surgery, Walter Cantídio University Hospital, Federal University of Ceará, Fortaleza, Brazil
3 Division of Oral Pathology, School of Dentistry, Federal University of Ceará, Campus Sobral, Sobral, Brazil
4 Division of Oral Radiology and Stomatology, School of Dentistry, Federal University of Ceará, Campus Sobral, Sobral, Brazil

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Date of Web Publication23-Sep-2013
 

   Abstract 

Salivary duct cyst (SDC) in an uncommon lesion of the minor salivary glands, with etiology related to obstruction of the salivary duct. It presents with a color similar to that of the mucosa, is mobile, measures between 3 and 10 mm and affects preferentially the bottom lip, floor of the mouth and the jugal mucosa. SDC have a predilection for the male and individual in elderly groups. The pathogenesis of SDC is associated with the formation of a mucous plug that causes partial or total obstruction of the salivary gland system duct, resulting in the dilation of the duct and increase in intraluminal pressure. They present clinical-pathological characteristics similar to those of salivary gland tumors, making diagnosis difficult and subject to errors in treatment. It is important for the dentist to include SDC in the differential diagnosis of lesions that affect the upper lip, although it is relatively rarely found in this anatomic site.

Keywords: Minor salivary gland, oral lesions, retention mucocele cyst, salivary duct cyst, sialocyst

How to cite this article:
Chaves FN, Carvalho FR, Pereira KA, Costa FG. Salivary duct cyst in the upper lip: Case report and review of the literature. Indian J Pathol Microbiol 2013;56:163-5

How to cite this URL:
Chaves FN, Carvalho FR, Pereira KA, Costa FG. Salivary duct cyst in the upper lip: Case report and review of the literature. Indian J Pathol Microbiol [serial online] 2013 [cited 2020 Feb 26];56:163-5. Available from: http://www.ijpmonline.org/text.asp?2013/56/2/163/118678



   Introduction Top


Cystic lesions represent around 6-9% of the total salivary gland disorders, particularly in the major salivary glands. [1] There are two types of cysts observed: The extravasation cyst (without epithelial lining) and the retention cyst (with epithelial lining). [2],[3],[4]

Various terminologies may be used to denominate these lesions; however, "salivary duct cyst (SDC)" is the most adequate by virtue of its origin being related to the epithelial lining of salivary gland ducts. [5] Clinically, the salivary gland duct present coloring similar to that of the mucosa, is mobile and is 3-10 mm in size. It affects different sites in the oral mucosa, preferably the bottom lip and rarely the upper lip. [3]

The aim of the present study was to relate a rare case of SDC in a minor salivary duct of a 78-year-old patient.


   Case Report Top


Patient, a 78-year-old man, presented to the stomatology service of the Federal University of Ceará - Sobral Campus, Brazil, for a dental consultation. On intraoral examination, a nodule was observed on the upper lip, with coloring similar to that of the mucosa, of a fibrous consistency, with a smooth surface, sessile implantation, with a size of approximately 2 cm 1 cm, painless and an undetermined time of development [Figure 1]. After the clinical evaluation, an excisional biopsy was performed [Figure 2], followed by histopathological evaluation. Microscopically, a dilated salivary gland duct was shown, containing eosinophilic material, compatible with mucin [Figure 3], an epithelial lining with two to three layers of cuboid cells and acini of salivary gland mucosa exhibiting discrete atrophy and congested blood vessels completed the histological condition examined [Figure 4], leading to the histopathological report of retention mucocele cyst. At present, patient has been followed-up for 1 year and there have been no signs of recurrence.
Figure 1: Aspect of lesion. Painless nodule on the upper lip, sessile, with coloring similar to that of the mucosa, of a fibrous consistency, with a smooth surface, with a size of approximately 2 cm × 1 cm

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Figure 2: Excisional biopsy. Removal of the lesion with the affected salivary gland duct, final aspect (a) and specimen (b)

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Figure 3: Photomicrograph H and E, ×40 — cystic cavity represented by the epithelial lining surround the amorphous eosinophilic material, compatible with mucin

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Figure 4: Photomicrograph H and E, ×400 — epithelial lining presents 2-3 layers of cuboid cells and discrete atrophy of the mucous acini of the affected salivary gland

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   Discussion Top


In the present study, a review of the literature was performed in association with the clinical case description. A total of 478 cases have been described from 1956 up to 2011. In the selected articles, it was observed that involvement of the oral mucosa occurred mainly in the age-range between the 1 st and 3 rd decades of life, distributed in a balanced manner and as from the 7 th decade of life. As regards gender, a discrete predilection for the male sex was observed, similar to the present case. With respect to location, the main sites were the bottom lip, floor of the mouth and jugal mucosa. Other sites affected were described as being the palate, tongue and retromolar region. [1],[3],[5] In the reported case, the site where the lesion was located was the upper lip, a rare location, according to the literature analyzed.

The pathogenesis of the salivary gland cyst is still uncertain. It is believed that with the increase in age, there is a natural reduction in salivary secretion, promoting the formation of a mucous plug that causes partial or total obstruction of the salivary gland system duct, resulting in dilation of the duct and increase in intraluminal pressure. [3],[5],[6]

Microscopically, the SDC is shown to have an epithelium lining of flattened duct cells similar to intercalated duct cells or bilayered duct cells similar to striated and excretory ductal cells. In some areas, plump papillary projections are developed. Occasionally, goblet cells and non-keratinized squamous epithelium may be present within the epithelial lining of the cyst. [1] A secretory material including crystalloids and focal papillary projections fills the lumen or epithelial transformation could be observed with squamous metaplasia or goblet-like cells. [7] In a review of 121 cases of SDCs, Eversole observed three histological subtypes: "True mucous retention cysts" lined by nononcocytic ductal epithelium and exhibiting minimal inflammation; "reactive oncocytoid cysts" exhibiting oncocytoid metaplasia and "mucopapillary cysts" with a papillary multicystic growth pattern and regions of pronounced mucous metaplasia. [5],[8]

SDCs may present clinical-pathological characteristics similar to those of salivary gland tumors, making diagnosis difficult and subject to errors in treatment. [9] Basal cell adenoma, pleomorphic adenoma, cystoadenoma and well-differentiated mucoepidermoid carcinoma or with a low degree of malignancy are oral lesions that may present in a similar manner to that of SDCs. [1],[7],[9]

Basal cell adenoma is an uncommon benign neoplasia of the salivary glands, preferentially located in the upper lip, normally firm to palpation and preferentially affects elderly patients, over 50 years of age. In the case described, the clinical characteristics found were similar to these. However, the trans-surgical macroscopic aspect of "bunches of grapes" observed in basal cell adenomas, was not observed in the present case. The pleomorphic adenoma is considered the most common salivary neoplasia of the oral cavity. However, its main site of location is the palate and it preferentially affects an age-range between 30 and 50 years, differing from the case under study, whose site of location was the upper lip in a 78-year-old patient. [10]

Some non-neoplastic lesions in salivary glands, such as mucoceles may represent a diagnostic challenge, by virtue of the clinical presentation similar to that of SDC. [6] Nevertheless, mucocele differs histologically from this lesion due to the absence of a ductal lining of epithelium. [5]

Cryosurgery, carbon dioxide laser surgery and conservative surgical excision are the treatment modalities that may be used. [1],[6],[9] By virtue of the fact that benign tumors of the salivary glands are able to clinically mimic SDCs, complete excision is mandatory.

Although the SDC is considered a rare condition in the oral cavity, it is important to include this lesion in the differential diagnosis of lesions that affect the upper lip.


   Acknowledgment Top


The authors would like to express your deep gratitude to Professor Roberta Barroso Cavalcante (DDS, MSc, PhD. Adjunct Professor, Division of Oral Pathology, University of Fortaleza, Fortaleza, Ceará, Brasil) for his example of dedication and study, enthusiastic encouragement and for all useful critiques for this work.

 
   References Top

1.Antoniades D, Epivatianos A, Markopoulos A, Kolokotronis A, Zaraboukas T. Coexistence of mucous retention cyst and basal cell adenoma arising from the lining epithelium of the cyst. Report of two cases. Med Princ Pract 2009;18:248-52.  Back to cited text no. 1
    
2.Martins-Filho PR, Santos Tde S, da Silva HF, Piva MR, Andrade ES, da Silva LC. A clinicopathologic review of 138 cases of mucoceles in a pediatric population. Quintessence Int 2011;42:679-85.  Back to cited text no. 2
    
3.Granholm C, Olsson Bergland K, Walhjalt H, Magnusson B. Oral mucoceles; extravasation cysts and retention cysts. A study of 298 cases. Swed Dent J 2009;33:125-30.  Back to cited text no. 3
[PUBMED]    
4.Cohen L. Mucoceles of the oral cavity. Oral Surg Oral Med Oral Pathol 1965;19:365-72.  Back to cited text no. 4
[PUBMED]    
5.Chi AC, Lambert PR 3 rd , Richardson MS, Neville BW. Oral mucoceles: A clinicopathologic review of 1,824 cases, including unusual variants. J Oral Maxillofac Surg 2011;69:1086-93.  Back to cited text no. 5
    
6.Hayashida AM, Zerbinatti DC, Balducci I, Cabral LA, Almeida JD. Mucus extravasation and retention phenomena: A 24-year study. BMC Oral Health 2010;10:15.  Back to cited text no. 6
[PUBMED]    
7.Seifert G. Mucoepidermoid carcinoma in a salivary duct cyst of the parotid gland. Contribution to the development of tumours in salivary gland cysts. Pathol Res Pract 1996;192:1211-7.  Back to cited text no. 7
[PUBMED]    
8.Eversole LR. Oral sialocysts. Arch Otolaryngol Head Neck Surg 1987;113:51-6.  Back to cited text no. 8
[PUBMED]    
9.Everson JW, Speight PM. Non-neoplastic lesions of the salivar glands: New entities and diagnostic problems. Curr Diagn Pathol 2006;12:22-30.  Back to cited text no. 9
    
10.Osterne RL, Costa FW, Mota MR, Vidal Patrocínio RM, Alves AP, Soares EC, et al. Lip lesions in a Brazilian population. J Craniofac Surg 2011;22:2421-5.  Back to cited text no. 10
    

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Correspondence Address:
Filipe Nobre Chaves
Ministro Joaquim Bastos, 471, Apt. 1502, Fátima, Fortaleza, Ceará, CEP: 60415-040
Brazil
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.118678

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]

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    Abstract
   Introduction
   Case Report
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