Pleomorphic adenoma of the cheek: A case report arising from accessory parotid gland around Stensen's duct orifice

Pleomorphic adenoma of the cheek: A case report arising from accessory parotid gland around Stensen's duct orifice

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ARTICLE IN PRESS Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2016) xxx–xxx

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Case Report

Pleomorphic adenoma of the cheek: A case report arising from accessory parotid gland around Stensen’s duct orifice Wang Dian-can a , Chen Yan b , Guo Chuan-bin a,∗ a Department of Oral and Maxillofacial Surgery, Peking University School and Hospital of Stomatology, 22 South Zhongguancun Avenue, Haidian District, Beijing 100081, PR China b Department of Oral Pathology, Peking University School and Hospital of Stomatology, 22 South Zhongguancun Avenue, Haidian District, Beijing 100081, PR China

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Article history: Received 1 December 2015 Received in revised form 4 February 2016 Accepted 19 February 2016 Available online xxx Keywords: Pleomorphic adenoma Stensen’s duct orifice Tadpole’s sign Operation Case report

a b s t r a c t Pleomorphic adenoma is the most common benign salivary tumor, originating from various areas besides major or minor salivary glands. We present a case of a patient with buccal mass. Tadpole’s sign in preoperative axial CT scan indicated the relation of tumor and Stensen’s duct orifice. Pathologic report was pleomorphic adenoma. To our knowledge, this is the first reported pleomorphic adenoma of Stensen’s duct orifice. Resection of the tumor with reconstruction of the orifice is the preferable treatment. © 2016 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.夽

1. Introduction Pleomorphic adenoma (mixed tumor) is the most common benign salivary gland neoplasm that affects both major and minor salivary glands, characterized by neoplastic proliferation of parenchymal glandular cells along with myoepithelial components. It can also occur, albeit most uncommonly, in lacrimal gland [1,2], external ear canal [3], TMJ area [4], sinonasal and skull base [5], epiglottis [6], larynx and nasopharynx [7], trachea [8], esophagus [9], lung [10], breast [11] and so on. Intraosseous pleomorphic adenoma [12], such as in anterior mandible [13] was also reported. We present a rare case of pleomorphic adenoma of Stensen’s duct orifice. 2. Case report A 38-year-old female was admitted to our department with a painless intraoral lump in her left cheek, which she had first noticed

夽 Asian AOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathology; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants. ∗ Corresponding author. Tel.: +86 10 82195295; fax: +86 10 62173402. E-mail address: [email protected] (G. Chuan-bin).

a year back. The lump did not enlarge since it was noticed and it did not bleed. Her parotid gland never became enlarged upon trying to eat. There was no history of trauma. She did not have fever. Physical examination revealed a 1.5 cm × 1.0 cm, painless, freely movable intraoral mass in the left cheek, inferior to the Stensen’s duct opening. Axial enhanced computerized tomography (CT) of the cheek area demonstrated the outline of a mass at the anterior border of the masseter muscle, without any radiological signs of malignancy. The mass was attached with the Stensen’s duct, which can be named as tadpole sign because the CT appearance of the way in which the mass attached with the Stensen’s duct is like a tadpole (Fig. 1). Under general anesthesia, a mucosal incision was given over the mass surface, 1 cm away from the Stensen’s duct orifice. After blunt and sharp dissection alternately, an encapsulated mass was exposed, which was attached to the Stensen’s duct end. The mass was excised completely with suitable margin of normal tissue (including the Stensen’s duct end). The Stensen’s duct opening was reconstructed with the endpoint of the duct sutured to the mucosal incision (Fig. 2). The surgical specimen was a yellowish, irregular lobulated mass and elastic in consistency, measuring 2 cm in diameter (Fig. 3). The cut surface revealed a central cystic space containing yellowish sticky fluid. Microscopic examination showed that the duct shares the capsule with the tumor (Fig. 4) and there were no salivary glands around the tumor, which confirmed that the tumor originated from

http://dx.doi.org/10.1016/j.ajoms.2016.02.007 2212-5558/© 2016 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.夽

Please cite this article in press as: Dian-can W, et al. Pleomorphic adenoma of the cheek: A case report arising from accessory parotid gland around Stensen’s duct orifice. J Oral Maxillofac Surg Med Pathol (2016), http://dx.doi.org/10.1016/j.ajoms.2016.02.007

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Fig. 3. The surgical specimen, measuring about 2 cm in diameter.

Fig. 1. Enhanced axial CT shows tadpole’s sign. A mass at the anterior border of the left masseter muscle, without any radiological signs of malignancy (the white arrow). The mass was attached to the Stensen’s duct (the hollow arrow), like a tadpole.

Fig. 4. The duct (the arrow) and the tumor share the same capsule, indicating that the tumor originated from the duct, 3×, H&E.

Fig. 2. The mass (the white arrow) was attached to Stensen’s duct end (the hollow arrow).

the duct. High magnification microscope revealed that it is a typical pleomorphic adenoma. The main components were cellular region and duct-like structures (Fig. 5). Other characteristic region specific to pleomorphic adenoma such as myxoid stroma was found too. The patient was discharged 3 days after the operation. After 6 months postoperatively, recurrent and mild swelling of the parotid secondary to salivary retention was noted and it gradually reduced. The reconstructed Stensen’s duct functioned well after 20 months follow-up. 3. Discussion Among tumors arising from Stensen’s duct, malignancies were most reported [14]. To our knowledge, this is the first reported

pleomorphic adenoma of Stensen’s duct orifice. Because buccal mucosa is the second most common affected site by intraoral minor salivary gland tumor, following palate [15–17], the initial provisional clinical diagnosis of this case was that of a minor salivary gland tumor originating from the buccal mucosa. But during the operation, we failed to separate the duct and the tumor through blunt dissection and the tumor could hardly be removed before the end of the Stensen’s duct was sacrificed. Thus the postoperative diagnosis of pleomorphic adenoma that originated from the Stensen’s duct orifice was established and was confirmed by the observation under optical microscope. This was different from those pleomorphic adenoma of buccal minor salivary gland [18]. Since the duct itself cannot be a source of pleomorphic adenoma, the very rare pleomorphic adenoma of Stensen’s duct orifice may be viewed as a tumor that originated from ectopic salivary gland tissue or a special accessory parotid gland tumor. Ectopic salivary gland tissue has been identified at various sites, including skin, neck, hypophysis, mediastinum, prostate, rectum, vulva, thyroid gland, mastoid bone, middle ear, maxilla, mandible

Please cite this article in press as: Dian-can W, et al. Pleomorphic adenoma of the cheek: A case report arising from accessory parotid gland around Stensen’s duct orifice. J Oral Maxillofac Surg Med Pathol (2016), http://dx.doi.org/10.1016/j.ajoms.2016.02.007

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indicator of duct orifice’s origin. Total resection of the tumor and reconstruction of the orifice with the residual duct is the preferable treatment. Conflicts of interest No conflicts of interest to declare. Funding None. Ethical approval Not required. Patient permission Not required. Fig. 5. Microscopic view is that of a typical pleomorphic adenoma, 40×, H&E.

References and others [13]. From embryological point of view, there are possibilities that the area along the parotid duct could harbor ectopic salivary gland tissue. In the sixth week of prenatal development, the parotid salivary glands appear as epithelial buds, located on the oral part of the cheek, next to the labial commissures of the primitive mouth. These glandular epithelial buds later grow posteriorly toward the otic placodes of the ears and branch to form solid cords with rounded terminal ends near the developing facial nerve. These cords are canalized and form ducts, with the largest becoming the parotid duct for the parotid gland. The rounded terminal ends of the cords form the acini of the glands and the supporting connective tissue of the gland develops from the surrounding mesenchyme. During the development of the gland, it is possible that some acini might have stayed near the duct and finally become original tissue to develop a pleomorphic adenoma. Ectopic salivary gland tissue along the Stensen’s duct is named as accessory salivary gland tissue. Normally, accessory parotid gland is found along Stensen’s duct along its middle third [19]. Total resection of freely movable, asymptomatic, small tumors in buccal area near to orifice of Stensen’s duct is preferable treatment. It needs to be preoperatively differiated from those originating from the minor salivary gland, buccal mucosa or the duct. More importantly, it should be assessed whether the duct is affected or not and whether the tumor is arising from duct. By computed tomography (CT), or magnetic resonance imaging (MRI), or ultrasonography, the basis of duct related findings, such as swelling of duct near the tumor or concurrent parotitis is suspicion of the Stensen’s duct tumor. In this case, we found that an interesting tadpole sign in axial CT scan or MRI, describing the relationship between duct orifice and tumor, may help clinicians achieve decisive conclusion and make an more appropriate treatment plan, mainly related to scarification or preservation of duct. 4. Conclusion Pleomorphic adenoma may originate at Stensen’s duct orifice. In axial CT scan or MRI, tadpole sign may be decisive and characteristic

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Please cite this article in press as: Dian-can W, et al. Pleomorphic adenoma of the cheek: A case report arising from accessory parotid gland around Stensen’s duct orifice. J Oral Maxillofac Surg Med Pathol (2016), http://dx.doi.org/10.1016/j.ajoms.2016.02.007