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Year : 2019  |  Volume : 14  |  Issue : 2  |  Page : 113-114

Keratinizing variant of pleomorphic adenoma


Department of Oral and Maxillofacial Pathology and Microbiology, D. Y. Patil University School of Dentistry, Navi Mumbai, Maharashtra, India

Date of Submission05-Feb-2018
Date of Decision18-Nov-2018
Date of Acceptance19-Aug-2019
Date of Web Publication25-Nov-2019

Correspondence Address:
Dr. Treville Pereira
Department of Oral and Maxillofacial Pathology and Microbiology, D. Y. Patil University School of Dentistry, Sector 7, Nerul, Navi Mumbai - 400 706, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdmimsu.jdmimsu_9_18

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How to cite this article:
Pereira T, Shetty S, Gotmare S, Kale S. Keratinizing variant of pleomorphic adenoma. J Datta Meghe Inst Med Sci Univ 2019;14:113-4

How to cite this URL:
Pereira T, Shetty S, Gotmare S, Kale S. Keratinizing variant of pleomorphic adenoma. J Datta Meghe Inst Med Sci Univ [serial online] 2019 [cited 2019 Dec 11];14:113-4. Available from: http://www.journaldmims.com/text.asp?2019/14/2/113/271557



Pleomorphic adenoma (PA) accounts for 73% of all salivary gland tumors, with parotid gland being the most commonly affected. It usually affects individuals in the fifth or sixth decades of life, and about 60% of them are women.[1] The neoplasm is usually slow-growing, painless, and firm on palpation. Possible complications to treatment include the risk of recurrence and malignant transformation.[2]

A 29-year-old male patient reported to our clinic with a complaint of pain and swelling in the left submandibular region for 2 years. There was a history of fever, with sudden weight loss over the past 6 months. The swelling was insidious in onset and had progressed to its present size of approximately 4 cm × 3 cm [Figure 1]a. It had irregular borders and was firm in consistency. It was initially small in size and had been growing slowly for the past 2 years to reach its present size. The slow painless progression and the poor socioeconomic status of the patient had discouraged the patient from seeking treatment. There was neither sensory nor motor deficiency nor any alteration in the general physical examination.
Figure 1: (a) Extraoral photograph of the patient showing swelling with irregular borders in the left submandibular region. (b) Magnetic resonance imaging showing a heterogeneous isotense lesion displacing the left submandibular gland

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Ultrasonography revealed a well-lobulated heterogeneously hypoechoic lesion within the left submandibular gland measuring 3.8 cm × 3.4 cm × 3.5 cm in size suggestive of neoplastic etiology. In addition, multiple enlarged nonnecrotic lymph nodes were noted bilaterally in the cervical region with the largest measuring 1.9 cm × 1.1 cm at right level IB/II superiorly. No obvious bony erosion was noted. A magnetic resonance imaging revealed a 4.8 cm × 4 cm × 3.3 cm heterogeneous isointense lesion with hyperintense cystic areas within [Figure 1]b. The isointense solid component showed heterogeneous postcontrast enhancement. The lesion was displacing the left submandibular gland giving an inference of a well-defined heterogeneously enhancing solid cystic lesion in the left submandibular region which was consistent with benign neoplastic etiology. Fine-needle aspiration cytology revealed the presence of cheesy material which was suggestive of an epidermoid cyst with a differential diagnosis of tuberculous lymphadenitis. Excision of the tumor was performed under general anesthesia along with the removal of the submandibular gland. On histopathological examination, a biphasic population of epithelial and mesenchymal cells was observed. The epithelial component being glandular in nature showed variable sized ducts lined by cuboidal epithelium with underlying myoepithelial cells. Stroma showed fibromyxoid areas along with focal areas of squamous metaplasia and the formation of keratin pearls. Histopathological features were suggestive of a PA with squamous metaplasia and keratin pearl formation [Figure 2]a. Postoperative course was uneventful.
Figure 2: (a) Tumor islands arranged in sheets and ducts, along with fibromyxoid areas (H and E, ×10), (b) Keratin pearl formation (Mallory, ×10)

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Squamous metaplasia may be observed in nonneoplastic lesions such as lymphoepithelial cysts, chronic sialadenitis, and sialometaplasia. A tumor which shows extensive squamous metaplasia can sometimes be misdiagnosed with squamous cell carcinoma. Varied stromal changes such as chondroid, osseous, myxoid, or mucous may be seen.[3],[4],[5] Cystic changes may be due to secretions from the ducts of salivary gland, hemorrhage, or necrosis. In the present case, there was the presence of keratin along with squamous metaplasia, which was confirmed using Mallory stain [Figure 2]b.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to b'e reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Frazell EL. Clinical aspects of tumors of the major salivary glands. Cancer 1954;7:637-59.  Back to cited text no. 1
    
2.
Reiland MD, Koutlas IG, Gopalakrishnan R, Pearson AG, Basi DL. Metastasizing pleomorphic adenoma presents intraorally: A case report and review of the literature. J Oral Maxillofac Surg 2012;70:e531-40.  Back to cited text no. 2
    
3.
Batrani M, Kaushal M, Sen AK, Yadav R, Chaturvedi NK. Pleomorphic adenoma with squamous and appendageal metaplasia mimicking mucoepidermoid carcinoma on cytology. Cytojournal 2008;6:5.  Back to cited text no. 3
    
4.
Su CC, Chou CW, Yiu CY. Neck mass with marked squamous metaplasia: A diagnostic pitfall in aspiration cytology. J Oral Pathol Med 2008;37:56-8.  Back to cited text no. 4
    
5.
Siddaraju N, Murugan P, Basu D, Verma SK. Preoperative cytodiagnosis of cystic pleomorphic adenoma with squamous metaplasia and cholesterol crystals: A case report. Acta Cytol 2009;53:101-4.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2]



 

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