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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 10  |  Issue : 1  |  Page : 60-63

Pleomorphic adenoma of tongue: A common entity at unusual location


Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India

Date of Submission01-Jul-2019
Date of Acceptance23-Jan-2020
Date of Web Publication16-Jul-2020

Correspondence Address:
Dr. Shruti Sangwan
Room No 416, Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, College Building, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohs.ijohs_32_19

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  Abstract 


Salivary gland tumors constitute 3% of salivary glands lesions. The most common tumor of salivary glands is pleomorphic adenoma (PA) commonly affecting major salivary glands. PA involving minor salivary glands accounts for <3% of all head and neck tumors. Palate is the most common site, followed by upper lips. The tongue is an unusual site and there are only 29 reported cases in literature as per our knowledge. Herein, we report a rare case of 63-year-old male presenting with non-healing ulcer on the left lateral border of the tongue. A biopsy was done and material sent for histopathological examination. Histopathological examination of biopsy revealed PA. Smooth muscle actin and p63 immunohistochemistry showed myoepithelial cells. This case suggests that PA of the tongue should be considered in the differential diagnosis of lesions of the tongue.

Keywords: Benign tumor, minor salivary gland, oral cavity, palate, tongue


How to cite this article:
Sangwan S, Sufian, Kaur N. Pleomorphic adenoma of tongue: A common entity at unusual location. Int J Oral Health Sci 2020;10:60-3

How to cite this URL:
Sangwan S, Sufian, Kaur N. Pleomorphic adenoma of tongue: A common entity at unusual location. Int J Oral Health Sci [serial online] 2020 [cited 2020 Aug 8];10:60-3. Available from: http://www.ijohsjournal.org/text.asp?2020/10/1/60/289875




  Introduction Top


Pleomorphic adenoma (PA) is a benign neoplasm affecting the salivary glands. They are mainly seen in major salivary glands, especially in the parotid glands. Minor salivary glands are more commonly affected by malignant neoplasms than benign ones. Benign and malignant tumors of the minor salivary glands are usually found on the palate, upper lip, gums, cheek, floor of the mouth, pharynx, larynx, and trachea.[1] Our case is rare of its type as it was present in an unusual site, i.e., the tongue.

The incidence of salivary gland neoplasms in minor glands varies from 9% to 22%. Approximately 8% of PA s involve the minor salivary glands. The involvement of the base of the tongue is rare. Malignant tumors such as adenocarcinoma, adenoid cystic carcinoma, and mucoepidermoid carcinoma involve the tongue more frequently.[2],[3],[4]

In the tongue, the most common benign tumor is the PA, whereas adenoid cystic carcinoma is the most common malignant tumor reported. Most of the studies indicate an overall ratio of approximately 1:6 for the benign/malignant lingual salivary gland tumors.[3] The high recurrence rate and frequent malignant conversion are its characteristics features. For most cases, the only clinical sign is an asymptomatic painless swelling with a slow growth rate. Microscopically, PA is characterized by variable, diverse structuring histological patterns consisting of fibrous, hyaline, myxoid, cartilaginous, and osseous areas, which are differentiated by myoepithelial cells. The occurrence of PA of the tongue is very rare, and till date, very few cases have been reported in the literature.[2],[4]

When they occur on the tongue, PAs are most commonly seen in the posterior, followed by anterior, and rarely in the lateral lingual gland (Ebner's gland) of the tongue.[5] Malignant tumors such as adenocarcinoma, adenoid cystic carcinoma, and mucoepidermoid carcinoma involve the tongue more frequently than their benign counterparts.[2],[3],[4] We hereby report a rare case of PA of the tongue in a 63-year-old male patient.


  Case Report Top


A 63-year-old Male patient presented to our outpatient department with a history of nonhealing ulcer in the tongue for the past 6 months. It was slow growing and not associated with difficulty in swallowing or movement of the tongue. The patient does not report any incidence of trauma to the area and is not a known diabetic or hypertensive. There was nothing suggestive in the personal or family history. After getting informed consent, the patient was examined, and there was a 1 cm by 0.5 cm ulcer on the left lateral border of the tongue that was firm, nontender with no restriction of the tongue mobility. No other abnormality was detected in the oral cavity. No cervical lymphadenopathy was seen. Rest of the ear, nose, throat examination was within normal limits. Magnetic resonance imaging (MRI) showed homogeneous mass of size 2.5 cm × 2 cm × 1 cm on the lateral border of the tongue, in the left side, not crossing midline. The muscles of the tongue were not seen to be infiltrated and features were suggestive of benign lesion. Biopsy was done under local anesthesia and material sent for histopathological examination. The postoperative period was uneventful. Grossly, we received a single gray-brown soft-tissue piece measuring 0.5 cm × 0.3 cm × 0.2 cm. Microscopic examination showed stratified squamous epithelium lined tissue. Underneath seen epithelial ductal elements embedded in chondromyxoid stroma consistent with the diagnosis of PA along with few normal minor salivary glands [Figure 1] and [Figure 2]. Smooth muscle actin and p63 immunohistochemistry were performed for myoepithelial cells. Both the markers show positivity in myoepithelial cells in PA as well as in minor salivary glands [Figure 3] and [Figure 4].
Figure 1: Epithelial elements in chondromyxoid stroma, H and E, ×100

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Figure 2: Epithelial elements in chondromyxoid stroma, H and E, ×400

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Figure 3: Smooth muscle actin positivity in myoepithelial cells, ×400

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Figure 4: p63 positivity in myoepithelial cells, ×400

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  Discussion Top


Misssen in 1874 first described PA. PA s arise in the parotid gland (80%) and then in the submandibular gland (10%) and the minor salivary glands (10%).[6] Palate, upper lip, gums, cheek, and floor of the mouth, pharynx, and trachea are the common sites of tumors of minor salivary glands.[1]

The tongue is a rare site. They have female preponderance and usually occur in 40–50 years of age group.[7],[8] Of 143 intraoral PA s studied by Eveson and Cawson, only 1 case involved tongue. As per article published by Tanigaki et al., there are only 26 cases reported in the world since 1960 as PA in the tongue[5] and 3 more cases have been reported after that.

The etiology is not known clearly. There are evidence to suggest that exposure to radiation and viruses (simian virus) have a role to play. Abnormalities in the chromosome loci 8q12 and 12q15 are also implicated. These tumors usually present as smooth masses that are slow growing and painless.[9] The symptoms depend on site and size may be asymptomatic or can cause dysphagia if large and located posteriorly in the oral cavity, respiratory difficulty, etc.

Histologically, PA s are mixed tumors, i.e., they have both epithelial and mesenchymal elements. Their proportion can vary, depending on which the consistency of the tumor also changes.[10] The origin of the PA is myoepithelial cells and intercalated duct cells.[3] The histopathologic appearance of a PA is mainly composed of epithelial and myoepithelial elements, with a variety of patterns ending up embedded in mucopolysaccharide stroma. Fibrosis of the surrounding salivary parenchyma forms a capsule, usually false.[2] PA of the minor salivary gland is known to have more cellular and fewer mesenchymal components. In cases of the elderly, malignant degeneration to carcinoma ex PA must be taken into consideration.[4]

These are usually well-demarcated or encapsulated but the extension of tumor into the capsule is a common feature and sometimes lobules of tumor may appear to be completely separated from the main tumor mass. PA of intraoral accessory glands seldom is allowed to attain a size >1–2 cm in diameter. Because this tumor causes the patient difficulties in mastication, talking and breathing, it is detected and treated earlier than other tumors of major glands. Imaging studies particularly CT and MRI provide useful information on the nature and extent of the lesion, involvement of the lymph nodes, and the status of the surrounding tissues. MRI is preferred because of its ability to define the soft tissues with higher resolution.[4]

There are variants such as PA with lipomatous change, myxolipomatous PA, and PA with squamous differentiation.[11] Studies show that there is the presence of S-100 protein and glial fibrillary active protein-related antigen in these tumors and may be beneficial in diagnosis.[12] MRI is the investigation of choice when avoided for fear of seeding. However, fine-needle aspiration is safe and is the investigation that is most commonly done.

Surgery is the definitive treatment of PA. In oral cavity, it can be removed with a safe margin. Care should be taken that no tissue is left out as recurrence can occur. Spiro in his study concluded that PA had a recurrence rate of around 6% in minor salivary glands.[13] Tumors in the anterior tongue, palate, and others in oral cavity can be removed transorally. Those of base of the tongue and soft palate may require combined transoral-transcervical, transpharyngeal, or transmandibular approaches according to the size and extension. Wherever the lesion and whatever the approach and technique used, a PA of the oral cavity should always be removed. Care must be taken to preserve the hypoglossal and lingual nerves. The use of carbon dioxide laser is becoming more widespread for resection. Recurrence is uncommon and may be attributed to partial excision or a multifocal origin of the tumor.[14],[15]

Differential diagnosis, we considered due to the clinical presentation and localization of the lesion, included mucocele, reactive lesions such as giant cell fibroma or focal fibrous hyperplasia, lipoma, granular cell myoblastoma, neurofibroma, neurilemmoma, vascular leiomyoma, and benign salivary gland neoplasm. Other rare tumors of the tongue are tuberculoma, amyloid tumor, mucoepidermoid carcinoma, and angioleiomyoma. Incisional biopsy gives the pathologist a better chance of diagnosis than fine-needle aspiration, without an increased morbidity. The diagnosis of these tumors is made on the basis of histopathological features. In the present case, biopsy was done for the diagnosis.[16],[17]

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 be 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.
Burbank PM, Dockerty MB, Devine KD. A clinicopathologic study of 43 cases of glandular tumors of the tongue. Surg Gynecol Obstet 1959;109:573-82.  Back to cited text no. 1
    
2.
Berry S, Tay H, Puentes CP. Pleomorphic adenoma of the base of the tongue. Ear Nose Throat J 2004;83:646, 648.  Back to cited text no. 2
    
3.
Gupta AK, Singhal SK, Mann SB, Bapuraj JR, Saran RK. Pleomorphic adenoma presenting as a base of tongue mass. J Laryngol Otol 1997;111:1177-8.  Back to cited text no. 3
    
4.
Yoshihara T, Suzuki S. Pleomorphic adenoma of tongue base causing dysphagia and dysphasia. J Laryngol Otol 2000;114:793-5.  Back to cited text no. 4
    
5.
Tanigaki Y, Mikami Y, Ono M, Tsukuda M. Pleomorphic adenoma of the lateral side of the tongue. Acta Otolaryngol 2004;124:649-51.  Back to cited text no. 5
    
6.
Grewal DS, Pusalkar AG, Phatak AM. Pedunculated pleomorphic adenoma of the tongue base manifesting with dysponea. A case report. J Laryngol Otol 1984;98:425-7.  Back to cited text no. 6
    
7.
van Heerden WF, Raubenheimer EJ. Intraoral salivary gland neoplasms: A retrospective study of seventy cases in an African population. Oral Surg Oral Med Oral Pathol 1991;71:579-82.  Back to cited text no. 7
    
8.
Jorge J, Pires FR, Alves FA, Perez DE, Kowalski LP, Lopes MA, et al. Juvenile intraoral pleomorphic adenoma: Report of five cases and review of the literature. Int J Oral Maxillofac Surg 2002;31:273-5.  Back to cited text no. 8
    
9.
Feinmesser R, Gay I. Pleomorphic adenoma of the hard palate: An invasive tumour? J Laryngol Otol 1983;97:1169-71.  Back to cited text no. 9
    
10.
Chau MN, Radden BG. A clinical-pathological study of 53 intra-oral pleomorphic adenomas. Int J Oral Maxillofac Surg 1989;18:158-62.  Back to cited text no. 10
    
11.
Kondo T. A case of lipomatous pleomorphic adenoma in the parotid gland: A case report. Diagn Pathol 2009;4:16.  Back to cited text no. 11
    
12.
Nakazato Y, Ishizeki J, Takahashi K, Yamaguchi H, Kamei T, Mori T. Localization of S-100 protein and glial fibrillary acidic protein-related antigen in pleomorphic adenoma of the salivary glands. Lab Invest 1982;46:621-6.  Back to cited text no. 12
    
13.
Spiro RH. Salivary neoplasms: Overview of a 35-year experience with 2,807 patients. Head Neck Surg 1986;8:177-84.  Back to cited text no. 13
    
14.
Bansal S, Kalsotra G, Mohammed AW, Bahl A, Gupta AK. Pleomorphic adenoma of base of tongue: Is midline mandibulotomy necessary for approaching benign base tongue lesions? Case Rep Otolaryngol 2012;2012:851501.  Back to cited text no. 14
    
15.
Nascimento LA, Vilela TG. Pleomorphic adenoma of the tongue base: Case report and review. Int Arch Otorhinolaryngol 2014;18:328-31.  Back to cited text no. 15
    
16.
Deitmer T, Stoll W. [Rare tumors of the base of the tongue and their therapy]. HNO 1985;33:366-9.  Back to cited text no. 16
    
17.
Vasudev S, Vakade C, Paramesh RC, Bansal BB. Pleomorphic adenoma: A rare sighting of a common tumour. Ann Dent Spec 2017;3:5.  Back to cited text no. 17
    


    Figures

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



 

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