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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 32  |  Issue : 1  |  Page : 73-76

Pleomorphic adenoma of dorsolateral surface of the tongue: A rarest clinical presentation


1 Department of Oral Medicine and Radiology, New Horizon Dental College and Research Institute, Raipur, Chhattisgarh, India
2 JNM Medical College, Department of Surgical Oncology, Raipur, Chhattisgarh, India
3 Oral and Maxillofacial Surgeon, Om Multispeciality Hospital, Raipur, Chhattisgarh, India
4 Department of Oral Medicine and Radiology, Swami Devi Dayal Hospital and Dental College, Panchkula, Haryana, India

Date of Submission26-Jan-2020
Date of Decision27-Feb-2020
Date of Acceptance15-Mar-2020
Date of Web Publication17-Apr-2020

Correspondence Address:
Dr. Ramanpal Singh Makkad
Department of Oral Medicine and Radiology, New Horizon Dental College and Research Institute, Bilaspur, Chhattisgarh
India
Ravleen Nagi
Department of Oral Medicine and Radiology, Swami Devi Dayal Hospital and Dental College, Panchkula, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_15_20

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   Abstract 


Pleomorphic adenoma is most common benign neoplasm of the major salivary glands that predominantly affects the superficial lobe of the parotid gland and rarely minor salivary glands. These tumors commonly occur in the fourth, fifth and sixth decades with increased predilection towards females and clinically manifests as unilateral, slow growing, firm and painless mass. Their occurrence on the tongue has been very rarely reported in the literature. This case report describes a rarest case of pleomorphic adenoma involving the right dorsolateral surface of tongue in a 28 year old male patient.

Keywords: Adenoma, mixed tumor, parotid gland, pleomorphic, tongue


How to cite this article:
Makkad RS, Agrawal G, Agrawal V, Nagi R. Pleomorphic adenoma of dorsolateral surface of the tongue: A rarest clinical presentation. J Indian Acad Oral Med Radiol 2020;32:73-6

How to cite this URL:
Makkad RS, Agrawal G, Agrawal V, Nagi R. Pleomorphic adenoma of dorsolateral surface of the tongue: A rarest clinical presentation. J Indian Acad Oral Med Radiol [serial online] 2020 [cited 2020 May 30];32:73-6. Available from: http://www.jiaomr.in/text.asp?2020/32/1/73/282605




   Introduction Top


Pleomorphic adenoma (PA) also known as mixed tumor, is most commonly encountered benign neoplasm affecting both major and minor salivary glands, and constitutes up to two third of all salivary gland neoplasms.[1] The term “pleomorphic Adenoma” has been proposed by “Willis” because of its characteristic histopathological features. These tumors have been reported to often arise in the superficial lobe of the parotid gland but may also involve minor salivary glands and the submandibular gland.[2] Other regions that are less commonly affected are lips, neck, oral and nasal cavity. Corresponding to minor salivary glands, palate is the most frequently encountered intraoral site due to preponderance of minor salivary glands followed by labial and buccal mucosa, tongue, floor of mouth, tonsils, pharynx, retro molar area and gingiva.[3]

PA occurs in individuals between fourth and sixth decade of age, and has predilection towards females, with female to male ratio 1.9:1. It clinically manifests as a slowly progressing, asymptomatic swelling of parotid gland without the involvement of facial nerve. The swelling is usually unilateral, firm in consistency that may enlarge to a huge size if left untreated.[4] Several cases of PA affecting various intra and extraoral sites have been documented in the literature and among them only two cases involving the tongue have been published.[5],[6] Tongue is unusual site of occurrence of PA, and when they occur they usually involve posterior, followed by anterior and rarely lateral lingual glands i.e. Von Ebner glands of the tongue. We present a case of PA involving the lateral lingual glands on the dorsum of the tongue, which is considered as a rarest site for its occurrence.


   Case Report Top


A 28-year-old male patient reported to the department with a complaint of slowly progressing painless swelling on right side of tongue since two years. The swelling was initially small in size and has gradually progressed to the present size. Associated symptoms of dysphagia and difficulty while talking were not present. There was no previous history of trauma, or any relevant past medical and dental history. On clinical examination, a solitary, well circumscribed oval mass measuring approximately 2 × 2 cm was observed on the right dorsolateral surface of the tongue. Overlying mucosa was intact and the surface appeared smooth and mildly erythematous [Figure 1]. On palpation growth was firm, non-tender, non-fluctuant, non-reducible, compressible and non-pulsatile. No bleeding or discharge was observed. Blanching was also not noted on application of digital pressure over the lesion. There were no clinically palpable lymph nodes. Based on history and clinical examination, provisional diagnosis of neurofibroma was considered with a differential diagnosis of hemangioma, vascular leiomyoma, granular cell myoblastoma, giant cell fibroma and benign salivary gland neoplasm.
Figure 1: Nodular well circumscribed, sessile growth on the right lateral dorsal surface of tongue

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After routine blood investigations, which were normal, incisional biopsy was done under local anesthesia and specimen was sent for histopathological examination that revealed non keratinized epithelium, round to oval islands of epithelial cells, myoepithelial cells, ductal differentiation and cartilaginous areas within the chondromyxoid connective tissue stroma. Mitotic figures or any type of vascular invasion was not seen. Microscopic features were suggestive of benign salivary gland neoplasm and final diagnosis of pleomorphic adenoma was established [Figure 2]. Later, the mass was surgically excised with wide margins under general anesthesia, excised mass was well encapsulated, measuring 2 × 2 cm with brownish surface [Figure 3]. The postoperative healing was uneventful. There has been no any sign of recurrence till date [Figure 4].
Figure 2: Histopathological examination (H and E, 100×) reveals non keratinized stratified epithelium, round to oval epithelial islands, myoepithelial cells, ductal differentiation and collage nous areas in chondromyxoid connective tissue stroma

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Figure 3: Excised mass measuring 2 × 2 cm with brownish surface

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Figure 4: Postoperative healing after 1 month follow up visit

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   Discussion Top


PAs account for 80% of salivary gland neoplasm's that chiefly arise in the parotid gland, and 8% occur in the minor salivary glands. Majority of PAs are benign and approximately 1.9 to 23.3% have been found to undergo malignant transformation. They are generally discovered during routine physical examination, as an asymptomatic mass.[7] Mostly patients are unaware of these tumors unless they enlarge to huge size and cause discomfort while chewing or during talking. When originating in the minor salivary glands, majority of PA occur on the palate due to the highest concentration of minor salivary glands and typically presents as a firm or rubbery submucosal mass without ulceration.[8] They rarely involve the tongue and to our best knowledge only two cases have been reported in the literature on the dorsolateral surface of the tongue.[5],[6] In the present case, solitary well-circumscribed sessile mass was observed on the right lateral half of dorsum of tongue in a 28 year old male patient, and involvement of lateral lingual glands is extremely uncommon.

The diagnosis of PA is usually confirmed from the patient's history, physical examination, cytology and by histopathological examination. An incisional biopsy helps the clinician in establishing proper diagnosis and treatment plan than fine needle aspiration cytology with minimal morbidity to the patient.[8] Clinically, painless nodular mass on the dorsal surface of the tongue could be suspected to be hemangiomas, vascular leimyomas, fibroma, neurofibroma, benign minor salivary gland neoplasm (pleomorphic adenoma) and granular cell myoblastoma. Hemangioma of tongue present as bluish or red painless mass, usually blanches on digital pressure and are pulsatile on palpation; vascular leiomyomas are rarely seen in the oral cavity, if occur they often manifest as painless submucosal mass, soft, freely mobile, compressible and exhibit slip sign on palpation; fibromas are non-tender, firm and fibrous on palpation; solitary oral neurofibroma is rare entity seen as painless, slowly progressive sessile mass, occasionally associated with paraesthesia or pain due to nerve compression; granular cell myoblastoma appears as pale yellowish painless, sessile submucosal nodular mass of gummy consistency [Table 1].[9],[10],[11],[12],[13],[14]
Table 1: Illustrates comparison of clinical and histopathological features of swellings of tongue

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Histopathological examination is necessary to establish the true nature of the lesion. In this case, definitive diagnosis was established by incisional biopsy and microscopic features such as presence of islands of epithelial cells, myoepithelial cells, ductal differentiation in chondromyxoid connective tissue stroma were suggestive of benign salivary gland neoplasm. As PA presents with a diversified histopathologic presentation, it may be confused with other tumors like myoepithelioma, adenoid cystic carcinoma, mucoepidermoid carcinoma and basal cell adenoma.[10]

In majority of the reported cases, the base of the tongue constituted 1% of intraoral minor salivary gland tumors. Mostly patients were male, aged between 29-87 years, who were not aware of the occurrence of tumor until they developed dysphagia, these tumors were detected on routine physical examination in some of the patients.[3],[6] As mentioned earlier, literature search revealed only two cases of PA affecting the dorsolateral surface of the tongue, first case was reported by Tanigaki et al.[5] in 2004 from Japan, in a 54-year-old woman on the lateral part of the tongue dorsum originating from Ebner's glands successfully treated by partial glossectomy. Second case was reported by Chhabra et al.[6] in 2019 from India, in a male patient aged 41 years on the left anterolateral dorsal surface of the tongue. Ours is the third case of PA arising from Ebner's glands on the right dorsolateral surface of the tongue and was treated by wide surgical excision under general anesthesia.

Although tumor is well encapsulated, wide surgical resection with adequate margins is recommended to prevent recurrence. Malignant transformation has been documented for 50% of intraoral minor salivary gland tumors to adenoid cystic carcinoma as common malignant tumor due to lack encapsulation in some of the tumors on the palate, buccal mucosa, tongue and lip. Yamada et al.[8] reported a case of histopathologically-diagnosed adenocarcinoma on the left anterior surface of the tongue in a 64-year-old male patient successfully treated by wide local excision with left upper neck dissection and no signs of recurrence were observed 22 months after surgery. In our patient, no recurrence was observed on the regular follow up visits till date. Recurrence rate of 5-30% has been found for PA which could be due to an inadequate excision or exposure of the capsule, but adenomas arising in the minor glands have little chances of recurrence.[10]


   Conclusion Top


Salivary gland lesions and painless nodular mass on the tongue present diagnostic challenge to both the clinician and the oral pathologist. The patient's history, complete clinical examination followed by histopathological examination play a key role in providing confirmatory diagnosis for proper treatment selection. Complete wide surgical excision is the treatment of choice and long term follow up of patients is necessary due to late recurrence and malignant transformation of this benign neoplasm.

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.
Alves FA, Perez DE, Almeida OP, Lopes MA, Kowalski LP. Pleomorphic adenoma of the submandibular gland: Clinicopathological and immunohistochemical features of 60 cases in Brazil. Arch Otolaryngol Head Neck Surg 2002;128:1400-3.  Back to cited text no. 1
    
2.
Ledesma-Montes C, Garces-Ortiz M. Salivary gland tumors in a Mexican sample. A retrospective study. Med Oral 2002;7:324-30.  Back to cited text no. 2
    
3.
Pons Vicente O, Almendros Marqués N, Berini Aytés L, Gay Escoda C. Minor salivary gland tumors: A clinicopathological study of 18 cases. Med Oral Patol Oral Cir Bucal 2008;13:E582-8.  Back to cited text no. 3
    
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Gbotolorun OM, Arotiba GT, Effiom OA, Omitola OG. Minor salivary gland tumours in a Nigerian hospital: A retrospective review of 146 cases. Odontostomatol Trop 2008;31:17-23.  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.
Chhabra S, Bhutani N, Jain P, Gill M, Nishisth N, Sen R. 'Pleomorphic adenoma of the tongue: A common entity at the uncommon location'. Ann Med Surg (Lond) 2018;;38:34-6.  Back to cited text no. 6
    
7.
Rahnama M, Orzędała-Koszel U, Czupkałło L, Lobacz M. Pleomorphic adenoma of the palate: A case report and review of the literature. Contemp Oncol (Pozn) 2013;17:103-6.  Back to cited text no. 7
    
8.
Yamada SI, Yanamoto S, Rokutanda S, Matsutani K, Kawasaki G, Kawano T, et al. Carcinoma ex pleomorphic adenoma in minor salivary glands of the anterior tongue: A case report. J Oral Maxillofac Surg Med Pathol 2013;25:197-200.  Back to cited text no. 8
    
9.
Patrick J, Bradley M. Recurrent salivary gland pleomorphic adenoma: Etiology, management and results. Curr Opin Otolaryngol Head Neck Surg 2001;9:100-8.  Back to cited text no. 9
    
10.
Pranitha V, Puppala N, Deshmukh SN, Jagadesh B, Anuradha S. Cavernous hemangioma of tongue: Report of two cases. J Clin Diagn Res 2014;8:15-7.  Back to cited text no. 10
    
11.
Kolte AP, Kolte RA, Shrirao TS. Focal fibrous overgrowths: A case series and review of literature. Contemp Clin Dent 2010;1:271-4.  Back to cited text no. 11
[PUBMED]  [Full text]  
12.
Roy R, Chakraborty S, Das S, Roy A. Solitary neurofibroma at the base of the tongue. A Rare presentation. Indian J Dermatol 2015;60:497-9.  Back to cited text no. 12
[PUBMED]  [Full text]  
13.
Rawal SY, Rawal YB. Angioleiomyoma (vascular leiomyoma) of the oral cavity. Head Neck Pathol 2018;12:123-6.  Back to cited text no. 13
    
14.
Eguia A, Uribarri A, Gay Escoda C, Crovetto MA, Martínez-Conde R, Aguirre JM. Granular cell tumor: Report of eight intraoral cases. Med Oral Patol Oral Cir Bucal 2006;11:425-8.  Back to cited text no. 14
    


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