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National Journal of Maxillofacial Surgery
 
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Table of Contents
CASE REPORT
Year : 2020  |  Volume : 11  |  Issue : 2  |  Page : 289-291  

Pleomorphic adenoma of the upper lip: A rare case report


1 Department of Maxillo-Facial Surgery, District Hospital Baramulla, Baramulla, Jammu and Kashmir, India
2 Department of General Surgery, District Hospital Baramulla, Baramulla, Jammu and Kashmir, India
3 Department of Hospital Administration, District Hospital Baramulla, Baramulla, Jammu and Kashmir, India

Date of Submission31-Mar-2017
Date of Acceptance28-May-2019
Date of Web Publication16-Dec-2020

Correspondence Address:
Dr. Afak Yusuf Sherwani
Department of General Surgery, District Hospital Baramulla, Baramulla, Jammu and Kashmir
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/njms.NJMS_20_17

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   Abstract 


Pleomorphic adenoma (PA), also called mixed tumor, is the most common benign tumor of the salivary glands that mostly occurs in the parotid or submandibular glands but may also occur in the minor salivary glands that are distributed throughout the oral cavity. The common sites of PA of the minor salivary glands are the palates followed by lips and cheeks. Surgical removal with adequate margins is the principal treatment. We present a case of PA (arising from the minor salivary gland of the upper lip) – a rare site in a 65-year-old female who presented with a swelling upper lip of 2-year duration. Fine-needle aspiration cytology of the swelling revealed PA which was confirmed by histopathological examination after complete excision of the swelling.

Keywords: Minor salivary gland, pleomorphic adenoma, upper lip


How to cite this article:
Shah BA, Singh AP, Sherwani AY, Ahmad SM. Pleomorphic adenoma of the upper lip: A rare case report. Natl J Maxillofac Surg 2020;11:289-91

How to cite this URL:
Shah BA, Singh AP, Sherwani AY, Ahmad SM. Pleomorphic adenoma of the upper lip: A rare case report. Natl J Maxillofac Surg [serial online] 2020 [cited 2021 Jan 18];11:289-91. Available from: https://www.njms.in/text.asp?2020/11/2/289/303498




   Introduction Top


Pleomorphic adenoma (PA), or benign mixed tumor, is the most common salivary gland neoplasm, accounting for 60%–65% of all major and minor salivary gland tumors.[1] Minor salivary glands are located in the palate, upper and lower lips, gingiva, floor of the mouth, cheek, tongue, tonsillar areas, nasal cavity, paranasal sinuses, ears, jaw, pharynx, larynx, trachea, and bronchi – where it may give rise to inflammatory conditions and benign and malignant tumors.[2] Although the hard and soft palates are the most common sites of minor salivary gland tumors, the upper lip is relatively uncommon site. Eighty percent of the minor salivary gland tumors located in the lip are benign.[3] It occurs frequently in females, with a female-male ratio ranging from 1.9:1 to 3.2:1 and with a peak incidence between the 5th and the 7th decades of life.[4],[5] This tumor presents as an asymptomatic firm mass with a long period of slow growth rate, whereas secondary to trauma, the clinical features may also include ulceration, pain, or bleeding.[6] Histologically, it is characterized by a large variety of tissues consisting of epithelial cells arranged in a cord-like cell pattern, together with areas of squamous differentiation or with plasmacytoid appearance myoepithelial cells which are responsible for the production of abundant extracellular matrix with chondroid, collagenous, mucoid, and osseous stroma.[7]


   Case Report Top


A 68-year-old female patient reported to our dental outpatient department with complaints of a painless swelling upper lip of 2-year duration which had gradually started increasing from the past 4 months. There was no antecedent history of trauma or any other medical history of significance. Extraoral examination revealed a swelling on the left side of the upper lip with normal overlying skin [Figure 1]. On palpation, the swelling was firm in consistency but nontender. On intraoral examination, a solitary well-circumscribed swelling about 2 cm × 2.5 cm, firm in consistency, mobile but nontender was noted in the upper lip on the left side of the midline. The overlying mucosa was smooth without any ulceration or toxicity [Figure 2]. The systemic examination was normal, and there was no regional lymphadenopathy. Routine baseline investigations were within normal limits. Fine-needle aspiration cytology (FNAC) of the swelling revealed epithelial components and myxoid stroma, suggesting the diagnosis of PA.Excision of the mass was planned under local anaesthesia [Figure 3] and [Figure 4]. Complete excision of the tumor was possible as the tumor was not fixed to the underlying structures, and a primary closure of the defect was done with good cosmetic result [Figure 5] and [Figure 6]. The histopathological section showed well-encapsulated soft-tissue mass consisting of epithelial and myoepithelial cell proliferation lining the ducts and chondromyxoid stroma with the final diagnosis of PA of the upper lip. The patient is on our follow-up without any signs of recurrence.
Figure 1: Extraoral view of the swelling

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Figure 2: Intraoral view of the swelling

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Figure 3: Intra Oral Mucosal Incision over the swelling

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Figure 4: Excision of Swelling

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Figure 5: Primary closure of the defect

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Figure 6: Excised specimen

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   Discussion Top


PA is the most common benign tumor of the minor salivary glands. Females are more affected than males. These tumors are most often diagnosed in the 4th and 6th decades of life. The typical presentation is slow-growing painless firm mass and nontender and tends to be mobile when small but fixed to the surrounding tissue with advanced growth.[8] Prognosis is good if the tumor is well excised. The most common sites among the major salivary gland are the parotid gland (approximately 75%), the submandibular gland (around 5%–10%), and the minor salivary gland (approximately 10%).[9] Among the minor salivary glands, the most common sites are the hard palate and the soft palate, and the lip is relatively uncommon.[10] The main etiopathogenesis of PA remains unclear. Cytogenetic and molecular studies have described that it is of epithelial origin with chromosomal abnormalities at 8q12 and 12q15.[7],[10]

PA of the lip is a rare neoplasm, and therefore, its diagnosis requires a high index of suspicion. It is an epithelial tumor of complex morphology, possessing epithelial and myoepithelial elements arranged in varieties of patterns and embedded in mucopolysaccharide stroma.

A complete wide surgical excision is the treatment of choice. Recurrence after many years of surgical excision as well as malignant transformation should be a concern, and therefore, long-term follow-up is necessary.


   Conclusion Top


PA in the minor salivary gland of the upper lip is very rare. FNAC is a valuable diagnostic adjuvant in preoperative evaluation of salivary gland lesions. It provides a preliminary diagnosis and preoperative assessment on which management decisions can be based. Although rare, PA should be considered as a differential diagnosis of swellings in the upper lip. This lesion, in most frequent cases, is asymptomatic, and the patient may not be aware of its existence and is discovered accidentally by a dentist. Although it is very difficult to diagnose the salivary gland diseases, histopathology is the only gold standard for diagnosis of all lesions.

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.
Forty MJ, Wake MJ. Pleomorphic salivary adenoma in an adolescent. Br Dent J 2000;188:545-6.  Back to cited text no. 1
    
2.
Rosai J, editor. Major and minor salivary glands. In: Ackerman's Surgical Pathology. 9th ed., Vol. 2. St. Louis: Mosby, Elsevier; 2004. p. 873-916.  Back to cited text no. 2
    
3.
Pires FR, Pringle GA, de Almeida OP, Chen SY. Intra-oral minor salivary gland tumors: A clinicopathological study of 546 cases. Oral Oncol 2007;43:463-70.  Back to cited text no. 3
    
4.
Yin WY, Kratochvil J, Stewart JC. Intraoral minor salivary gland neoplasm: Review of 213 cases. J Oral Maxillofac Surg 1989;18:158-62.  Back to cited text no. 4
    
5.
Toida M, Shimokawa K, Makita H, Kato K, Kobayashi A, Kusunoki Y, et al. Intraoral minor salivary gland tumors: A clinicopathological study of 82 cases. Int J Oral Maxillofac Surg 2005;34:528-32.  Back to cited text no. 5
    
6.
Bentz BG, Hughes CA, Lüdemann JP, Maddalozzo J. Masses of the salivary gland region in children. Arch Otolaryngol Head Neck Surg 2000;126:1435-9.  Back to cited text no. 6
    
7.
Lotufo MA, Júnior CA, Mattos JP, França CM. Pleomorphic adenoma of the upper lip in a child. J Oral Sci 2008;50:225-8.  Back to cited text no. 7
    
8.
Eisele DW, Johns ME. Salivary gland neoplasm. In: Bailey BJ, editor. Head & Neck Surgery-Otolaryngology. Philadelphia: Lippincott Williams & Wilkins; 2001. p. 1279.  Back to cited text no. 8
    
9.
Huvos AG. Paulino AF. Salivary glands. In: Sternberg's Diagnostic Surgical Pathology. 4th ed., Vol. 2. Philadelphia, PA, USA: Lippincott Williams & Wilkins; 2004. p. 933-62.  Back to cited text no. 9
    
10.
Debnath SC, Adhyapok AK. Pleomorphic adenoma (benign mixed tumour) of the minor salivary glands of the upper lip. J Maxillofac Oral Surg 2010;9:205-8.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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