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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 28  |  Issue : 1  |  Page : 101-104

Submandibular pleomorphic adenoma in a male: An unusual case report


1 Department of General Surgery, Kakatiya Medical College, Warangal, Telangana, India
2 Department of General Surgery, Dr. NTR University of Health Sciences, Srikakulam, Andhra Pradesh, India
3 Department of General Surgery, GMERS Medical College, Gandhinagar, Gujarat, India

Date of Submission02-Mar-2022
Date of Decision15-Apr-2022
Date of Acceptance04-May-2022
Date of Web Publication14-Jul-2022

Correspondence Address:
Dr. Tarun Kumar Suvvari
Dr. NTR University of Health Sciences, Srikakulam, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ksj.ksj_6_22

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  Abstract 


Pleomorphic adenoma (PA) of the submandibular gland is a very unusual neoplasm and comprises only 8%–10% of all benign tumours, most commonly affecting females between 30 and 50 years of age. In this case report, we described a case of a 36-year-old male patient who presented with swelling in the left neck region for 5 years, associated with dull aching pain and increased salivation. The ultrasonography and plain computed tomography (CT) of the neck revealed iso-hypointense lesions arising from the left submandibular gland. Fine needle aspiration cytology of the swelling revealed sheets and clusters of cells with an eccentric nucleus in the chondromyxoid and haemorrhagic background, and a probable diagnosis of PA was made based on Milan's scoring system. The patient underwent an excision of the gland. Post-operative biopsy showed islands of mesenchymal and epithelial components with intervening fibrous tissue and areas of haemorrhage, confirming the diagnosis of submandibular PA.

Keywords: Chondromyxoid stroma, Milan's scoring, pleomorphic adenoma, Wharton's duct


How to cite this article:
Guguloth MN, Kuchana SK, Suvvari TK, Gopu AR, Mamidi SR, Mittal RJ. Submandibular pleomorphic adenoma in a male: An unusual case report. Kerala Surg J 2022;28:101-4

How to cite this URL:
Guguloth MN, Kuchana SK, Suvvari TK, Gopu AR, Mamidi SR, Mittal RJ. Submandibular pleomorphic adenoma in a male: An unusual case report. Kerala Surg J [serial online] 2022 [cited 2022 Sep 24];28:101-4. Available from: http://www.keralasurgj.com/text.asp?2022/28/1/101/350906




  Introduction Top


Salivary gland tumours are uncommon and comprise only up to 3% of head and neck tumours.[1] The parotid gland is the most commonly affected salivary gland that is as high as in 70% of cases, followed by submandibular gland in 5%–10% of cases and at last the sublingual gland that is only 1% of cases. Minor salivary glands are affected in 5%–15% of cases.[2]

The term 'Pleomorphic adenoma (PA)' was coined by Willis. In the early years, it used to be referred to as a mixed tumour, enclavoma, endochroma, etc.[3] PA of the submandibular gland is quite uncommon and involves only up to 8%–10% of all benign neoplasms of salivary glands frequent in the third to fifth decade of life with a sexual predilection towards females (female: male 3.5:1).[1] They are characterised by painless enlargement of the submandibular triangle.[4] If ignored, can achieve grotesque proportions and weigh up to many kilograms and can cause facial disfigurement and rarely airway compromise.[5]

The investigations include computed tomography (CT), which is considered the gold standard radiological tool. The fine needle aspiration helps in differentiating the tumour from inflammatory conditions and enlargement of lymph nodes.[1] The treatment includes complete excision of the gland to forestall recurrences.


  Case Report Top


A 36-year-old male patient was presented with swelling in the left neck region for 5 years to the outpatient department of general surgery. The swelling was insidious in onset, gradually progressive associated with dull aching pain radiating to the jaw and increased in salivation [Figure 1]. On extra-oral examination, globular swelling in the left submandibular region of approximate size 4 cm × 3 cm, 3 cm lateral to the symphysis menti and 2 cm medial to the angle of mandible, was seen with well-defined borders, firm consistency, not adherent to above skin and mobile in all planes.
Figure 1: Globular swelling of 4 cm × 3 cm in size can be seen 3 cm lateral to symphysis menti and 2 cm medial to angle of mandible

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The workup included ultrasonography (USG) of the neck which revealed a 3 cm × 2 cm well-defined hypo-echoic lesion in the left submandibular gland. Coronal and axial planes of CT of the plain neck revealed a well-defined homogeneously iso-hypointense lesion measuring 3 cm × 3 cm arising from the left submandibular gland, suggestive of adenoma of the submandibular gland. A fine needle aspiration was done, and the cytology revealed sheets and clusters of cells with some plasmacytoid looking cells with an eccentrically placed nucleus in a chondromyxoid fragment and haemorrhagic background [Figure 2]. Presumptive diagnosis of PA was made using Milan's scoring system, and decision was taken to surgically excise lesion under general anaesthesia.
Figure 2: Fine needle aspiration cytology revealing sheets and clusters of cells with plasmacytoid appearance and eccentric nuclei in a chondromyxoid and haemorrhagic background

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A horizontal incision was given on the left submandibular gland and deepened through the platysma, the gland was identified and separated from the surrounding lingual nerve and facial vein, and the entire left submandibular gland was excised along with the left Wharton's duct [Figure 3]. Haemostasis is secured and the wound is closed in layers. Post-operative and healing of wounds are uneventful.
Figure 3: Surgically resected left submandibular gland along with Wharton's duct

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Post-operative biopsy revealed normal serous acini with partially encapsulated tumour tissue arranged in sheets and islands showing mesenchymal and epithelial components. Epithelial cells are arranged in an acinar pattern, few gland-like, round to oval with the round nucleus. Myoepithelial cells are spindle-shaped, also showing mesenchymal components with the myxoid area and intervening fibrous tissue and areas of haemorrhage [Figure 4] confirming the diagnosis of PA.
Figure 4: Postoperative biopsy showing well-encapsulated tumour tissue with epithelial and mesenchymal components (epithelial components with ductal epithelial cell arranged in cords and surrounded by chondromyxoid stroma)

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The patient was discharged after 10 days of surgery, and the follow-up was uneventful. The patient was followed up for 3 months, there was no recurrence, and the patient was still under follow-up.


  Discussion Top


The term PA was coined by Willis. In the early years, it used to be referred to as a mixed tumour, enclavoma, endochroma, etc.[3] Submandibular tumours are rare, comprising less than 2% of head and neck neoplasms. PA is 36% of all common neoplasms of the submandibular gland which encompasses adenoid cystic carcinoma (25%), mucoepidermoid carcinoma (12%) and malignant mixed tumour (10%).[4] The differential diagnosis must include basal cell carcinoma, adenocarcinoma and lymphoma.

PA commonly presents as slow growing, unilateral and painless mass in the third to fifth decades of life. It has a female preponderance, with a ratio of 3.5:1 and usually occurs in the parotid gland (90% of benign neoplasms of parotid) followed by submandibular and sublingual glands (8%–10%).[1],[6] They are characterised by painless enlargement of the submandibular triangle.[4] If ignored, can achieve grotesque proportions and weigh up to many kilograms and can cause facial disfigurement and rarely airway compromise.[5]

A single institutional study by Subhashraj in India confirmed that out of 684 cases of salivary gland tumours analysed retrospectively, 422 (62%) were benign, and out of them, 61% were in the parotid gland and 17% in the submandibular glands.[7] A comparable find out via Alves et al., Brazil, revealed that out of 60 cases of PA, 37 of them were female.[8] A systematic literature on reports of PA from 2000 to 2018 by Almeslet revealed that the mean age of PA was 44.14 years (range: 13–75 years) with woman predilection with a male-female ratio of 8:13 and the most often placed site was the left or right side of the neck (42.8%).[3] However, in our case, swelling of the left side of the neck was seen in a 36-year-old male patient.

CT determines the vicinity and dimensions of the tumours, the distinction between the tumour and surrounding tissues, detection of its capsule and any bone resorption around the tumour.[9] PA has a risk of malignant transformation of about 1.5% up to 5 years and will increase to 9.5% after more than 15 years. Fine needle aspiration helps in differentiating between tumour and inflammatory conditions of salivary glands or enlarged lymph nodes.[1] The stage of the accuracy of fine needle aspiration cytology (FNAC) in distinguishing malignancy and benign is 79.1%.[10] In our case, CT was performed to understand the extension of the lesion, and the preoperative diagnosis was finished through FNAC.

The treatment of choice for submandibular gland PA is submandibular gland excision in toto along with tumour.[11] Even though the tumours are well encapsulated, there are pseudopodic microscopic extensions into the surrounding tissues because of dehiscence in the false capsule.[12] A retrospective case series employed by Munir and Bradley in a tertiary referral centre in the UK (1988–2004) revealed that 37 cases had been surgically excised – 41% with extra-capsular gland excision and 59% through selective level Ib, IIa and III neck dissection.[13]

The final pathological diagnosis is always established by post-operative biopsy with complex morphology, possessing epithelial and myoepithelial elements intermingled with mucoid, myxoid or chondroid tissue embedded in mucopolysaccharide stroma.[14] Because of the varied histopathology of PA, it is frequently stressed with myoepithelioma and mucoepidermoid carcinoma. Myoepithelioma and mucoepidermoid lacks glandular ductal differentiation and absence of chondromyxoid stroma.[15]


  Conclusion Top


In our case, submandibular PA presented as a slow-growing swelling and affected a male patient while it is more common in females. Surgical excision is curative; however, due to dehiscence in the false capsule, local excision is not recommended. Since there is a significant rate of recurrence in the tumour bed, submandibular gland excision along with tumour is preferred.

Acknowledgement

Our sincere thanks to Squad Medicine and Research for their support and guidance.

Informed consent

Taken from the patient.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Rai S, Sodhi SP, Sandhu SV. Pleomorphic adenoma of submandibular gland: An uncommon occurrence. Natl J Maxillofac Surg 2011;2:66-8.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Patil P, Burde K, Naikmasur VG, Thorawat A. Pleomorphic adenoma of submandibular gland: A case report with review of literature. Dent Res J (Isfahan) 2014;11:411-4.  Back to cited text no. 2
    
3.
Almeslet AS. Pleomorphic adenoma: A systematic review. Int J Clin Pediatr Dent 2020;13:284-7.  Back to cited text no. 3
    
4.
Hanna E, Lee S, Fan C, Suen J. Benign neoplasms of the salivary glands. In: Cummings C, Flint P, Harker L, Haughey B, Richardson M, Robbins T, et al., editors. Cummings Otolaryngology Head & Neck Surgery. Philadelphia: Mosby; 2005. p. 1348-77.  Back to cited text no. 4
    
5.
Perumal CJ, Meyer M, Mohamed A. A giant pleomorphic adenoma of the submandibular salivary gland: A case report. Craniomaxillofac Trauma Reconstr 2012;5:185-8.  Back to cited text no. 5
    
6.
Becerril-Ramírez PB, Bravo-Escobar GA, Prado-Calleros HM, Castillo-Ventura BB, Pombo-Nava A. Histology of submandibular gland tumours, 10 years' experience. Acta Otorrinolaringol Esp 2011;62:432-5.  Back to cited text no. 6
    
7.
Subhashraj K. Salivary gland tumors: A single institution experience in India. Br J Oral Maxillofac Surg 2008;46:635-8.  Back to cited text no. 7
    
8.
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. 8
    
9.
Firdaus MA, Pulungan MR. Penatalaksanaan Adenoma Pleomorfik Parotis. Bagian Telinga Hidung Tenggorok Bedah Kepala Leher Fakultas Kedokteran Universitas Andalas/RSUP Dr. M. Djamil, Padang, Malaysia; 2011.  Back to cited text no. 9
    
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Mubeen K, Vijayalakshmi K, Abhisek R, Girish B, Singh C. Benign pleomorphic adenoma of minor salivary gland of palate. J Dent Oral Hygiene 2011;3:82-8.  Back to cited text no. 10
    
11.
Eveson JW, Cawson RA. Salivary gland tumours. A review of 2410 cases with particular reference to histological types, site, age and sex distribution. J Pathol 1985;146:51-8.  Back to cited text no. 11
    
12.
Sergi B, Limongelli A, Scarano E, Fetoni AR, Paludetti G. Giant deep lobe parotid gland pleomorphic adenoma involving the parapharyngeal space. Report of three cases and review of the diagnostic and therapeutic approaches. Acta Otorhinolaryngol Ital 2008;28:261-5.  Back to cited text no. 12
    
13.
Munir N, Bradley PJ. Pleomorphic adenoma of the submandibular gland: An evolving change in practice following review of a personal case series. Eur Arch Otorhinolaryngol 2007;264:1447-52.  Back to cited text no. 13
    
14.
Van der Wall I. Salivary gland neoplasm. In: Prabhu SR, Wilson DF, Daftary DK, Johnson NW, editors. Oral Disease in the Tropics. New York: Oxford University Press; 1992. p. 478-86.  Back to cited text no. 14
    
15.
Civelek S, Ercan B, Cakir B, Basak T, Turgut S. Malignant myoepithelial carcinoma arising in a pleomorphic adenoma of the submandibular gland. KBB Forum 2006;5:4.  Back to cited text no. 15
    


    Figures

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



 

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