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Table of Contents   
CASE REPORT  
Year : 2021  |  Volume : 32  |  Issue : 1  |  Page : 120-123
Asymptomatic palatal mass: A case report


1 Department of Oral Medicine and Radiology, Faculty of Dental Sciences, Sri Ramachandra Medical College, Chennai, Tamil Nadu, India
2 Department of Radiation Oncology, Faculty of Dental Sciences, Sri Ramachandra Medical College, Chennai, Tamil Nadu, India
3 Oral and Maxillofacial Surgery, Faculty of Dental Sciences, Sri Ramachandra Institute of Higher Education and Research (DU), Chennai, Tamil Nadu, India
4 Oral Pathology, Faculty of Dental Sciences, Sri Ramachandra Institute of Higher Education and Research (DU), Chennai, Tamil Nadu, India

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Date of Submission23-Nov-2018
Date of Decision04-Jan-2020
Date of Acceptance25-Feb-2021
Date of Web Publication13-Jul-2021
 

   Abstract 


Accidental detection of asymptomatic clinically suspicious lesions is a common occurrence in routine clinical examination of the oral cavity. In certain cases, these lesions may mimic benign lesions further adding to confusion. In this case report, we have discussed the case of a 51-year-old male patient with an asymptomatic palatal mass which was detected during a routine dental check-up and subsequently confirmed to be an adenoid cystic carcinoma (ACC) arising from the minor salivary glands. The patient underwent right partial maxillectomy followed by radiation therapy and has been disease-free for 6 years and is on follow-up. ACC is a malignant tumour of the salivary glands commonly occurring in the palate, characterized by a slow indolent growth phase, with a high predilection for late recurrences which can be local or systemic. This article describes the importance of recognizing this clinical entity as a differential diagnosis in the evaluation of asymptomatic palatal lesions to enable early diagnosis and institution of appropriate treatment to successfully treat the disease.

Keywords: Adenoid cystic carcinoma, maxillectomy, metastasis, radiation therapy

How to cite this article:
Divyambika C V, Srinivas K S, Subramanian S S, Elengkumaran S, Malathi N. Asymptomatic palatal mass: A case report. Indian J Dent Res 2021;32:120-3

How to cite this URL:
Divyambika C V, Srinivas K S, Subramanian S S, Elengkumaran S, Malathi N. Asymptomatic palatal mass: A case report. Indian J Dent Res [serial online] 2021 [cited 2021 Nov 27];32:120-3. Available from: https://www.ijdr.in/text.asp?2021/32/1/120/321388



   Introduction Top


Adenoid cystic carcinoma (ACC) is often described as a slow-growing, malignant neoplasm, arising from the major and minor salivary glands of the head and neck.[1] ACC is most commonly seen in the oral cavity with a predilection to involve the hard palate.[2],[3] Presenting as painless, non-ulcerated swellings these lesions can clinically masquerade as benign salivary gland tumours, most commonly pleomorphic adenoma involving the palate. Other differential diagnoses include lymphoproliferative diseases like non-Hodgkin's lymphoma, lymphoid hyperplasia, and neoplasm involving maxillary sinus, with infrastructure erosion and extension to oral cavity presenting as a non-ulcerative mass.[4],[5] ACC is associated with a higher rate of local recurrence after surgery with a distinctive tendency for perineural invasion. Distant metastasis, although uncommon in ACC, may occur years after successful treatment of the primary tumour, which is a unique feature of this neoplasm.[6] We herewith report a 51-year-old patient, who came for a routine dental check-up, was found to have a palatal mass. The patient was diagnosed to have ACC and was subsequently treated.


   Case Report Top


A 51-year-old male patient reported to the Department of Oral Medicine and Radiology for a routine dental check-up. On intraoral examination, there was evidence of a well-defined non-ulcerated swelling around 1.5 cm in diameter in the posterior aspect of the palate distal to 18 adjacent to the maxillary tuberosity region [Figure 1]. Mucosa over the swelling appeared erythematous. On palpation, the swelling was non-tender, soft in consistency, non-fluctuant and fixed to the underlying palate. A single right submandibular lymph node was palpable measuring around 1.5 × 2 cm, which was firm, non-tender, and freely mobile.
Figure 1: The presence of intraoral swelling in the posterior hard palate

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Since the patient was not aware of the swelling, the duration of the swelling could not be elicited. The patient gave no history of previous trauma or symptoms such as pain or paraesthesia in the right maxillary region.

Patient consent was obtained for the rest of the procedures planned. Intraoral periapical radiograph in relation to 18, orthopantomogram and paranasal sinus (PNS) view revealed no pathological findings [Figure 2], [Figure 3], [Figure 4]. Computed tomography (CT) of the PNS with contrast revealed an ill-defined heterogeneously enhancing lesion involving the posterior aspect of the hard palate on the right side [Figure 5]. The greater palatine foramen was not widened ruling out the perineural spread. A provisional diagnosis of benign minor salivary gland neoplasm was given.
Figure 2: Intraoral periapical radiograph of 18, with no evidence of pathology

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Figure 3: PNS view, with no evidence of pathology

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Figure 4: Reveals orthopantomogram, with no evidence of pathology

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Figure 5: Sagittal and axial sections of CT PNS with contrast respectively, which revealed an ill-defined heterogeneously enhancing lesion involving the posterior aspect of the hard palate on the right side

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Incisional biopsy was carried out under local anaesthesia and the tissue was histopathologically evaluated. The section showed proliferating ductal and basaloid cells arranged in a cribriform pattern with eosinophilic coagulum. Overlying stratified squamous epithelium along with few mucinous acini was evident, suggestive of ACC.

The patient underwent right partial maxillectomy with selective neck dissection [Figure 6]. Postoperative histopathological examination was suggestive of monomorphic basaloid hyperchromatic cells arranged in a ductular pattern along with mucin-filled spaces suggestive of grade 1 cribriform type of ACC with no lymphovascular and perineural invasion. The immunohistochemical evaluation showed positivity for C kit, confirming the diagnosis of ACC [Figure 7], [Figure 8], [Figure 9]. Ki67 immunoexpression was done, which showed weak positivity [Figure 10].Subsequently, the patient underwent postoperative radiotherapy (RT) to the right maxillary bed, base of the skull, and right-side upper neck to a dose of 60 Gy in 30 fractions using intensity-modulated radiation therapy (IMRT) with 6 MV photons. The patient-tolerated RT well and completed the planned radiation protocol within the stipulated time of 6 weeks. At 8 weeks post-radiation, the patient underwent whole-body positron emission tomography (PET) with FDG-18 isotope and the PET scan showed no evidence of residue and local/distal recurrence. Patient is currently under follow up and is disease free since 6 years post-treatment. At the time of the last evaluation, he was clinically in remission and annual PET CT was negative for local or metastatic recurrence [Figure 11].
Figure 6: Postoperative photograph

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Figure 7: H and E 10 × showing numerous cystic spaces along with tumour islands

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Figure 8: H and E 40× showing swiss cheese pattern

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Figure 9: 20× view showing C-kit membrane positivity

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Figure 10: Weak positivity of Ki 67

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Figure 11: Five years post-treatment PET CT with no metabolically active disease

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   Discussion Top


ACC is a relatively rare neoplasm of the head and neck. It primarily involves the minor salivary glands of the palate, and the other sites of involvement include PNSs, nasopharynx, trachea, lacrimal gland, breast, external auditory canal, cervix, and vulva.[3],[7],[8] This tumour is more common in the elderly age group with the highest incidence in the fourth to the seventh decades.[8] The gender predilection is not very certain, however, the study done by Rodriguez et al. in 263 patients with ACC shows a slight female predilection of 56.5%.[8] Most of the cases tend to be asymptomatic for a long course of time and exhibit a slow growth phase. Mitra et al. reported a case of ACC presenting as an asymptomatic ulcer involving the palate undiagnosed for 15 years.[7] The present case adds to clinical significance as the patient was unaware of the asymptomatic swelling and was detected only during a routine intraoral examination. This emphasises the importance of a proper routine clinical examination for early detection of such lesions. Despite their indolent clinical phase, the unique feature of this neoplasm is its inherent tendency for perineural invasion, local recurrence, and distant metastasis, most commonly to lungs.[8],[9] The three histological variants identified are the solid, tubular, and cribriform patterns. The present case belonged to the cribriform type, which has been the most commonly encountered subtype.[8] Immunopositivity with C kit (CD117) is strongly associated with ACC and has been found to be a valuable marker in differentiating ACC from polymorphous low-grade adenocarcinoma (PLGA).[10],[11] A strong immunopositivity to C kit was observed in the present case, further confirming the diagnosis of ACC. The treatment regimen includes surgical excision followed by RT because of the risk of neural invasion, local recurrence, and distant metastasis.[12] In a study done by Spiro et al. in 22 patients diagnosed with ACC, it has been suggested that radical resection combined with RT, improves outcome in ACC.[2] The present case underwent surgical excision (R0 resection) followed by adjuvant radiation therapy to improve local control. PET-CT is a valuable diagnostic imaging modality in detecting local recurrence and distant metastasis.[13] In the present case, PET-CT, taken 5 years post-therapy showed the absence of metabolically active disease. However, such patients need to be followed up for a longer period because of its unpredictable clinical course. In a retrospective study, certain parameters such as high T classification, positive surgical margins, positive nodal status, neural invasion, lymphovascular invasion, and tumour site such as the floor of mouth, tongue, sinonasal tract, palate were identified as significant predictors of decrease in overall survival rate.[14] For predicting the tumour progression, Ki-67 expression is found to be higher in tumours failing treatment, and hence, may serve as a prognostic indicator of the tumour.[15] The present case had a favourable survival rate because of the disease stage, absence of perineural invasion, and low Ki-67 value. However long-term follow up of such patients is mandatory because these tumours are characterised by high 5-year survival rates and very low 10–20-year survival rates.[16]


   Conclusion Top


Adequate clinical examination aids in the early diagnosis of such asymptomatic ACC which helps in better prognosis, thus preventing the associated complications. Long-term monitoring of the patient is necessary for identifying any relapse and reduce the associated morbidity and mortality.

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.
Barnes L, Everson JW, Reichart P, Sidransky D. “Tumors of the salivary glands: WHO and TNM classification”. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. Pathology and Genetics: Head and Neck Tumours. Lyon: IARC Press; 2005. p. 210-11.  Back to cited text no. 1
    
2.
Spiro RH, Huvos AG, Strong EW. Adenoid cystic carcinoma: Factors influencing survival. Am J Clin Pathol 1979;138: 579-83.  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 Pathol Oral Cir Bucal 2008;13:E582-8.  Back to cited text no. 3
    
4.
Regezzi JA, Sciubba JJ, Jordan RC. Oral Pathology Clinical Pathologic Correlations. 5th ed. Delhi, India: Saunders Elsevier Inc.; 2009.  Back to cited text no. 4
    
5.
Neville BW, Damm DD, Allen CM, dan Bouquot JE. Oral and Maxillofacial Pathology. 2nd ed. Philadelphia: WB Saunders Company; 2002.  Back to cited text no. 5
    
6.
Huang M, Ma D, Sun K, Yu G, Guo C, Gao F. Factors influencing survival rate in adenoid cystic carcinoma of the salivary glands. Int J Oral Maxillofac Surg 1997;126:435-9.  Back to cited text no. 6
    
7.
Mitra S, Kundu S, Pattari SK, Ghosal AG. Metastatic pleural effusion: A rare presentation of salivary gland adenoid cystic carcinoma. Indian J Chest Dis Allied Sci 2011;53:107-10.  Back to cited text no. 7
    
8.
Martínez-Rodríguez N, Leco-Berrocal I, Rubio-Alonso L, Arias-Irimia O, Martínez-González JM. Epidemiology and treatment of adenoid cystic carcinoma of the minor salivary glands: A meta-analytic study. Med Oral Patol Oral Cir Bucal 2011;16:e884-9.  Back to cited text no. 8
    
9.
Rinaldo A, Shaha AR, Pellitteri PK, Bradley PJ, Ferlito A. Management of malignant sublingual salivary gland tumors. Oral Oncol 2004;40:2-5.  Back to cited text no. 9
    
10.
Andreadis D, Epivatianos A, Poulopoulos A, Nomikos A, Papazoglou G, Antoniades D, et al. Detection of C-KIT (CD117) molecule in benign and malignant salivary gland tumors. Oral Oncol 2006;42:57-65.  Back to cited text no. 10
    
11.
Tariq H, Anjum S, Din HU, Akhtar F. Diagnostic utility of C-kit protein (CD117) expression in differentiating adenoid cystic carcinoma and polymorphous low-grade Adenocarcinoma. Pak J Med Sci 2017;33:1376-80.  Back to cited text no. 11
    
12.
Soares EC, Carreiro Filho FP, Costa FW, Vieira AC, Alves AP. Adenoid cystic carcinoma of the tongue: Case report and literature review. Med Oral Patol Oral Cir Bucal 2008;13:e475-8.  Back to cited text no. 12
    
13.
Tewari A, Padma S, Sundaram PS. Detection of atypical metastases in recurrent adenoid cystic carcinoma of the parotid gland. J Can Res Ther 2013;9:148-50.  Back to cited text no. 13
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14.
Ouyang D, Liang L, Zheng G, Ke Z, Weng D, Yang W, et al. Risk factors and prognosis for salivary gland adenoid cystic carcinoma in Southern China: A 25-year retrospective study. Medicine 2017;96:e5964.  Back to cited text no. 14
    
15.
Park S, Nam SJ, Keam B, Kim TM, Jeon YK, Lee SH, et al. Overexpression in predicting poor overall survival in adenoid cystic carcinoma Cancer Res Treat 2016;48:518-26.  Back to cited text no. 15
    
16.
Jang S, Patel PN, Kimple RJ, Mcculloch TM. Clinical outcomes and prognostic factors of adenoid cystic carcinoma of the head and neck. Anticancer Res 2017;37:3045-52.  Back to cited text no. 16
    

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Correspondence Address:
Dr. C V Divyambika
Department of Oral Medicine and Radiology, Faculty of Dental Sciences, Sri Ramachandra Institute of Higher Education and Research (DU), Chennai - 600 116, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.IJDR_853_18

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]



 

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