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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 10  |  Issue : 3  |  Page : 158-160

Adenoid cystic carcinoma of palate: A rare case report


1 Department of Oral Pathology and Oral Microbiology, Index Institute of Dental Sciences, Indore, Madhya Pradesh, India
2 Department of Dentistry, B.K.L. Walawalkar Rural Medical College and Hospital, Ratnagiri, Maharashtra, India
3 Department of Pedodontics and Preventive Dentistry, Government College of Dentistry, Index Institute of Dental Sciences, Indore, Madhya Pradesh, India
4 Department of Pharmacology, ICARE Institute of Medical Sciences and Research, Haldia, West Bengal, India
5 Department of Dentistry, Darbhanga Medical College and Hospital, Laheriasarai, Bihar, India

Date of Submission06-Apr-2019
Date of Acceptance26-Jun-2019
Date of Web Publication15-Oct-2019

Correspondence Address:
Dr. Sukanta Sen
Department of Pharmacology, ICARE Institute of Medical Sciences and Research, Banbishnupur, Purba Medinipur, Haldia - 721 645, West Bengal
India
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DOI: 10.4103/srmjrds.srmjrds_29_19

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  Abstract 

A 40-year-old female patient reported to the department with the complaint of a single growth and pain in the upper right back tooth region for 1 year. Intraoral examination revealed a single unilateral exophytic growth seen on the right upper pterygomandibular raphe region. Extraoral examination revealed no facial asymmetry and no evidence of lymphadenopathy. Histopathological examination revealed a hyperparakeratotic stratified squamous epithelium of variable thickness with broad and flattened rete ridges. Lesional tissue showed large sheets of cells surrounded by condensed collagen component. The lesional cells were basaloid in appearance with hyperchromatic nucleus predominately arranged in a cribriform pattern along with eosinophilic material. No evidence of perineural invasion was observed in serial sections. On the basis of these findings, the diagnosis of adenoid cystic carcinoma was established.

Keywords: Adenoid cystic carcinoma, palate, salivary gland


How to cite this article:
Kumar P, Wanjari S, Sukhija H, Agarwal A, Sen S, Kolay SK. Adenoid cystic carcinoma of palate: A rare case report. SRM J Res Dent Sci 2019;10:158-60

How to cite this URL:
Kumar P, Wanjari S, Sukhija H, Agarwal A, Sen S, Kolay SK. Adenoid cystic carcinoma of palate: A rare case report. SRM J Res Dent Sci [serial online] 2019 [cited 2019 Nov 23];10:158-60. Available from: http://www.srmjrds.in/text.asp?2019/10/3/158/269212


  Introduction Top


Adenoid cystic carcinoma (ACC) is a malignant salivary gland tumor which was first described by Theodar Bilroth in 1856 as “cylindroma.” In 1954, this carcinoma was christened by Ewing (Foote and Frazel) as ACC. 22% of all salivary gland malignancies have been accounted as ACC. It is one of the most common malignant tumors among minor salivary glands and seromucous glands.[1] This carcinoma may often be deceptive clinically due to its small size as well as its pattern to be slow growing. ACC has been noticed to have a marked tendency to metastasis early, thereby making the prognosis usually questionable. This is a slow-growing tumor although it is considered to be an aggressive neoplasm which has been observed to be characterized by a remarkable recurrence capacity.


  Case Report Top


A female patient aged 40 years reported to the department with the complaint of a single growth and pain in the upper right back tooth region for 1 year. The patient reported intermittent and throbbing pain in the region of the growth. The patient did not report any aggravating or relieving factor for the pain. The patient did not have any significant medical history, and there was no history of fever and unexplained weight loss since the observation of the growth by the patient. Intraoral examination revealed a single unilateral exophytic growth on the right upper pterygomandibular raphe region approximately measuring 0.5 cm × 0.5 cm and appearing oval shaped. It appeared red in color [Figure 1]. Palpation revealed a well-defined exophytic growth with rough surface. The lesion elicited tenderness on palpation and did not bleed spontaneously or on palpation.
Figure 1: Adenoid cystic carcinoma of palate (single unilateral exophytic growth seen on the right upper pterygomandibular raphe region)

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Extraoral examination revealed no facial asymmetry and no evidence of lymphadenopathy. Panoramic radiography showed no evidence of bone changes in both the jaws. These features were suggestive clinically of squamous papilloma. The lesion was surgically excised and was sent to the department for histopathological examination. Histopathological examination revealed a hyperparakeratotic stratified squamous epithelium of variable thickness with broad and flattened rete ridges [Figure 2]. Lesional tissue showed large sheets of cells surrounded by condensed collagen component [Figure 3]. The lesional cells were basaloid in appearance with hyperchromatic nucleus. This appeared as predominately arranged in cribriform pattern along with eosinophilic material [Figure 4]. No evidence of perineural invasion was observed in serial sections. On the basis of the aforementioned findings, ACC was diagnosed.
Figure 2: Histopathological examination revealed a hyperparakeratotic stratified squamous epithelium of variable thickness with broad and flattened rete ridges

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Figure 3: Lesional tissue showing large sheets of cells surrounded by condensed collagen component

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Figure 4: (a and b) The lesional cells are basaloid in appearance with hyperchromatic nucleus predominately arranged in cribriform pattern along with eosinophilic material

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  Discussion Top


ACC is a rare tumor of epithelial origin displaying slow yet persistent growth pattern. The World Health Organization (WHO) defines ACC as a “basaloid tumor consisting of epithelial and myoepithelial cells in various morphological configurations including tubular, cribriform, and solid patterns.”[2] The most frequent subtype has been stated to be the cribriform pattern. This pattern histopathologically is seen to be composed of basaloid cell islands surrounding cyst-like spaces of variable sizes displaying the Swiss cheese- or sieve-like pattern. These cyst-like spaces are known as pseudocysts since they do not show true glandular lamina. It usually has been reported to have fatal outcome.

The clinical presentation of ACC involves a slow-growing, firm, and unilocular mass.[3] The patient usually complains of pain as an important associated symptom which may occasionally develop before clinical evidence of the malignancy. The case presented shows small exophytic growth with throbbing intermittent pain.

Histopathologically, ACC is known to be of three variants, namely the solid, tubular, and cribriform types. ACC grading is done based on the histological patterns as Grade I to be the combination of cribriform and tubular types, Grade II to be a combination of tubular, solid, and cribriform patterns, and Grade III as showing only the solid pattern.[4],[5] The presenting case reported was of cribriform pattern and classified as Grade I. The mitotic figures are generally less in the cribriform pattern.[3],[6] Prognosis of ACC depends on the presence of perineural invasion, tumor grade, and tumor site. Cribriform and tubular types have been reported to have better prognosis than the solid pattern.[7],[8]

Due to rarity of this lesion, it mostly presents as a diagnostic as well as treatment challenge. Most literature available with regard to ACC is based on a small number of cases published, and therefore, there is a requirement for more publication of information related to its clinical behavior, its various treatment modalities, and its prognosis. ACC has been reported to occur at any age though, in most of the cases, the age of the patients has been observed to range from 24 to 78 years.[9] The palate has been reported to be the most commonly involved site among the minor salivary glands affected. Among the malignant tumors of the minor salivary glands, mucoepidermoid carcinoma has been reported to be the most common which has been observed to be followed by polymorphous low-grade adenocarcinoma, and the third most common neoplasm was ACC.[9],[10] ACC affecting the minor salivary glands has been reported to have the worst prognosis compared to ACC of the major salivary glands.[9],[10],[11]

Pain is a common clinical feature in ACC due to perineural invasion.[12] Perineural invasion may occur due to spread of the neoplasia along perineural spaces or due to within the nerve itself.[13] The presenting case did not show any metastasis although literature reveals metastasis to lungs, bone, liver, and brain.[14] According to the WHO, influence of perineural invasion on the survival of the patient needs further confirmation. This was mentioned to have no prognostic significance in few studies. However, some authors stated that ACC has a poor prognosis because of its greater tendency toward distantly metastasizing.[11],[12],[13] Grade III ACC shows long-term low survival. According to some authors, distant metastasis has been seen to occur in 25%–50% of the cases with the most common site of metastasis to be the lungs. Authors have stated that the 5-year survival rate after treatment has been seen to be 75%, but the long-term survival rate has been seen to be relatively lower.[9]


  Conclusion Top


ACC is a rare salivary gland malignancy. ACC displays unique histopathological features and its tendency toward perineural invasion. Careful assessment of tumor grading, perineural invasion, and clinical margin status continue to be important criteria for prognosis. Long-term follow-up of cases at regular intervals is advised and mandatory.

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.
Jaso J, Malhotra R. Adenoid cystic carcinoma. Arch Pathol Lab Med 2011;135:511-5.  Back to cited text no. 1
    
2.
Ajila V, Hegde S, Nair GR, Babu SG. Adenoid cystic carcinoma of the buccal mucosa: A case report and review of the literature. Dent Res J (Isfahan) 2012;9:642-6.  Back to cited text no. 2
    
3.
Gondivkar SM, Gadbail AR, Chole R, Parikh RV. Adenoid cystic carcinoma: A rare clinical entity and literature review. Oral Oncol 2011;47:231-6.  Back to cited text no. 3
    
4.
Mahajan A, Kulkarni M, Parekh M, Khan M, Shah A, Gabhane M. Adenoid cystic carcinoma of hard palate: A case report. Oral Maxillofac Pathol J 2011;2:127-31.  Back to cited text no. 4
    
5.
Naik KL, Shetty P, Hegde P. Adenoid cystic carcinoma of buccal mucosa with extensive hyalinization: A unique case report. Ann Trop Med Public Health 2013;6:571-4.  Back to cited text no. 5
  [Full text]  
6.
Kumar AN, Harish M, Alavi YA, Mallikarjuna R. Adenoid cystic carcinoma of buccal mucosa. BMJ Case Rep 2013;2013. pii: bcr2013009770.  Back to cited text no. 6
    
7.
Huang M, Ma D, Sun K, Yu G, Guo C, Gao F, et al. Factors influencing survival rate in adenoid cystic carcinoma of the salivary glands. Int J Oral Maxillofac Surg 1997;26:435-9.  Back to cited text no. 7
    
8.
Khan AJ, DiGiovanna MP, Ross DA, Sasaki CT, Carter D, Son YH, et al. Adenoid cystic carcinoma: A retrospective clinical review. Int J Cancer 2001;96:149-58.  Back to cited text no. 8
    
9.
Chundru NS, Amudala R, Thankappan P, Nagaraju CD. Adenoid cystic carcinoma of palate: A case report and review of literature. Dent Res J (Isfahan) 2013;10:274-8.  Back to cited text no. 9
    
10.
Neville BW, Damm D, Allen CM, Bouquot JE. Salivary gland pathology. In: Neville, editor. Oral and Maxillofacial Pathology. 3rd ed. Missouri: Saunders; 2009. p. 495-7.  Back to cited text no. 10
    
11.
Buchner A, Merrell PW, Carpenter WM. Relative frequency of intra-oral minor salivary gland tumors: A study of 380 cases from Northern California and comparison to reports from other parts of the world. J Oral Pathol Med 2007;36:207-14.  Back to cited text no. 11
    
12.
Hashemi Pour MS, Zarei MR, Chamani G, Rad M. Malignant salivary glands tumors in Kerman province: A retrospective study. Dent Res J 2007;4:4-10.  Back to cited text no. 12
    
13.
Nascimento AG, Amaral AL, Prado LA, Kligerman J, Silveira TR. Adenoid cystic carcinoma of salivary glands. A study of 61 cases with clinicopathologic correlation. Cancer 1986;57:312-9.  Back to cited text no. 13
    
14.
Dal Maso M, Lippi L. Adenoid cystic carcinoma of the head and neck: A clinical study of 37 cases. Laryngoscope 1985;95:177-81.  Back to cited text no. 14
    


    Figures

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



 

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