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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
CASE REPORT Table of Contents   
Year : 2004  |  Volume : 8  |  Issue : 2  |  Page : 87-90
 

Salivary duct carcinoma of minor salivary gland


1 Department of Oral Pathology, Ragas Dental College & Hospital, Chennai, India
2 Department of Oral Surgery, Ragas Dental College & Hospital, Chennai, India

Correspondence Address:
S Nalin Kumar
Department of Oral Pathology, Ragas Dental College and Hospital, #2/102, East Coast Road, Uthandi, Chennai - 600119
India
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Source of Support: None, Conflict of Interest: None


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   Abstract 

Salivary duct carcinoma (SDC) is an uncommon aggressive malignancy seen predominantly in the parotid gland. Intra oral cases of SDC arising from minor salivary glands are rare and only a few have been reported in literature. This malignancy is well known for its cestructive behaviour, metastasis to distant sites and high mortality rate. In this article, we present a case of salivary duct carcinoma of palate of a 44-year-old male patient.


Keywords: Salivary duct carcinoma, minor glands, immuno-histochemistry


How to cite this article:
Nalin Kumar S, Ranganathan K, Nathan J A, Saraswathi T R. Salivary duct carcinoma of minor salivary gland. J Oral Maxillofac Pathol 2004;8:87-90

How to cite this URL:
Nalin Kumar S, Ranganathan K, Nathan J A, Saraswathi T R. Salivary duct carcinoma of minor salivary gland. J Oral Maxillofac Pathol [serial online] 2004 [cited 2020 Mar 29];8:87-90. Available from: http://www.jomfp.in/text.asp?2004/8/2/87/40973



   Introduction Top


Salivary duct carcinoma( SDC) is a rare. invasive malignancy arising in the ductal epithelium of salivary glands [1] . It is usually seen in the major salivary glands especially the parotid gland [2] . It was established as a distinct clinicopathologic entity and delineated from ''adenocarcinoma(NOS)" in 1991 by the World Health Organization [1],[4] .Salivary duct carcinoma affecting the minor salivary glands has been reported in only 4% of SDC cases and constitutes for 2%, of all salivary gland malignant neoplasms [1],[2],[3] . In this article, we report a case of salivary gland carcinoma present in the palate of a 44-year-old male patient.


   Case Report Top


A 44-year-old male patient was referred with a complaint of pain in the left upper posterior region of maxilla for the past 2 months. The patient had a history of cigarette smoking fix the past 20 years. He was not suffering from any systemic illness and there were no abnormalities detected on extra oral examination. On until oral examination, an ulcerated lesion was seen in the left side of the hard palate extending from the premolar to third molar region [Figure 1]. The patient gave a history of duration of the lesion as six months, the lesion initially being a small swelling with rapid growth and ulceration eventually. The lesion was 3.5 x 2.5 cm in size. crythematous with irregular margins and indurated. rolled out borders. On palpation. the lesion was tender and soft to Jinn in consistency. On lymph node examination, there were no lymph nodes detected. A provisonal diagnosis of squamous cell carcinoma/ mucoepidermoid carcinoma was given based on the clinical details. An incisional biopsy was performed under local anaesthesia and the specimen was sent for histopathological evaluation.

The histopathologic features of the lesion revealed numerous infiltrating islands and cords of neoplastic glandular epithelium in a fibrovascular connective tissue [Figure 2]. Most of the neoplastic islands exhibited central "comedonecrosis [Figure 3]. The neoplastic cells were cuboidal to polygonal in shape and exhibited cellular pleomorphism, eosinophilic cytoplasm, nuclear hypercchromtism and prominent nucleoli [Figure 4]. Numerous blood vessels and mitoses were present. The lesion was covered by a parakeratinized stratifieds squamous surface epithelium. Based on the histopathologic features a diagnosis, of salivary duct carcinoma was made.


   Discussion Top


Salivary duct carcinoma (SDC) was First described by Kleinsasser el al in I968, who reported a histopathologic similarity to ductal carcinoma of breast [1] . Other terms used for SDC are cribriform salivary carcinoma of excretory ducts; intraductal carcinoina and infiltrating salivary duct carcinoma [2],[5]. The term salivary duct carcinoma is considered to be inaccurate as other salivary . tumors are also ofcxcretory or terminal duct origin [6] .

A review of the existing literature on SDC revealed the rarity of this lesion in minor salivary glands. Only 28 cases of SDC have been reported in minor salivary glands. Kung-HoE Hub et al have published a review of 20 cases of minor salivary gland SDC [1] . In addition to this, othercase reports by can Heerden et al (5 cases) [5] . Lopes MA et al (I case) [2] . Takeda et al (I case) [7] and Urban et al (I case) [8] were found in English literature.

SDC occurs predominantly in males with a male to female ratio of 2:1.The age of these patients ranged front 23-80 with more cases being reported in the fifth and sixth decides [1],[2],[4],[5],[6] . Intra-orally, the common sites of occurrence are the palate, followed by the buccal mucosa /vestibule, upper lip, maxilla and mandible [1] . It usually presents as a painful or painless, firm mass of rapid growth. Facial nerve palsy was associated in few cases of SDC affecting the parotid gland [6] . Based on the clinical features, the differential diagnoses for this lesion are mucoepidemtoid carcinoma. adenoid cystic carcinoma and stluamous cell carcinoma [1] .

Hisiopathologic features of SDC consist of atypical cuhoidal or polygonal cells arranged in papillary cribriform and solid growth patterns alone with duct like structures [1] . Tumor cells are polygons in shape with granular eosinophilic cytoplasm. enlarged hyperchormatic. pleomorphic nuclei and prominent nucleoli [1],[4] . pseudocyst formation and central comedonecrosis are seen in the neoplastic islands [11] Vascular invasion anal perineural infiltration have been reported in some cases [1],[9] . Atypical mitotic figures are seen in most of the lesions [2] . In sonic cases, hands of epithelium arch over luminal space, to create a' roman bridge" appearance [6] . Dystrophic calcifications were seen in some cases [9] .

Histopathologically, the differential diagnoses for SDC are mucoepidennoid carcinoma, nncocytie carcinoma, acinic cell carcinoma, polymorphous low-grade carcinoma [4] . adenoid cystic carcinoma and adenocarcinoma (not otherwise specified) [5]. SDC can be distinguished from the above lesions by histopathologic features like comedonecrosis. cosinophilic cytoplasm of atypical cells and cellular plenmorphism [2],[4]. Metastases from breast and prostrate gland should be ruled out before giving a final diagnoses ofSDC [5] .

SDC can occur de novo or as the malignant component in carcinoma ex pleomorphic adenoma [5],[6] and also as a hybrid tumour in association with squamous cell carcinoma [7] . adenoid cystic carcinoma [5] and warthins tumour [11] . SDC of minor salivary gland of maxilla involving the cavernous sinus has been reported as a result of retrograde vascular tumour seeding [8] .

SDC is considered to he a high-grade malignancy as it is aggressive and invasive with a tendency for regional and distant metastasis [1],[2] . Metastasis of SDC to lungs, bones and liver has been reported with a high mortality rate [6] . However, a low-grade variant of SDC has also been described in literature [5],[12] . In another report by Delgado et al, 10 cases were described as low-grade SDC characterized by intraductal growth pattern, less aggressive cytomorphology and lack of comedonecrosis [13] .

Ultrastructural studies of SDC showed the presence of cuboidal or polygonal cells forming duct like structures with microvilli and apical vesicles. Myoepithelial cells were not present. The ultrastructural features of SDC exhibited similarities to that of the excretory duct [9],[14] .

Immunohistochemical analysis ofSDC shows a positive reaction of the tumour for cytokeraiin and epithelial membrane antigen (EMA). SDC is positive for CK7 and CK8 [14] . C K 14 is useful in demonstrating in situ ductal growth pattern in low-grade variant of SDC [14] . PCNA. Ki-67, p53, c-erhB-2 and cathepsin-D [10] immunopositivity have been reported in some cases of SDC [2],[6] . SDC is negative for S-I00, Estrogen receptor, Progesterone receptor [4],[10] , vimentin, smooth muscle actin [2],[5],[15] and GFAP [6] . Androgen receptor (AR) is strongly expressed in SDC [16] .

Androgen receptor (AR) mediates the effect ofandrogen by binding to specific DNA sequences, which influence transcription and translation of' various genes. It is suggested that androgen may play a role in the pathogenesis of' SDC Through the mediation of an epidermal growth factor and transforming growth Iaclor­ a pathway. Experimental studies have shown that androgen influences the expression of proto-oncogenes like c-myc and bcI-2 in lacrimal and salivary gland tissues [16] .

Lymph node metastases have reported in 22% of SDC cases in minor salivary glands as compared to 83% in SDC of major salivary glands [1] . However, Multiple metastases and high mortality rate of 60-75% are associated with minor salivary gland SDC [1],[6] . Recurrence rate ol'33-35%, has been reported in a study of SDC patients [2],[6] .

Treatment of this lesion involves radical surgical excision of lesional tissue and associated structures with concomitant neck dissection, followed by post-operative radiation therapy [2],[4],[9] . Hormonal therapy using an anti-androgen like goserelin has been tried out which resulted in partial remission of' the salivary duct carcinoma. Given the hypothesis of hormonal involvement in the pathogenesis of this lesion, further studies on anti-androgen therapy needs to be carried out on a larger sample size [16] .

 
   References Top

1.Kyung-Hoe Huh. Min-Suk Hoe. Sam-Sun Lee et al. Three new cases of salivary duct carcinoma in the palate: A radiologic investigation and review of literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endol 2003:95: 752-60.  Back to cited text no. 1    
2.Lopes MA. de Abreu Alves F. Levy BA et al. Infra-oral salivary duct carcinoma: Case report with immunohistochemical observations. Oral Surg Oral Mcd Oral Pathol Oral Radiol Endol 2001:91: 689-92.  Back to cited text no. 2    
3.Lopes MA. Kowalski LP, Almeida OP et al. C'linicopathologic study of 196 intraoral minor salivary gland tumours. J Oral Pathol Mcd 1999;28(6):264-7.  Back to cited text no. 3    
4.'Toyoda II, Yamaguchi K, Miura K et al. Salivary duct carcinoma in the submandibular region. Int_ J. Oral. Maxillofac. Surg 2003; 32: 427-9.  Back to cited text no. 4    
5.Van Heerden WFP, Raubenheimer EJ, Swart TJP et al. Intraoral salivary duct carcinoma: A report of 5 cases. J. Oral. Maxillofac_ Surg 2003; 61:126-131.  Back to cited text no. 5    
6.Madrigal B, Garcia J and do Vicente JC Salivary duct carcinoma: An unusual case of long-term evolution. Oral Surg Oral Med Oral Pathol Oral Radiol Endol 1999; 88: 597-602.  Back to cited text no. 6    
7.Takeda Y. Yamamoto H. Oral collision carcinoma: salivary duct carcinoma of minor salivary gland origin and squamous cell carcinoma of oral mucosa. J. Oral Sci 1999; 41: 129.  Back to cited text no. 7    
8.Urban SD, Hall JM, Bentkover SH et al. Salivary ducts carcinoma of minor salivary gland origin: Report of a case involving the cavernous sinus. J.Oral. Maxillofac. Surg2002; 60:958-962.  Back to cited text no. 8    
9.Barnes L, Rao U, Krause J et al. Salivary duct carcinoma. Part I Clinicopathologic evaluation and DNA image analysis of 13 cases with review of literature. Oral Sung Oral Med Oral Pathol 1994: 78: 64-73.  Back to cited text no. 9    
10.Barnes I., Rao U. Krause ,1 et al. Salivary duct carcinoma. Part II Immunohistochemical evaluation of 13 cases fir estrogen and progesterone receptors. cathepsin D and c­erbB-2 protein. Oral Surg Oral Med Oral Pathol 1994;78:74-80.  Back to cited text no. 10    
11.Yoshiniura Y, Tawara K, Yoshigi J et al. Concomitant salivary duct carcinoma of minor salivary gland and papillary cystadenoma lymphomatosum of a cervical lymph node: Report of a case and review of the literature. J. Oral. Maxillofac. Surg 1995:53:448-453.  Back to cited text no. 11    
12.Tatemoto Y, Ohno A and Osaki T. Low malignant intraductal carcinoma on the hard palate: A variant of salivary duct carcinoma'? Oral oncol, EurJ cancer 1996 - ,32B: 275-277.  Back to cited text no. 12    
13.Delgado R, Klimstra I), Albores-Sayeedra J. Low grade salivary duct carcinoma. A distinct variant with low-grade histology and predominant intraductal growth pattern. Cancer 1996; 78: 958-67.  Back to cited text no. 13    
14.Dc Araujo VC, Loducca SVI., Sobral APV et al. Salivary duct carcinoma: cytokcratin 14 as a marker of in-situ intraductal growth. Histopathology 2002:41:244-2-19,  Back to cited text no. 14    
15.Suruki H, Hashimoto K. Salivary duell carcinoma in the mandible: Report of a case with immunohistochemical studies. Br J Oral Maxillofac Surg 199 1 37: 67-69.  Back to cited text no. 15    
16.Nasser SMf, Faquin WC and Dayal Y. Expression of androgen. estrogen and progesterone receptors in salivary gland tulnollm Frequent expression of androgen receptor in a subset of malignant salivary gland tumours. Am J C lin pathol 2003; 119: 801-6.  Back to cited text no. 16    


    Figures

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



 

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    Abstract
    Introduction
    Case Report
    Discussion
    References
    Article Figures

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