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CASE REPORT Table of Contents   
Year : 2005  |  Volume : 16  |  Issue : 4  |  Page : 167-70
Salivary duct carcinoma of the palate


1 Oral and Maxillofacial pathology, TN Gov't. Dental College and Hospital, Chennai, India
2 Oral Medicine and Radiology, TN Gov't. Dental College and Hospital, Chennai, India
3 Oral and Maxillofacial Surgery, TN Gov't. Dental College and Hospital, Chennai, India
4 TN Gov't. Dental College and Hospital, Chennai, India
5 Oral and Marillo facial Pathology, TN Gov't. Dental College and Hospital, Chennai, India

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   Abstract 

Salivary duct carcinoma is a rare high-grade neoplasm that more frequently affects the parotid gland. Though neoplasms of this type are infrequent in minor salivary glands, they are less aggressive and may lead to early diagnosis before distant metastases could occur. Salivary duct carcinoma is also the most frequent epithelial component of carcinosarcoma. The present article reports a case of SDC of the palate in a 26-year-old male and discusses SDC as a malignant epithelial component in carcinosarcoma.

Keywords: Salivary duct carcinoma, minor salivary glands; comedonecrosis; carcinosarcoma

How to cite this article:
Ponniah I, Murali G M, SureshKumar P, Kumaran M G, Shaheen A. Salivary duct carcinoma of the palate. Indian J Dent Res 2005;16:167

How to cite this URL:
Ponniah I, Murali G M, SureshKumar P, Kumaran M G, Shaheen A. Salivary duct carcinoma of the palate. Indian J Dent Res [serial online] 2005 [cited 2017 Jul 27];16:167. Available from: http://www.ijdr.in/text.asp?2005/16/4/167/29902

   Introduction Top


Salivary duct carcinoma (SDC) is a rare, typically high-grade malignant epithelial neoplasm composed of structures that resemble expanded salivary gland ducts. Kleinsasser and Klein first applied the term in 1968 [1] to describe a malignant neoplasm of salivary gland having resemblance to the ductal carcinoma of breast. Other terms include infiltrating salivary duct carcinoma[2], cribriform salivary carcinoma of excretory duct [3]. and low grade salivary duct carcinoma [4]. Recently, a variant of SDC sarcomatoid salivary duct carcinoma has been proposed [5]. Over 80 % of the tumors are found in the parotid gland more commonly in elderly men, with peak in the sixth decade. [2],[3], [6],[7],[8],[9],[10] Although it was first described over three decades ago, reports of SDC involving minor salivary glands of the oral cavity is rare, with only fewer than 20 such tumors described in the literature [1],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20],[21]. We report a case of SDC involving the palate in a 26-year­old male. In addition, we discuss SDC as a malignant epithelial component in carcino sarcoma.


   Case report Top


In January 2001, a 26-year-old male patient presented with the chief complaint of swelling in the roof of his mouth on the right side for the past 1 year. History revealed that the swelling started as a small one and gradually progressed to the present size. On his account, the swelling was painless initially and reported that he developed pain only in the last few weeks. He stated that the pain was localized, continuous and gnawing in nature. There was no history of nasal obstruction or discharge. His past medical and dental history was unremarkable.

On physical examination, there was no palpable lymph node. hitra orally, a single lobulated swelling was noted on the right side of the hard palate, measuring 3 cm x 2cm in size, irregular in shape. The lesion extended anteriorly to the 14 region, posteriorly to the junction of hard and soft palate and medially crossing the midline to the left side of the palate [Figure - 1] a. The mucosa over the swelling appeared smooth but showed slight bluish tinge with a bluish black discoloration on the posterior aspect. Few engorged blood vessels were also seen over the swelling.

On palpation, the lesion was firm, tender and slightly compressible. Aspiration was attempted but did not yield any positive results. Occlusal radiograph of maxilla showed widening of periodontal ligament space in relation to 11, which also showed extrusion. There was also loss of trabecular pattern and ill-defined radiolucency indicating bone destruction [Figure - 1]b. The lesion was provisionally diagnosed as an adenoma of the palate. An incisional biopsy was performed and subjected to histopathological examination.

Histopathological examination showed beneath a non-dysplastic surface epithelium, variably sized circumscribed nests of dysplastic epithelium with prominent central comedonecrosis and intraductal papillary projections [Figure - 2]a and b. The nests of epithelium also exhibited solid and cribriform growth patterns [Figure - 2]c. The stroma was predominantly fibro vascular and characteristically showed vascular invasion in many regions of the section [Figure - 2]d. The histomorphology of the lesion was consistent with salivary duct carcinoma.

The lesion was excised completely free of margins, as observed by the study of surgical specimen. There was no evidence of local recurrence or distant metastasis during the 4 years of follow-up.


   Discussion Top


Review of the literature showed that SDC of the oral cavity is more common in the palate [1],[13],[16],[17], [19],[20],[21]and is similar to its counterpart in the parotid gland with respect to age and gender. Most of the reported cases occurred in patients over 50 yeas of age and is uncommon before the third decade, only one case had been reported previously in a 23 year old patient [19], ours being the second case to the best of our knowledge.

SDC is considered to be a high-grade carcinoma with dismal short term prognosis [3],[6] death eventually occurring within the first 3 years after diagnosis [3],[7],[21],[22]. In contrast, others believe that there exist different sub types, one of which is characteristically aggressive, whereas the other less invasive purely intraductal, perhaps representing pre-invasive phase of high-grade SDC [4],[10],[1]. SDC is characterized microscopically by intraductal tumor cell islands with papillary, cribriform or solid configurations of cells, often with central comedonecrosis [15].

Combination of different patterns are frequently found in the same tumor [6],[10], as observed in our case. Perineural and vascular invasion as well as intralymphatic emboli may also be seen.

Delgado et al [4], suggested that cribriform growth pattern and comedonecrosis are more frequently encountered in high-grade than in low-grade SDC. Progression is evidenced by the acquisition of higher cytologic grades such as, monomorphous cellular composition, well­ defined cytoplasmic borders and rigid cellular arrangement [4], In our case, cribriform growth pattern and comedonecrosis were prominent and also higher cytologic grades were observed. However this lesion is found to be less aggressive similar to most other reports of SDC of oral cavity [1], [10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20].

To make a definitive diagnosis of SDC, Garland et al ,[8] require circumscribed epithelial nest with central comedonecrosis. Interestingly, in most cases of carcinosarcoma, a rare biphasic tumor consisting of carcinomatous and sarcomatous features [23] the malignant epithelial component is that of SDC.[24],[25],[26],[27] Furthermore, both SDC[5],[7],[9],[13],[22],[28] and carcinosarcoma may arise from pre-existing pleomorphic adenoma [23], [25],[26],[27]. Does the presence of residual pleomorphic areas have any significance to the prognosis? The fact is that the presence of pre-existent pleomorphic adenorna does not have a significant prognostic factor in SDC[7],[9]. Similarly, the degree of lethality is also not dependent on the histological type of carcinoma in carcinosarcoma[27].

Immunohistochemical findings in carcinosarcoma suggest that both epithelial and mesenchymal component have a common precursor cell, possibly myoepithelial cell with a spectrum of differentiation [23],[24],[25]. In contrast Alvarez-Canas and Rodilla [26] concluded that the malignant epithelial component resembling similar to SDC in carcinosarcoma, is composed entirely of ductal cells without evidence of myoepithelial differentiation.

Recently, sarcomatoid SDC has been described in an attempt to define those carcinosarcomas that manifest SDC microscopically, as a variant of SDC rather than carcinosarcoma [5],[28]. The concept of sarcomatoid SDC was based on the immunohistochemical expression pattern of epithelial specific markers [5],[28]. Further evidence for this concept is the ultra structural observation of interdigitating cytoplasmic process and scattered junctional complexes in addition to the lack of tonofilaments or intracellular lumina in the sarcomatoid component [5]. Therefore, it is likely that both carcinomatous and sarcornatous component share a common histogenetic pathway [28]. It is appropriate that the use of the term carcinosarcoma be restricted to those carcinosarcomas in which the malignant epithelial component does not resemble SDC.

In conclusion, although these terminologies for closely related neoplasm in terms of prognosis may generate academic interest, it is difficult to predict any difference in their biologic behaviour. It is important that more cases of this neoplasm should be reported to understand any significant biologic differences that would help to establish a definite conclusion about their nature. Until more is known about their nature, it is widely believed that both SDC and SDC in carcinosarcoma are rare high-grade carcinomas capable of distant metastases with a poorer outcome in major salivary glands[2],[3], [5],[6],[7], [24],[25],[27]. In contrast SDC of minor salivary gland origin arerelatively less aggressive and carries a better prognosis [1], [10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20].



 
   References Top

1.Kleinsasser O, Klein ILL Hubner G. Speichelgangcarcinome. Fin den Milchgancarcinomen der Brustdruse analoge Gruppe von Speicheldrusentumoren. Arch Klin Exp Ohren NasenKehlkopfheilkd,192:100-5, 1968.  Back to cited text no. 1    
2.Chen KT, Hafez GR. Infiltrating salivary duct carcinoma. A clinicopathologic study of five cases. Arch Otolaryngol, 107:37-9,1981.   Back to cited text no. 2    
3.Brandwein MS, Jagirdar J, Patil T, Biller H, Kaneko M. Salivary duct carcinoma [Cribriform Salivary Carcinoma of Excretory Ducts]. A clinicopathologic and immunohistochemical study of 12 cases. Cancer, 65:2307-2314,1990.   Back to cited text no. 3    
4.Delgado R, Klimstra D, Albores-Saavedra J. Low grade salivary duct carcinoma: A distinct variant with a low grade histology and a predominant intraductal growth pattern. Cancer, 78:958-67,1996.  Back to cited text no. 4    
5.Henley JD, Seo IS, Dayan D, Gnepp DR. Sarcomatoid Salivary duct carcinoma of the parotid gland. Than Pathol, 31:208-213,2000.   Back to cited text no. 5    
6.Ruiz CC, Romero MP, Perez MM. Salivary duct carcinoma: A report of nine cases. J Oral Maxillofac Surg, 51:641-46,1993.  Back to cited text no. 6    
7.Lewis JE, McKinneyBC, Weiland LIE Ferreiro JA, Olsen KD. Salivary duct carcinoma: clinicopathologic and inummohistochemical review of 26 cases. Cancer, 77:223-30,1996.   Back to cited text no. 7    
8.Garland TA, Innes DJ, Fechner RE. Salivary duct carcinoma: an analysis of four cases with review of literature. Am J Clin Pathol, 81:436-41,1984.  Back to cited text no. 8    
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11.Chen KTK. hitraductal carcinoma of the minor salivary gland. J Laryngol Otol, 97:189­191,1983.  Back to cited text no. 11    
12.Pesce C, Colacino R, Buffa P. Duct carcinoma of the minor salivary glands: A case report. J Laryngol Oto1,100:611-13,1986.  Back to cited text no. 12    
13.Delgado R, Vuitch F, Albores-Saavedra J. Salivary duct carcinoma. Cancer, 72;1503-12, 1993.  Back to cited text no. 13    
14.Watatani K, Shirasuna K, AikawaT, Matsuya T. Intracluctal carcinoma of the tongue. Report of a case. hit J Oral Maxillofac Slug, 20:175-76, 1991.  Back to cited text no. 14    
15.Yoshimura Y, Tawara K, Yoshigi J, Nagaoka S. Concomitant salivary duct carcinoma of a minor buccal salivary gland and papillary cystadenoma lymphomatusum of a cervical lymph node: Report of a case and review of the literature. J Oral Maxillofac Surg, 53:448-53,1995.   Back to cited text no. 15    
16.TatemotoY, Ohno A, Osaki T. Low malignant intraductal Carcinoma on the hard palate: a variant of salivary duct carcinoma 7EurJcancer B Oral Oncol, 32(PtB):275-277,1996.  Back to cited text no. 16    
17.Epivatianos A, Dimitrakopoulos J, 'Trigonidis G. Intra oral salivary duct carcinoma: a clinicopathological study of four cases and review of the literature. Ann Dent, 54:36­40,1995.  Back to cited text no. 17    
18.Cheuk W, Miliauskas JR, Chan JKC. Intraductal carcinoma of the oral cavity: a case report and a reappraisal of the concept of pure ductal carcinoma in situ in salivary duct cacinoma. Am J Surg Pathol, 28:266-70, 2004.   Back to cited text no. 18    
19.Huh KH, Heo MS, Lee SS, Choi SC. Three new cases of salivary duct carcinoma in the palate: a radiologic investigation and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod,95:752-60, 2003.  Back to cited text no. 19    
20.Ide F, Mishima K, Saito I. Circumscribed salivary duct carcinoma of the palate: a non­threatening variant. Histopathol, 45:89­91,2004.  Back to cited text no. 20    
21.Lopes MA, Fabio de Ab reu A, Levy BA, Paes de Almedia O, Kowalski LP. Intraoral salivary duct carcinoma: case report with immunobisto chemical observations. Oral Surg Oral Med Oral Pathol Oral Radiol Endod.2991; 91:689-92.   Back to cited text no. 21    
22.Nagao T, Gaffey TA, Visscher DW, Kay PA, Minato H, Serizawa H, et al . Invasive micropapillary salivary duct carcinoma: a distinct histologic variant with biologic significance. Am J Surg Patho1,28:319-26, 2004.   Back to cited text no. 22    
23.Ford AC, Petruzzelli GL McClatchey KD, Candel AG. Carcinosarcoma of the parotid gland: Report of a case with parapharyngeal presentation. Am J Otolaryngol, 21:136­140, 2000.  Back to cited text no. 23    
24.Carson HL Tojo DP, Chow JM, Hammadeh R, Raslan WE. Carcinosarcoma of salivary glands with unusual stromal components. Report of two cases and review of the literature. Oral Sung Oral Med Oral Pathol Oral Radiol Endod, 79:738-46,1995.  Back to cited text no. 24    
25.Pang PCW, To EWH, Tsang WM, Liu TL. Carcinosarcoma (Malignant mixed tumor) of the parotid gland: A case report. J Oral MaxillofacSurg, 59:583-87,2001.   Back to cited text no. 25    
26.Alvarez-Canal C, Rodilla IG. True malignant mixed tumor (Carcinosarcoma) of the parotid gland. Report of a case with immunohistochemical study. Oral Surg Oral Med oral Pathol Oral Radiol Endod, 81:454-58,1996.  Back to cited text no. 26    
27.Stephen J, Batsakis JG, Luna MA, HeydenUVD,Byers, RM.True malignant mixed tumours (Carcinosarcoma) of the salivary glands. Oral Surg Oral med Oral Pathol, 61:597-602,1986.  Back to cited text no. 27    
28.Nagao T, Gaffey TA, Serizawa H, Iwaya K, Watanabe A, Yoshida T, et al . Sarcomatoid variant of salivary duct carcinoma: clinicopathologic and immunohistochemical study of eight cases with review of the literature. Am J Clin Patho1,122:222-31, 2004.  Back to cited text no. 28    

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Correspondence Address:
I Ponniah
TN Gov't. Dental College and Hospital, Chennai
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.29902

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[Figure - 1], [Figure - 2]

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