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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 7  |  Issue : 1  |  Page : 32-34

Salivary duct carcinoma of tongue: A rare entity


1 Consultant, Head Neck, Reconstructive and Robotic Services, Reconstructive and Robotic Services HCG Cancer Centre, Ahmedabad, India
2 Clinical Fellow, Department of Head and Neck, Reconstructive and Robotic Services HCG Cancer Centre, Ahmedabad, India
3 Head, Department of Histopathology, Reconstructive and Robotic Services HCG Cancer Centre, Ahmedabad, India
4 Head and Consultant, Head Neck, Reconstructive and Robotic Services HCG Cancer Centre, Ahmedabad, India

Date of Web Publication26-Jul-2019

Correspondence Address:
Dushyant S Mandlik
Room No. 205, OPD Building, HCG Cancer Centre, Science City, Ahmedabad - 380 060, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jhnps.jhnps_24_19

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  Abstract 


Salivary duct carcinomas (SDCs) occur almost exclusively in the major salivary glands. The parotid gland is most commonly affected. SDC of the tongue, arising from a minor salivary gland, is a rare entity. The standard treatment for SDCs is radical surgery in combination with radiotherapy. There is a paucity of cases of this entity in literature, especially affecting the tongue. Hereby, we report a case of an SDC of the anterior tongue.

Keywords: Salivary duct carcinoma, salivary tumour, tongue


How to cite this article:
Mandlik DS, Dubey T, Mankiwala S, Kanhere S, Patel KD. Salivary duct carcinoma of tongue: A rare entity. J Head Neck Physicians Surg 2019;7:32-4

How to cite this URL:
Mandlik DS, Dubey T, Mankiwala S, Kanhere S, Patel KD. Salivary duct carcinoma of tongue: A rare entity. J Head Neck Physicians Surg [serial online] 2019 [cited 2019 Aug 18];7:32-4. Available from: http://www.jhnps.org/text.asp?2019/7/1/32/263514




  Introduction Top


Salivary duct carcinoma (SDC) is a rare, aggressive malignancy arising in the ductal epithelium of salivary glands. SDC is common in the parotid gland of elderly people and predominates in males.[1] SDC arising from a minor salivary gland is extremely rare.[2] The term salivary duct carcinoma was coined by Kleinsasser et al. in 1968, after which few studies of this entity have been reported.[3] The standard treatment for SDC is surgery in combination with radiotherapy; however, the prognosis of SDC is poor with frequent distal metastasis.[4] We present a case of SDC of the tongue with bilateral neck nodes.


  Case Report Top


A 61-year-old male presented with the complaint of multiple neck swellings for 1 week. Clinical examination revealed around 1 cm-sized swelling at the ventral surface of the anterior tongue. There were bilateral, multiple, hard, palpable neck nodes [Figure 1].
Figure 1: Clinical picture of the tongue lesion

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His ultrasonogram (USG) of the neck showed bilateral submandibular and jugular nodes. His USG-guided needle cytology from tongue lesion showed moderately differentiated adenocarcinoma. His positron emission tomography (PET)-computed tomography (CT) showed high-grade, fluorodeoxyglucose (FDG)-avid, 24 mm × 10 mm × 18 mm–sized, well-defined exophytic hypodense lesion in the anterior ventral surface of the tongue in the midline. High-grade, FDG-avid, heterogeneously enhancing partly necrotic enlarged multiple neck nodes were seen in bilateral Levels II, III, and IV. USG-guided tru-cut biopsy from left cervical nodes was reported as metastatic moderately differentiated adenocarcinoma [Figure 2] and [Figure 3].
Figure 2: Positron emission tomography-computed tomography image

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Figure 3: Color Doppler image with a protruded tongue showing high vascularity

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He underwent anterior partial glossectomy with bilateral modified radical neck dissection. His final histopathology report showed SDC of the anterolateral tongue (tumor size was 25 mm × 15 mm × 15 mm [depth of invasion]). Immunohistochemical staining with gross cystic disease fluid protein, androgen receptor, and S100 demonstrated focal antibodies with a positive pattern of reaction, which was confirmatory for SDC. Staining with mammaglobin, DOG1, and p63 antibodies was negative. Multiple neck nodes were positive on both sides. As per the American Joint Committee on Cancer 8th Edition, he was staged as pT4aN3bM0 [Figure 4].
Figure 4: Histopathology image – Cribriform invasive tumor island comprising large cells with a significant nuclear pleomorphism

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The patient was subjected to postoperative adjuvant radiation (60 Gr over 30 fractions of radiotherapy) after discussion in the multidisciplinary tumor board.

The patient was in regular follow-up with us. At his 2-year follow-up, his clinical examination is oncologically normal. His PET-CT scan at 2-year follow-up is also normal with no locoregional or distant disease.


  Discussion Top


In 2005, SDC was defined as an independent entity by the World Health Organization, labeling it as “an aggressive adenocarcinoma, which resembles high-grade breast ductal carcinoma and exhibits intraductal and invasive components.” Many terms have been used to report SDC, including cribriform salivary carcinoma of excretory ducts, intraductal carcinoma, and infiltrating SDC, which makes a comprehensive literature search difficult.[5]

Intraoral SDC is a rare malignant neoplasm, accounting for <2% of all malignant minor salivary gland tumors of the oral cavity in a recent series. Although it is difficult to determine the predominant location of SDC in the minor salivary glands because of the limited number of cases, they occur most frequently on the palate followed by buccal mucosa or vestibule, tongue maxilla, mandible, and upper lip.[1],[2],[6],[7]

SDC appears to show an aggressive behavior, with around 25% disease-free survival at 20 months in some series and overall survival of around an average of 56 months.[1],[2]

Watatani et al. reported intraductal carcinoma originating from the salivary tissue in the tongue, with few infiltrative components and a less aggressive appearance.[8] In contradiction, Suzuki et al. reported a similar case of SDC of the mobile portion of the tongue where the histological findings were compatible with an aggressive neoplasm with a poor prognosis.[9] Advanced nodal disease, lymphovascular invasion, and extracapsular spread have a detrimental survival impact.[10] Aggressive surgical treatment with adjuvant radiation remains cornerstone management as they are locally aggressive with more than 50% regional metastasis.[1],[2]

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.

Disclosure

This material has never been published and is not currently under evaluation in any other peer reviewed publication.

Ethical approval

The permission was taken from Institutional Ethics Committee prior to starting the project. All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed consent

Informed consent was obtained from all individual participants included in the study.



 
  References Top

1.
Anwer AW, Faisal M, Adeel M, Waqas O, Abu Bakar M, Qadeer S, et al. Clinicopathological behavior and treatment-related outcome of rare salivary duct carcinoma: The Shaukat Khanum Memorial Cancer Hospital experience. Cureus 2018;10:e3139.  Back to cited text no. 1
    
2.
Jaehne M, Roeser K, Jaekel T, Schepers JD, Albert N, Löning T. Clinical and immunohistologic typing of salivary duct carcinoma: A report of 50 cases. Cancer 2005;103:2526-33.  Back to cited text no. 2
    
3.
Kleinsasser O, Klein HJ, Hübner G. Salivary duct carcinoma. A group of salivary gland tumors analogous to mammary duct carcinoma. Arch Klin Exp Ohren Nasen Kehlkopfheilkd 1968;192:100-5.  Back to cited text no. 3
    
4.
Hosal AS, Fan C, Barnes L, Myers EN. Salivary duct carcinoma. Otolaryngol Head Neck Surg 2003;129:720-5.  Back to cited text no. 4
    
5.
Seifert G, Brocheriou C, Cardesa A, Eveson JW. WHO international histological classification of tumours. Tentative histological classification of salivary gland tumours. Pathol Res Pract 1990;186:555-81.  Back to cited text no. 5
    
6.
van Heerden WF, Raubenheimer EJ, Swart TJ, Boy SC. Intraoral salivary duct carcinoma: A report of 5 cases. J Oral Maxillofac Surg 2003;61:126-31.  Back to cited text no. 6
    
7.
Tatemoto Y, Ohno A, Osaki T. Low malignant intraductal carcinoma on the hard palate: A variant of salivary duct carcinoma? Eur J Cancer B Oral Oncol 1996;32B:275-7.  Back to cited text no. 7
    
8.
Watatani K, Shirasuna K, Aikawa T, Matsuya T. Intraductal carcinoma of the tongue. Report of a case. Int J Oral Maxillofac Surg 1991;20:175-6.  Back to cited text no. 8
    
9.
Suzuki M, Suzukawa K, Ogawa M, Suzuki T. Salivary duct carcinoma with comedonecrosis in the mobile portion of the tongue. J Laryngol Otol 2006;120:e13.  Back to cited text no. 9
    
10.
Johnston ML, Huang SH, Waldron JN, Atenafu EG, Chan K, Cummings BJ, et al. Salivary duct carcinoma: Treatment, outcomes, and patterns of failure. Head Neck 2016;38 Suppl 1:E820-6.  Back to cited text no. 10
    


    Figures

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



 

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