Salivary duct carcinoma of minor salivary gland: A case report with review of literature

Arshdeep Kaur; Chetan Dev Singh Boparai; Anusha Rangare Lakshman; Gogineni Subhas Babu

doi:10.4103/jorr.jorr_23_18

CASE REPORT

Year : 2019 | Volume : 11 | Issue : 1 | Page : 25-28

Salivary duct carcinoma of minor salivary gland: A case report with review of literature

Arshdeep Kaur¹, Chetan Dev Singh Boparai², Anusha Rangare Lakshman³, Gogineni Subhas Babu⁴
¹ Department of Oral Medicine and Radiology, Sri Guru Ram Das Institute of Dental Sciences and Research, Amritsar, Punjab, India
² Department of Orthodontics, Sri Guru Ram Das Institute of Dental Sciences and Research, Amritsar, Punjab, India
³ Department of Oral Medicine and Radiology, Century International Institute of Dental Sciences and Research Centre, Kasaragod, Kerala, India
⁴ Department of Oral Medicine and Radiology, A. B. Shetty Memorial Institute of Dental Sciences, Mangalore, Karnataka, India

Date of Submission	23-Aug-2018
Date of Acceptance	24-Nov-2018
Date of Web Publication	6-Mar-2019

Correspondence Address:
Arshdeep Kaur
Department of Oral Medicine and Radiology, Sri Guru Ram Das Institute of Dental Sciences and Research, Amritsar, Punjab
India

Source of Support: None, Conflict of Interest: None

Check

DOI: 10.4103/jorr.jorr_23_18

Abstract

Salivary duct carcinoma (SDC) is a rare, highly aggressive neoplasm representing about 1%–3% of all malignant salivary gland tumors. Approximately, 85% of the cases occur in the parotid gland followed by the submandibular gland and minor salivary gland. Pathomorphologically, these tumors showed great similarities to ductal carcinoma of the female breast, which is why they described this tumor as “SDC.” This article highlights a case of rapidly growing swelling in the left mandibular region in a 60-year-old female. As this entity is rare and highly aggressive, an early detection followed by appropriate treatment is very important to improve the survival rate of the patient.

Keywords: Salivary duct carcinoma, submandibular, swelling

How to cite this article:
Kaur A, Singh Boparai CD, Lakshman AR, Babu GS. Salivary duct carcinoma of minor salivary gland: A case report with review of literature. J Oral Res Rev 2019;11:25-8

How to cite this URL:
Kaur A, Singh Boparai CD, Lakshman AR, Babu GS. Salivary duct carcinoma of minor salivary gland: A case report with review of literature. J Oral Res Rev [serial online] 2019 [cited 2023 May 30];11:25-8. Available from: https://www.jorr.org/text.asp?2019/11/1/25/253428

Introduction

Salivary duct carcinoma (SDC) is a rare tumor which accounts for 1%–3% of all malignant salivary gland tumors.^[1] Initially, a group of malignant salivary gland tumors characterized by ductal formations and central necrosis was first described by Kleinsasser et al. in 1968.^[2] In the literature, very few cases have been reported resulting in limited data regarding the biologic and the immunohistochemical characteristics of this tumor.

Case Report

A 60-year-old female patient reported with a complaint of swelling in the left mandibular region of 2 months' duration which progressed rapidly in size within 2 weeks. It was associated with continuous, dull aching pain. Medical history revealed that she was a known case of idiopathic thrombocytopenic purpura. She did not report of any constitutional symptoms such as fever, weight loss, and night sweats. She gave a history of betal quid chewing since 20 years.

Extraoral examination showed a well-defined swelling on the left lower third of face swelling measuring approximately 3 cm × 4 cm. The overlying skin appeared to be slightly erythematous, stretched, and shiny [Figure 1]. It was firm, tender, and indurated on palpation. Paresthesia was absent. The right submandibular lymph node could not be assessed due to the extent of the swelling, and other lymph nodes were not palpable. Intraorally, a proliferative growth was noticed in mandibular left second and third molar region which appeared to arise buccally from the alveolus measuring approximately 2 cm × 1 cm in size and partially obliterating the buccal vestibule [Figure 2]. The growth was firm, nontender, and partially movable on palpation. Bleeding was also noticed on provocation and left mandibular molars showed Grade II mobility. A provisional diagnosis of squamous cell carcinoma was considered based on the clinical findings followed by metastatic lesion and mucoepidermoid carcinoma as differential diagnosis.

Figure 1: Extraoral photograph showing a swelling of left side of the face

Click here to view

Figure 2: Intraoral photograph showing a proliferative gingival growth

Click here to view

Panoramic radiograph showed horizontal bone loss in the 36–38 region [Figure 3]. Ultrasonography of neck showed a well-defined hypoechoic lesion adjacent to left ramus of mandible measuring approximately 2.85 cm × 2.16 cm in size. Submandibular gland, parotid gland, and thyroid gland were normal and 2–3 submandibular lymph nodes were also noted adjacent to the lesion with largest of them measuring approximately 1.8 cm in its greatest dimension. Computed tomography scan revealed a soft-tissue density lesion in the left submandibular region suggestive of lymph node mass. Submandibular, parotid and thyroid gland were normal [Figure 4]. Hematological investigations revealed thrombocytopenia. Chest X-ray was clear, and ultrasound abdomen was normal. Then, incisional biopsy of the intraoral growth was performed which revealed epithelium proliferating into the connective tissue in the form of islands and sheets showing dysplastic features. Areas of necrosis were also noted at some areas. It was suggestive of a poorly differentiated squamous cell carcinoma. Then, hemimandibulectomy with neck dissection was planned followed by postoperative radiation therapy. Later, excisional biopsy microscopically showed tumor cells which were round to polygonal in shape and were showing predominantly clear cytoplasm and cellular atypia [Figure 5]a and were arranged in circumscribed organoid nests showing central comedonecrosis [Figure 5]b.

Figure 3: Panoramic radiograph showing crestal bone loss in 36–38 region

Click here to view

Figure 4: Axial section of computed tomography scan showing a soft-tissue density lesion in the left submandibular region

Click here to view

Figure 5: (a) photomicrograph shows round to polygonal tumor cells with clear cytoplasm and cellular atypia. (b) photomicrograph shows tumor cells arranged in organoid circumscribed nests with central comedonecrosis

Click here to view

Based on histopathological findings, a diagnosis of SDC was suggested. Clinical examination of breast, mammography, and ultrasonography was done to rule out breast carcinoma.

Discussion

SDC is an uncommon high grade, aggressive malignant tumor which was first described by Kleinsasser in 1968.^[2] It was only recently recognized as a distinct clinicopathological entity. These are commonly seen in the major salivary glands, especially in the parotid gland. SDC rarely involves the submandibular gland and minor salivary glands. Intraorally, the common sites of occurrence are palate, followed by buccal mucosa/vestibule, upper lip, and maxilla/mandible. SDC occurs predominantly in males, with a male to female ratio of 2:1. Moreover, most cases being reported in the fifth and sixth decades of life.^[3] Patients usually present with a rapidly growing mass associated with facial nerve involvement. Metastases are common at the time of presentation to lymph nodes and extralymphatic sites such as lung, bone, and brain.^[4]

Histologically, it is characterized by a striking resemblance to ductal carcinoma of the breast. The terms, “cribriform salivary carcinoma of the excretory duct” and “infiltrating salivary duct carcinoind” have been recommended for SDC, to distinguish it from other salivary carcinomas, many of which may also be “ductal” in origin. However, the designation, “SDC” has been accepted because it is used in the WHO classification of tumours.” The separation of SDC from the category, “adenocarcinoma not otherwise specified,” is warranted by its aggressive behavior.^[3]

SDC shows a wide range of histological features.^[5] The histopathological features of SDC consist of atypical cuboidal or polygonal cells which are arranged in papillary cribriform and solid growth patterns along with duct-like structures. The tumor cells show granular eosinophilic cytoplasm, enlarged hyperchromatic, pleomorphic nuclei, and prominent nucleoli. Pseudocyst formation and central comedonecrosis are seen in the neoplastic islands. Vascular invasion and perineural infiltration have been reported in some cases. Atypical mitotic figures are seen in most of the lesions. Dystrophic calcifications are seen in some cases.^[6]

The differential diagnosis of SDC includes a range of low-grade and high-grade salivary gland malignant neoplasms. The most common ones are papillary cystadenocarcinoma, papillary cystic acinic cell carcinoma, and metastatic adenocarcinoma. Lymph node metastases have been reported in 83% SDC cases of the major salivary glands.^[7]

SDC has a very poor prognosis and is considered to be more aggressive than other types of salivary gland carcinomas. There is a 60%–70% mortality rate with over 55% of the patients died within 3 years of clinical presentation. This is due to its metastatic potential.^[8]

The recommended mode of therapy is complete local resection with concomitant neck dissection, followed by postoperative radiation therapy.^[4] Chemotherapy is generally reserved for metastatic forms of the disease.^[8] Kuroda et al. reported the utility of both anti-androgen therapy and chemotherapy for a patient with advanced SDC.^[9]

Conclusion

As the SDC originating in minor salivary glands is a rare and highly aggressive entity, early detection followed by appropriate treatment and regular follow-up is essential to rule out regional and distant metastasis and to improve the survival rate of the patient.

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

1.	Jaehne M, Roeser K, Jaekel T, Schepers JD, Albert N, Löning T, et al. Clinical and immunohistologic typing of salivary duct carcinoma: A report of 50 cases. Cancer 2005;103:2526-33.
2.	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.
3.	Nalin Kumar S, Ranganathan K, Nathan JA, Saraswathi TR. Salivary duct carcinoma of minor salivary gland. J Oral Maxillofac Pathol 2004;8:87-90.
4.	Butterworth DM, Jones AW, Kotecha B. Salivary duct carcinoma: Report of a case and review of the literature. Virchows Arch A Pathol Anat Histopathol 1992;420:371-4.
5.	Delgado R, Vuitch F, Albores-Saavedra J. Salivary duct carcinoma. Cancer 1993;72:1503-12.
6.	Rajini KM, Prakash AR, Reddy PS. Salivary duct carcinoma of the minor salivary glands: A rare case report. J Clin Diagn Res 2011;5:361-2.
7.	Motoori K, Iida Y, Nagai Y, Yamamoto S, Ueda T, Funatsu H, et al. MR imaging of salivary duct carcinoma. AJNR Am J Neuroradiol 2005;26:1201-6.
8.	Mlika M, Kourda N, Zidi YSH, Aloui R, Zneidi N, Rammeh S, Zermani R, Jilani SB. Salivary duct carcinoma of the parotid gland. J Oral Maxillofac Pathol 2011;6:134-6.
9.	Kuroda H, Sakurai T, Yamada M, Uemura N, Ono M, Abe T, et al. Effective treatment by both anti-androgen therapy and chemotherapy for a patient with advanced salivary duct carcinoma. Gan To Kagaku Ryoho 2011;38:627-30.