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Year : 2019  |  Volume : 15  |  Issue : 3  |  Page : 737-738

Synchronous double malignancy of lip and submandibular salivary gland with rare histology

1 Department of Surgery, JIPMER, Puducherry, Tamil Nadu, India
2 Department of Surgery, Government Kanyakumari Medical College, Nagercoil, Tamil Nadu, India
3 Consultant Pediatrician, CSI Kalyani Multispeciality Hospital, Mylapore, Chennai, Tamil Nadu, India

Date of Web Publication29-May-2019

Correspondence Address:
Dr. C Danny Darlington
No. 49, G3 Ananti Apartment, Santhome High Road, Santhome, Chennai - 600 004, Tamil Nadu
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jcrt.JCRT_270_17

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 > Abstract 

In the modern era, there is an increase in the incidence of double malignancies owing to the remarkable improvement in cancer diagnostics and patient survival. Double malignancies can be either synchronous or metachronous. Synchronous double malignancy can present either at the same time or within 6 months of diagnosis of the first one. We present a case of double malignancy of lip and submandibular salivary gland, diagnosed in a 55-year-old male, who presented with ulceroproliferative lesion of the upper lip and a hard swelling in the right submandibular gland. The tissue diagnosis was suggestive of squamous cell carcinoma of the lip and mucoepidermoid carcinoma of the submandibular gland. The patient underwent successful wide local excision of the lip and submandibular gland along with prophylactic supraomohyoid neck dissection. This case is reported for the rarity in site and histopathology of double malignancy.

Keywords: Carcinoma of lip, double malignancy, mucoepidermoid carcinoma, squamous cell carcinoma, synchronous

How to cite this article:
Darlington C D, Joseph S C, Anitha G F. Synchronous double malignancy of lip and submandibular salivary gland with rare histology. J Can Res Ther 2019;15:737-8

How to cite this URL:
Darlington C D, Joseph S C, Anitha G F. Synchronous double malignancy of lip and submandibular salivary gland with rare histology. J Can Res Ther [serial online] 2019 [cited 2021 Dec 8];15:737-8. Available from: https://www.cancerjournal.net/text.asp?2019/15/3/737/244447

 > Introduction Top

Double malignancies of the oral cavity are rarely reported. Synchronous cancers in head and neck can be explained by the concept of field cancerization. Malignancy of the lip commonly presents as squamous cell carcinoma (SCC), whereas adenocarcinoma is common in salivary glands. We report a rare combination of double malignancy with SCC of the lip and mucoepidermoid carcinoma (MEC) of the submandibular salivary gland, diagnosed in a 55-year-old male.

 > Case Report Top

A 55-year-old male presented to the outpatient department with a painless swelling of the upper lip for a duration of 3 months. He was a chronic smoker and alcoholic, and there was no significant past or family history. Local examination revealed an ulceroproliferative lesion of size 3 cm involving the outer one-third of the upper lip on the right side and the commissure, which was mobile and not tender on palpation [Figure 1]. The buccal mucosa was free of growth. Examination of the neck showed a hard, mobile swelling of size 2 cm involving the right submandibular gland [Figure 2], which was also confirmed by intraoral palpation. Biopsy of the lip lesion revealed a well-differentiated SCC. Fine-needle aspiration cytology of the submandibular swelling was done which was suggestive of a moderately differentiated MEC. The patient's laboratory investigations and chest X-ray were within normal limits. Computerized tomography of the neck showed a heterogeneous tumor with minimal small cystic areas confined to the submandibular gland. Metastatic workup was negative.
Figure 1: Clinical photograph of the lip lesion

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Figure 2: Clinical photograph of the submandibular region

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The patient underwent wide local excision of the lip malignancy with reconstruction of the upper lip using Estlander flap. The submandibular gland excision was done along with the right supraomohyoid neck dissection, as the neck was clinically N0. Postoperative histopathology was reported as well-differentiated SCC of the lip with all margins free of tumor (p T2N0M0). The submandibular gland tumor was a moderately differentiated MEC of stage pT2N0M0. No adjuvant therapy was given in view of the organ-confined disease. The patient is doing well after one year of follow-up.

 > Discussion Top

MEC occurs predominantly in major salivary glands. It represents about 5% of salivary gland tumors. The risk factors for these cancers include exposure to ionizing radiation, radiotherapy history, nickel, chemical solvents, leather, and formaldehyde exposure.[1] Our patient is a farmer and had no such exposure. These tumors consist of three types of cells - mucous cells, epidermoid squamous cells, and poorly differentiated intermediate cells. When the epidermoid cells predominate, the tumor resembles SCC and is considered high grade. When the mucous cells predominate, it is low-grade MEC. Moderately differentiated tumors have predominant population of intermediate cells.[1] The treatment of choice of MEC is always surgical excision. Postoperative adjuvant radiotherapy can be given for high-grade MECs.[2]

SCC of the lip is the most common oral cavity malignancy. The main risk factors include smoking, tobacco chewing, and chronic alcoholism. Exposure to viruses - HPV16 and HPV24 and HSV1 and HSV2 - is a predisposition, especially in immunosuppressed individuals. While SCC is more common in the lower lip than the upper lip, in this case, it involved the upper lip. Our patient was a smoker and alcoholic for 20 years which is a significant risk factor. Wide local excision of the primary with supraomohyoid neck dissection (SOHND) has been the standard of care for early lip cancers, especially in patients with large and deep tumors, commissure involvement, perineural infiltration, or recurrences. SOHND is done as a method of sampling and also to remove any occult metastases in the cervical nodes. Small defects of the lip can be closed primarily. Lip reconstruction using Estlander flap was needed in this case as the commissure was involved.[3]

Double malignancies are diagnosed only when they fulfill the Warren and Gates criteria, as in this case.[4] Synchronous double malignancies of MEC and SCC of head and neck have never been reported so far. In this case, an early diagnosis and a high index of suspicion lead to a better outcome.

 > Conclusion Top

A high index of suspicion is needed when dealing with head-and-neck malignancies. Field cancerization should be considered, and there should be a thorough search for second primary. This case is reported for the rare histopathological combination of double malignancies involving the head and neck.

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.

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Conflicts of interest

There are no conflicts of interest.

 > References Top

Mesolella M, Iengo M, Testa D, DI Lullo AM, Salzano G, Salzano FA, et al. Mucoepidermoid carcinoma of the base of tongue. Acta Otorhinolaryngol Ital 2015;35:58-61.  Back to cited text no. 1
Kolude B, Lawoyin JO, Akang EE. Mucoepidermoid carcinoma of the oral cavity. J Natl Med Assoc 2001;93:178-84.  Back to cited text no. 2
Moretti A, Vitullo F, Augurio A, Pacella A, Croce A. Surgical management of lip cancer. Acta Otorhinolaryngol Ital 2011;31:5-10.  Back to cited text no. 3
Acharya P, Ramakrishna A, Kanchan T, Magazine R. Dual primary malignancy: A rare organ combination. Case Rep Pulmonol 2014;2014:760631.  Back to cited text no. 4


  [Figure 1], [Figure 2]

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