Dental Science - Case Report

Lymphoepithelial of the

A. Saneem Ahamed, V. Sadesh Kannan1, K. Velaven2, G. R. Sathyanarayanan3, J. Roshni, E. Elavarasi4

Departments of ABSTRACT Consultant Maxillofacial Lymphoepithelial are benign, slowly growing unilocular or multilocular lesions that appear in the head Surgeon, Appasamy Multispeciality Hospital, and neck. They are also called Branchial cyst. The head and neck sites are the salivary glands(more commonly Arumbakkam, 2Impacts parotid and rarely submandibular gland) and the oral cavity (usually the floor of the ). there are various 32 Dental Clinic, Old methods of investigation available today, of which Fine needle aspiration cytology (FNAC) can be used to Pallavaram, Chennai, provide an immediate diagnosis of a lymphoepithelial cyst. The other investigations include, Ultrasonogram,and 1BeWell Hospitals and Computed tomography.It usually occurs due to the process of lymphocyte-induced cystic ductular dilatation and The Dental Clinic, the confirmatory diagnosis is always made postoperatively by histopathological examination. The mainstay in Pakkamudayanpet, the treatment of a lymphoepithelial cyst remains the surgical approach, which includes complete enucleation 4 Consultant Oral and of the cyst along with total excision of the involved . This is a report of a lymphoepithelial cyst Maxillofacial Radiologist, involving the submandibular salivary gland and its management. Pudupet, Lawspet, Puducherry, 3Neu Face Hospitals, Thanjavur, Tamil Nadu, India

Address for correspondence: Dr. A. Saneem Ahamed, E‑mail: saneem.omfs@gmail. com

Received : 30-03-14 Review completed : 30-03-14 Accepted : 09-04-14 KEY WORDS: Lyphoepithelial cyst, sub-mandibular gland, benign neck lesion malignant transformation

ymphoepithelial cysts are benign, slowly growing occur without involving the salivary glands, most commonly in L unilocular or multilocular lesions that appear in the head the anterior triangle of the neck. If not treated, there is a high and neck. They are also called Branchial cyst. The head and neck chance of lymphoepithelial cysts transforming into malignant sites are the salivary glands (more commonly parotid and rarely lesions such as malignant lymphomas, which has the capacity submandibular gland) and the oral cavity (usually the floor of to involve the extranodal sides secondarily. This is a case report the mouth). These cysts are usually seen in adults and only of lymphoepithelial cyst involving the submandibular gland. occasionally in children. They range in size from 0.5 to 5.0 cm, and they can cause considerable cosmetic deformity and Case Report physical discomfort. Sometimes, the lymphoepithelial cyst may The case we present here is about a 32‑year‑old, female patient Access this article online who reported to our hospital with the complaint of a painless Quick Response Code: swelling on the left side of the neck for past 8 months. The Website: swelling was small initially and has progressed to the present www.jpbsonline.org size [Figure 1]. The swelling did not cause any functional restriction but was cosmetically evident, which was the DOI: patient’s concern. On extraoral examination, there was a soft, 10.4103/0975-7406.137464 fluctuant, compressible, nontender swelling of size 8-10 cm in the left submandibular region. The swelling extended

How to cite this article: Ahamed AS, Kannan VS, Velaven K, Sathyanarayanan GR, Roshni J, Elavarasi E. Lymphoepithelial cyst of the submandibular gland. J Pharm Bioall Sci 2014;6:S185-7.

Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1 S185  Ahamed, et al.: Lymphoepethelial cyst of submandibular gland anteriorly from the parasymphysis region of , and component usually arise on the lateral cervical area including extended posteriorly until the angle region of the mandible. the lymphnode. They have been classically described to occur Superiorly the swelling did not go beyond the lower border anterior to the upper third of the sternomastoid. Anyways, of mandible and inferiorly extended until the level of thyroid they have been known to occur in other areas of neck, oral cartilage. On intraoral examination, there was no abnormal cavity, salivary glands, thyroid, and mediastinum and even finding. The orifice of the left submandibular duct and the flow was normal.

On intraoral examination, there were no abnormal finding and the patient did not complaint of any dryness of mouth or pain along the floor of the mouth or at the opening of the submandibular duct.

An aspiration was done from the swelling using a wide bore needle, which showed the presence of a Straw colored fluid. The aspirated fluid subjected for fine‑needle aspiration cytology (FNAC), which revealed the presence of lymphocytes, few histiocytes, few plasma cells and squamous cells dispersed in a proteinaceous material. This almost proved that the lesion is a cyst from a lymphoepithelial origin.

A computed tomography (CT) scan was done to assess the size and extent of the swelling which revealed the presence Figure 1: Preoperative photo showing cyst on the left side sub- of a well circumscribed and encapsulated lesion measuring mandibular region about 10 cm × 6 cm. The lesion did not show any invasion into the mandible or major blood vessels, except it was pushing the facial artery and vein medially into the neck. The capsule of the lesion was slightly adherent to the superficial part of the left side submandibular gland [Figure 2]. A routine blood investigation was performed. The patient was tested for human immunodeficiency (HIV) both with enzyme linked immunosorbent assay and Western blot test, both of which were negative.

The treatment was very definitive and the patient was planned for excision of the lesion under general anesthesia. The surgical approach was made with a horizontal neck incision along the neck crease. Facial artery and vein were seen adherent to the cystic wall and hence they were ligated. Marginal mandibular Figure 2: Computed tomography of the submandibular region shows nerve was identified and preserved. The lesion was exposed a well encapsulated mass seen on the left submandibular region being and was completely enucleated along with the excision of attached to superficial part of left submandibular gland the superficial part of the submandibular salivary gland. Hemostasis was achieved and closure was done. The specimen was sent for histopathological examination, which revealed the specimen consisting of cyst lined by glandular epithelium consisting of lymphoid follicles [Figure 3].

Discussion

Lymphoepithelial cysts or the branchial cysts are the benign, slow growing lesion that occurs mostly in adults with a predilection for gender with 60-80% being female. It occurs in the second and third decade of life and the swelling is usually diffuse, fluctuant and nontender. The lymphoepithelial cysts are usually associated with the salivary gland, especially the and rarely the submandibular gland.[1] In this kind of lesion, as the glands are affected, the duct of the gland is also affected as in the above case, submandibular gland and duct.[2] The branchial cysts which do not involve the salivary Figure 3: Postoperative photo after 2 weeks of excision

 S186 Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1 Ahamed, et al.: Lymphoepethelial cyst of submandibular gland in pancreas. It occurs due to lymphocyte induced cystic differential diagnosis include lymphoepithelial , ductular dilatation and it is always diagnosed postoperatively HIV‑associated , chronic sclerosing by histopathlogic examination. sialadenitis, Warthin tumor, and extranodal marginal zone B‑cell lymphoma, salivary duct retention cyst (mucocele), There are various theories proposed as the cause for a branchial dysgenetic polycystic disease of the salivary gland, mucosa cyst. They can be broadly classified as congenital theories and associated lymphoid tissue lymphoma. Patients with salivary lymph node theories. Of the two theories, the most accepted gland lymphoepithelial cyst are at the increased risk of is the lymphnode theory suggested by kings. It proposed developing lymphoma and that is reason such cyst should be that cysts developed due to the cystic transformation of the treated as early as possible. lymphnodes. Lymphoepithelial cysts can arise with other condition like Sjogren’s syndrome, mikulicz’s disease and Treatment of the lyphoepithelial cyst includes both myoepithelial sialadinitis. In Sjogren’s syndrome minor salivary conservative as well as surgical approach. The conservative glands are involved, but generally lack a lymphoepithelial approach includes decompression the cyst by aspirating the component.[3] fluid out thereby reducing the pressure. Such a procedure should be considered in an immudeficient patient like The lymphoepiyheliail cyst has been associated with HIV HIV positive in which the surgical management is clearly infection as part of a diffuse infiltrative lymphocytosis outweighed by the risk. Other conservative treatment includes syndrome. HIV‑associated salivary gland disease is a lymphoid external radiotherapy. The definitve treatment is the surgical hyperplasia in the parotid and sometimes submandibular, management by complete enucleation of the cyst along gland with lymphoepithelial cyst and lymphoepithelial lesions with the excision of the involved gland. Most patients are in HIV positive patients. The incidence of HIV‑associated completely cured by excision and never get a recurrence.[6] salivary disease is about 3-10% among the HIV positive patient. Children and adults, males and females are affected. Salivary Conclusion gland disease usually develops before AIDS and sometimes it is the first manifestation of HIV infection. It is usually bilateral It should kept in mind that the lymphoepithelial cysts are and is accompanied by cervical lymphadenopathy.[4] benign in nature and should be treated as early as possible as it can transform itself into a malignant lesion such as malignant The investigation modalities include FNAC, which is usually lymphoma, , therapeutic as well as a confirmatory test if performed and surgery remains the main‑stay in the treatment of all accurately. The presence of proteinaceous background and a lymphoepithelial and lateral cervical cyst.[4] mixed population of lymphocytes, histiocytes, plasma cells, and metaplastic squamous cells in the cytologic findings on References the fine‑needle aspiration of a major salivary gland lesion, the [5] diagnosis of lymphoepithelial cyst should be considered. 1. Ioachim HL. Lymphoid hyperplasias and lymphomas of salivary CT helps in determining the nature of cyst, if it is encapsulated glands. Pathol Case Rev 2004;9:206-13. or breaching and invading into other structures. The most 2. Tiwari A, Kini H, Pai RR, Rau AR. HIV lymphadenitis of the salivary gland: A case with cytological and histological correlation. J Cytol important is that the CT reveals the extent of the lesion in 2009;26:146‑8. all direction before planning for surgical excision. Magnetic 3. Panchbhai AS, Choudhary MS. Branchial cleft cyst at an unusual resonance imaging (MRI) with contrast, which would clearly location: A rare case with a brief review. Dentomaxillofac Radiol show thin rim enhancement. Ultrasonogram can be performed, 2012;41:696‑702. 4. Rojas R, Di Leo J, Palacios E, Rojas I, Restrepo S. Parotid gland which can very well‑demonstrate a cystic lesion and a vascular lymphoepithelial cysts in HIV infection. Nose Throat J lesion. can be done to detect any obstruction 2003;82:20‑2. within the gland and duct. Both MRI and sialography was not 5. Lester J. Gopez EV. Cystic lesions of the salivary glands: Cytologic features in fine-needle aspiration biopsies 2002; 27:4,197-204. done as the patient was unaffordable. 6. Habib S, Rahman MM, Chaudhury AA, Kamal M. Benign lymphoepithelial cyst. Bangladesh J Otorhinolaryngol 2010;16. The lesions of salivary glands with a prominent lymphoid component are a heterogeneous group of diseases that Source of Support: Nil, Conflict of Interest: None declared. include benign reactive lesions and malignant neoplasms. The

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