|LETTER TO EDITOR
|Year : 2014 | Volume
| Issue : 2 | Page : 251-253
Hair discharging from the frenulum: Unusual presentation of a lingual dermoid cyst
Archana A Arya1, Shridhar V Babanagare2, Chetana Naik1, Sanjay D Deshmukh2
1 Department of ENT, Smt. Kashibai Navale Medical College, Narhe, Pune, Maharashtra, India
2 Department of Pathology, Smt. Kashibai Navale Medical College, Narhe, Pune, Maharashtra, India
|Date of Web Publication||10-Apr-2015|
Archana A Arya
Department of ENT, Smt. Kashibai Navale Medical College, Narhe, Pune, Maharashtra
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Arya AA, Babanagare SV, Naik C, Deshmukh SD. Hair discharging from the frenulum: Unusual presentation of a lingual dermoid cyst. Natl J Maxillofac Surg 2014;5:251-3
|How to cite this URL:|
Arya AA, Babanagare SV, Naik C, Deshmukh SD. Hair discharging from the frenulum: Unusual presentation of a lingual dermoid cyst. Natl J Maxillofac Surg [serial online] 2014 [cited 2021 May 9];5:251-3. Available from: https://www.njms.in/text.asp?2014/5/2/251/154858
Dermoid cysts are benign congenital lesions with three histological types namely epidermoid, dermoid and teratoma. Their incidence is: 6.9% in the head and neck and 1.6% within the oral cavity. They represent less than 0.01% of all oral cavity cysts. 
Dermoid cysts present either at birth or in the 2 nd and 3 rd decade of life.  Patients generally present with the complaints of swelling or symptoms related to rupture of the cyst.
Our patient was a 24-year-old male with the complaint of hair like structures and pus discharging from the frenulum of the tongue since 3 years. There was no history of difficulty in chewing, swallowing, dyspnea or pain. There was no history of trauma. On examination, a long white hair like structure was noted protruding from the fistulous opening at the frenulum. The hair like fiber was removed and sent for histopathological evaluation, which confirmed it to be a hair shaft.
A computed tomography with sinogram showed a well-defined, contrast enhancing cystic lesion measuring 1 cm, situated in the anterior third of the tongue with a fistulous opening at the frenulum [Figure 1].
|Figure 1: Computed tomography scan (contrast) - axial cuts showing the fistulous tract along with the cyst in the tongue|
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Surgical excision of the lesion was planned and an elliptical incision was taken around the fistulous opening at the frenulum. The tract was traced and complete excision of the fistulous track and cyst was carried out [Figure 2]. The post-operative period was uneventful with no recurrence after 1 year of follow-up.
|Figure 2: Midline attachment of dermoid cyst (arrow) after dissection from the tongue|
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On histological examination, a cyst lined by stratified squamous epithelium was seen. Presence of skin adnexal structures including hair follicles as well as sebaceous glands was noted [Figure 3] and [Figure 4]. The lumen of the cyst showed exfoliated cellular debris admixed with hair shafts. These features established the diagnosis of dermoid cyst.
|Figure 3: Cyst lined by stratified squamous epithelium with skin adnexal structures in the cyst wall (H and E, ×40)|
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|Figure 4: Cyst wall showing hair shaft surrounded by tissue reaction consisting of acute on chronic inflammatory infiltrate (H and E, ×400)|
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Epidermoid cysts, dermoid cysts and teratomas are distinct lesions with some overlapping features. Epidermoid cysts are derived from ectoderm and their walls are lined solely by squamous epithelium. 
Dermoid cysts are also derived from ectodermal layer, but with skin adnexa present in the wall of the cyst. Teratomas are true neoplasms that contain tissues that are either foreign to the primitive site of origin or histologically diverse and represent more than one of the embryogenic germ layers.  The literature search revealed only 14 reported cases of dermoid cysts of the tongue. Of these, seven were of the epidermoid type, three were teratomas and only four were true dermoid cysts of the tongue.  Ours is the fifth case of true intralingual dermoid cyst and the only reported case with a presenting symptom of a fistulous tract with hair discharging out of the frenulum. In none of the cases published in the literature, such an unusual presenting symptom was described. Dermoid cysts are congenital in origin and occur either due to failure of surface ectoderm to separate from underlying structures, sequestration of surface ectoderm or implantation of surface ectoderm probably during 3 rd to 5 th weeks of gestation. 
Dermoid cyst can arise all over the body, but the most common location is the anus (44.5%), followed by the ovary (42%).  The incidence in the head and neck region is 6.9%, where the lateral aspect of the eyebrow, pinna, nose, neck and lip are commonly involved. , In the mouth, the floor is the most frequent location. Other cystic lesions that may present in the tongue are cystic hygroma, ranula, thyroglossal cyst and foregut duplication cyst. 
All the reported cases of lingual dermoid cysts have presented early in age with difficulty in swallowing and breathing.
Surgery is the mode of treatment for these lesions. The experience of this case indicates that whenever one encounters cystic lesions in tongue, the possibility of dermoid cyst should be kept in mind and a complete resection including the tract should be planned. Prognosis is very good with no recurrence if the resection is complete.
| References|| |
Ozan F, Polat HB, Ay S, Goze F. Epidermoid cyst of the buccal mucosa: A case report. J Contemp Dent Pract 2007;8:90-6.
Smirniotopoulos JG, Chiechi MV. Teratomas, dermoids, and epidermoids of the head and neck. Radiographics 1995;15:1437-55.
Shaari CM, Ho BT, Shah K, Biller HF. Lingual dermoid cyst. Otolaryngol Head Neck Surg 1995;112:476-8.
Suga K, Muramatsu K, Uchiyama T, Takano N, Shibahara T. Congenital epidermoid cyst arising in soft palate near uvula: A case report. Bull Tokyo Dent Coll 2010;51:207-11.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]