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z

REVIEW

A case of large dermoid cyst of the tongue

Imen Mehri Turki   a,*, Yosr Mouaffak-Zidi   b, Hatem Rajhi   c, Hassen Triki   a,Sonia Naija   a, Sarra Ben Jilani   b, Najla M’Niff   c, Ali Adouani   a

a Departement of Maxillo-Facial Surgery, Charles Nicolle Hospital, Tunis, Tunisiab Departement of Histopathology, Charles Nicolle Hospital, Tunis, Tunisiac Departement of Radiology, Charles Nicolle Hospital, Tunis, Tunisia

Received 29 July 2011; accepted 22 October 2011

Available online 25 November 2011

KEYWORDS

Dermoid cyst;

Tongue;

Magnetic resonance imaging;

Surgery

Abstract   Objective:   We report a case of a large dermoid cyst of the tongue with a review of the

literature.

Case report:   We present a 14-year-old boy who presented an enlarging tongue with slight diffi-

culty of speech. Complete excision of the cyst through an intra-oral approach was possible when

aspiration of the fluid cyst was done at the moment of surgery.

Discussion:  Dermoid cysts are squamous epithelial-lined cavities with variable number of skin

adnexae in the capsule. We discuss clinical, radiological and histological presentation, aetioliogy,

and surgical management of these rare entities in head and neck.

Conclusion:   Although dermoid cysts are rare they should be part of the differential diagnosis of 

tongue masses. The surgical management depends on the size and the extension of the intra-lingual

cyst in the floor of the mouth. The aspiration of the fluid cyst to reduce its size is helpful without any

complication.

ª  2011 Egyptian Society of Ear, Nose, Throat and Allied Sciences.

Production and hosting by Elsevier B.V. All rights reserved.

2090-0740  ª   2011 Egyptian Society of Ear, Nose, Throat and Allied

Sciences. Production and hosting by Elsevier B.V. All rights reserved.

Peer review under responsibility of Egyptian Society of Ear, Nose,

Throat and Allied Sciences.

doi:10.1016/j.ejenta.2011.10.001

Production and hosting by Elsevier

* Corresponding author. Address: Departement of Maxillo-FacialSurgery, 9 April Street, Tunis, Tunisia. Tel.: +216 94789008.

E-mail address:  turkiymen@yahoo.fr (I. Mehri Turki).

Egyptian Journal of Ear, Nose, Throat and Allied Sciences (2011)  12, 171 – 174

Egyptian Society of Ear, Nose, Throat and Allied Sciences

Egyptian Journal of Ear, Nose, Throat and AlliedSciences

www.esentas.org

http://dx.doi.org/10.1016/j.ejenta.2011.10.001mailto:turkiymen@yahoo.frhttp://dx.doi.org/10.1016/j.ejenta.2011.10.001http://dx.doi.org/10.1016/j.ejenta.2011.10.001http://dx.doi.org/10.1016/j.ejenta.2011.10.001http://dx.doi.org/10.1016/j.ejenta.2011.10.001http://www.sciencedirect.com/science/journal/20900740http://www.sciencedirect.com/science/journal/20900740http://dx.doi.org/10.1016/j.ejenta.2011.10.001http://dx.doi.org/10.1016/j.ejenta.2011.10.001mailto:turkiymen@yahoo.frhttp://dx.doi.org/10.1016/j.ejenta.2011.10.001

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Contents

1. Introduction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 172

2. Case report. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 172

3. Discussion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 173

References . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 174

1. Introduction

Dermoid cysts are uncommon lesions in the head and neck,

with approximately 7% of dermoid cysts presenting in this re-

gion, usually observed in the lateral third of the eyebrow, fol-

lowed by the floor of the mouth and rarely in the tongue with

only 8 cases reported in the literature. The aetiology of these

cysts is still controversy. Usually lingual dermoid cysts are dis-

covered at birth or at the first year of life. However when they

are developed in the ventral base of the tongue, their clinical

appearance will be later, at childhood or at early adult life.

The only effective treatment of dermoid cyst of the tongue is

the complete enucleation. The histological examination distin-

guishes this entities from epidermoid cyst and teratoid cyst.1

We report a case of a 14-year-old male presenting a large

intra-glossal dermoid cyst, who underwent surgical excision.

2. Case report

A 14-year-old boy was referred with a gradually swelling of the

ventral base of the tongue without any protrusion. The mass

was covered by a normal mucosa. Normal saliva could be ex-

pressed from the orifices of Wharton’s ducts. Despite this,

there was only a slight difficulty in speech.

The lesion was heterogeneous, and had a predominant low

signal in T1-weighted images on MRI and a high signal on T2-weighted images which containing some area with low signal.

That means the presence of a fat component in the tumour.

The lesion didn’t enhance with gadolinium. It is localized in

the tongue pushing the floor of the mouth without any exten-

sion under the mylohyoid muscle (Figs. 1 and 2).

The patient was operated under general anesthesia with

nasotracheal intubation. A mucosal incision was performed

 just above the level of wharton’s orifices and extending along

Figure 1   T1-weighted coronal MRI shows an heterogeneous

intra-lingual mass.

Figure 2   T2-weighted axial MRI shows a predominant high

signal.

Figure 3   Peroperative view: intra-oral approach, the specimen

and the aspirated fluid cyst.

172 I. Mehri Turki et al.

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the ventral surface of the tongue with a V shaped incision. The

roof of the cyst was identified and once the dissection of the

upper pole of the cyst was completed, we aspirated the cystic

fluid reducing the mass of the lesion surrounded by the fibers

of the genio-glossus muscles that were displacing laterally,

and the genio-hyoid muscles inferiorly. Then a blunt dissection

was continued until a finger could be inserted behind the cyst

so that it could be completely removed (Fig. 3). The wound

was closed after a minucious hemostasis with vicryl 4/0 with-

out using a drainage tube. The patient recovered without com-

plication and was discharged 3 days after operation.

Histological examination concluded to a typical dermoid cyst( Figs. 4 and 5). There was no recurrence at 18 months

follow-up.

3. Discussion

The anterior two third of the tongue develops from fusion of 

three swellings of primitive mesenchyme: two lateral swellings

(the lateral processes) and a median swelling (the tuberculum

impar of Hiss). During the fourth week of embryonic develop-

ment, the two lateral processes migrate medially to fuse over

the tuberculum impar forming the corpus of the tongue. The

musculature of the tongue is formed three weeks later and

originates from the migration of occipital somites.2,3 During

this fusion, entrapment and proliferation of epithelial debris

form dermoid cyst. This is the most popular theory for the eti-

opathogeny of congenital dermoid cyst. A second hypothesis

states that acquired dermoid cyst arise from epidermal and

dermal cells trapped following trauma.4

Shafer postulated that some of the cells enclaved during

these fusion processes are totipotent blastomeres and, there-

fore, derivatives of the three basic germ layers may be present.5

This can explain the histological classification of Meyer that

distinguish dermoid, epidermoid and teratoid cyst.

The epidermoid cyst is lined with simple squamous epithe-

lium and surrounding connective tissue without any skin

appendages. The dermoid type, which our case had, is an epi-

thelial lined cavity with skin appendages of hair follicles, seba-

ceous glands, sweet glands. The cavity is filled with

desquamative cells, keratin and hair. The teratoid type is also

an epithelial lined cavity that contains skin appendages and

connective tissues derivatives such as muscle, bone, teeth and

mucous membrane. However cysts of this type are commonly

confined to the ovaries and testes.6–8

Dermoid cysts in the mouth account for less then 0, 01% of 

all oral cysts. Usually located in the sublingual region butrarely within the tongue.9 A simulnaneous dermoid cyst   of 

the tongue and the midline of the neck has been reported. 10

Most cases reported of lingual dermoid cyst follow a bimodal

distribution with a peak during the adolescence and a smaller

peak during the first year of life. Male and female are affected

equally.10,11

Clinically dermoid cyst may be asymptomatic, often discov-

ered by a painless swelling growing slowly and surrounding by

a normal mucosa. The cyst can enlarge to a point that the

patient present difficulty of articulation, mastication and

deglutition and airway compromise. He can also be unable

to close his mouth with sometimes lingual displacement.12

Recurrent infections can occur dissecting the tongue musclesin the zone of low resistance and opening up the midline of 

the dorsum of the tongue forming a sinus tract. Although an

embryologic theorie stipulate that the sinus is like the   cyst,

an epithelial remnant persisting after midline closure.13 It al-

lows drainage of the keratin and glandular products. On the

other side, the sinus tract is an entry for organisms of the oral

flora.4,14 Malignant transformation of a dermoid cyst to squa-

mous cell carcinoma is exceptionally rare in head and neck.

Only one case in the litterature of sublingual dermoid cyst

has been   malignant transformation but never a lingual der-

moid cyst.15

The differential diagnosis of a painless swelling in the mid-

line of the tongue include cystichygroma, lymphatic cyst, neu-

rofibroma, haemangioma, lingual thyroid, enteric duplicationcyst and dermoid cyst.16,17

Magnetic resonance imaging (MRI) is an accurate, non-

invasive modality for the diagnosis and procure follow-up

evaluation for lingual dermoid cyst. It has the advantage to

delineate the cyst and precise its possible extension through

the floor of the mouth. Dermoid cysts are iso- or hypointense

to muscle on T1-weighted images and hyperintense or hetero-

geneous on T2-weighted images. The diagnosis is suggested

when a   fat component exist in the tumour on MRI, like in

our case.18,19

Complete removal of the cyst is the effective treatment. An

intra-orally approach with a mucosal incision usually through

Figure 4   The cyst wall is lined by a stratified squamous

epithelium similar to normal skin, and contains mature epidermal

appendages (HE   ·10).

Figure 5   The cyst lies on the muscular layer of the tongue.

A case of large dermoid cyst of the tongue 173

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the ventral surface of the tongue is appropriated. The surgeon

should give a lot of care to avoid submandibular ducts. When

the cyst is so large or extending under the mylohyoid muscle,

an extra-oral approach is required. Some authors described

the advantage of aspiration of the fluid cyst at the moment

of surgery. This can provide easier access of large cyst with

only an intra-oral approach. We didn’t note, as documented

in the literature, any complication neither recurrence after

aspiration. Because of the consequent thickness of   dermoidcyst, blunt dissection is safe to remove the entire cyst.20–22

Recurrence would be rare although another cyst can arise

from cyst fragments or sinus tract left by surgery. Other

authors suggest an unlikely alternative that a daughter cyst

can be detached from the main one and grows separately until

its discovery, of course, after the first operation of the main

cyst.7,14,23

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