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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: [email protected] (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:[email protected]://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:[email protected]://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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