Asian J Oral Maxillofac Surg. 2009;21:51-53.

CASE REPORT

Dermoid Cyst with Fistula on the Dorsum of the Tongue

Yoshiyuki Mori, Hideto Saijo, Hisako Fujihara, Yoko Tanaka, Yujiro Maeda, Nobuko Hayashi,
Daichi Chikazu, Mitsuyoshi Iino, Tsuyoshi Takato
Department of Oral and Maxillofacial Surgery, Dentistry and Orthodontics, University of Tokyo Hospital, Tokyo, Japan

Abstract
A 32-year-old woman presented with difficulty in swallowing and swelling of the tongue. A fistula was
seen on the dorsum of the tongue. As T2-weighted magnetic resonance imaging showed a high signal
intensity lesion with poorly defined margins, a provisional diagnosis of tongue lipoma or angiomyolipoma was made. The lesion was surgically removed including the fistula. Sebaceous glands were seen
in the epithelial lining on histopathological analysis, which suggested a dermoid cyst. The patients
postoperative course was satisfactory, and no recurrence has been observed during 18 months of
follow-up after surgery.
Key words: Angiomyolipoma, Dermoid cyst, Fistula, Lipoma, Sebaceous glands

Introduction
Dermoid cysts are believed to arise from aberrations of
ectodermal tissue in the foetus or from aberrations in the epithelia due to trauma or surgery after birth. These cysts appear
in various parts of the body, and occurrence in the mouth is
reported to be around 1.6%.1 Patients with dermoid cysts accompanied by fistula on the dorsum of the tongue have only
been reported previously by Rise,2 and Korchin and Juan3
and only 1 such report was from Japan.4 This report is of a
patient in whom fistula formation was seen on the dorsum of
the tongue, which was possibly due to recurrent swelling of
the tongue over a long period.

Case Report
The patient was a 32-year-old woman who presented in
February 2007 with difficulty in swallowing and swelling of
the tongue. From the age of 3 years, the patient had repeatedly experienced swelling of the tongue from an unknown
cause. The swelling had occurred more frequently in recent
years. Magnetic resonance imaging (MRI) revealed a lesion
in the tongue, and the patient was referred to the Department
of Oral and Maxillofacial Surgery, Dentistry and Orthodontics, University of Tokyo Hospital, Tokyo, Japan, for detailed
examination. The patient had no history of oral trauma or
surgery. Systemic findings were normal. Intraorally, a firm
Correspondence: Yoshiyuki Mori, DDS, PhD, Department of Oral and
Maxillofacial Surgery, Dentistry and Orthodontics, University of Tokyo
Hospital, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8655, Japan.
Tel: (81 3) 5800 8669; Fax: (81 3) 5800 6832;
E-mail: mori-ora@h.u-tokyo.ac.jp

2009 Asian Association of Oral and Maxillofacial Surgeons.

swelling was noted on the right side of the dorsum of the

tongue. The surface mucosa of the tongue over the lesion
appeared normal. A tumour was suspected.
As previous T2-weighted MRI showed a high signal
intensity lesion with poorly defined margins, a provisional
diagnosis of tongue lipoma or angiomyolipoma was made.
Tissue biopsy was scheduled, but the swelling located on the
right part of the tongue became red and tender in May 2007.
The lesion was elastic-firm, movable, and had clear margins.
Clinical laboratory tests indicated the presence of inflammation (white blood cells, 7100/NL; C-reactive protein,
4.47 mg/dL). Computed tomography (CT) revealed a cystic
lesion with ring enhancement (24 t 19 mm) in the right deep
portion of the anterior half of the tongue (Figure 1a). The
swelling decreased after antibiotics were administered (intravenous piperacillin sodium 2.0 g/day for 6 days followed by
oral cefditoren pivoxil 300 mg/day for 4 days). MRI taken
after the inflammation had resolved showed a lesion (size, 7
t 6 t 19 mm) with distinct margins and a luminal structure
in the middle of the tongue (Figure 1b).
The lesion was surgically removed after the inflammation
had subsided completely. Before surgery, another elevated
lesion (size, 2 mm in diameter), which had not been noted
previously, was seen on the surface of the dorsum of the
tongue. A pale yellow cystic fluid was expressed from the
dorsum of the tongue when pressure was applied to the floor
of the oral cavity. A probe was inserted through the fistula
and its tip reached the floor of the oral cavity (Figure 2). The
fistula periphery was cut and detached along the fistulous
tract, and a portion of the cyst wall was detected. The lesion
was then approached from the floor of the mouth, the cyst
detached from the surrounding tissue and extirpated entirely,
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Dermoid Cyst with Fistula

Figure 1. Preoperative imaging. (a) Computed tomography during acute inflammation showed a cystic lesion in the center of the tongue
(arrow). (b) T1-weighted magnetic resonance imaging during resolution of inflammation showed a luminal structure in the centre of the
tongue (arrow).

Figure 2. Intraoperative photographs. (a) Pale yellow pus is seen leaking from the fistula on the dorsum of the tongue (arrow). (b) A probe
inserted through the fistula reached the floor of the mouth.

including the fistula. The extirpated specimen was 34 t 12

t 6 mm in size. Histopathologically, the fistulous tract was
found to originate from the tongue surface and was connected to the cystic lesion. The wall of the cystic lesion was lined
with stratified squamous epithelium, and parakeratosis was
seen. Although the contents of the cyst were not confirmed, a
diagnosis of dermoid cyst was made based on the sebaceous
glands observed in the connective wall of the cyst (Figure 3).
The patients postoperative course was satisfactory, and no
recurrence has been observed 18 months after surgery.

Discussion
Dermoid and epidermoid cysts have been reported to occur in all body parts, but occurrence in the oral region is
comparatively rare.5 These cysts are mostly considered to be
congenital, generally produced by aberrant introduction of
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ectodermal tissue into the union of the first and second brachial arches during the foetal period.6,7 In the oral region, the
floor of the mouth is a common site of onset. Cysts occurring
after birth are probably due to epithelial aberration because
of trauma or surgical treatment. Therefore, unlike congenital
cysts, dermoid or epidermoid cysts that are caused by trauma
or surgical treatment often manifest away from the midline.8
It is thus essential that patients in whom cysts are found
away from the midline be interviewed regarding any history
of trauma or surgery. Reports of dermoid cysts accompanied
by fistulae are extremely rare. An extensive literature review
revealed reports of only 2 patients from other countries2,3 and
1 in Japan.4
In the present patient, while recurrent swelling of the
lower part of the tongue had been experienced since a very
young age, the condition was not examined closely or
diagnosed definitively. A fistula was seen in the centre of the
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Mori, Saijo, Fujihara, et al

even if imaging is done at these times. The fistula was not

clear at initial presentation for this patient, but could be identified on the surface of the dorsum of the tongue when inflamed.
Where cysts are accompanied by fistulae, inflammation tends
to recur due to the existence of the fistula, and the cyst may
not enlarge considerably or swelling may stop while still mild
due to drainage through the fistula. As a result, it is difficult to
arrive at a definitive diagnosis for such patients and detection
is delayed. Therefore, special attention should be given during
diagnostic imaging. Malignant transformation of sublingual
dermoid cysts has also been reported,12 and this calls for
careful monitoring during clinical management.

References
Figure 3. Histopathological examination of the extirpated specimen
showed sebaceous glands in the connective wall of the cyst, which
was lined with stratified squamous epithelium (haematoxylin and
eosin; original magnification, 100).

1.

the mouth: diagnostic imaging by sonography, computed tomography

and magnetic resonance imaging. Br J Radiol. 1995;68:205-7.
2.

tongue for this patient. Taking into account the development

of fistulae, as swelling was experienced repeatedly from an
early age, it was assumed that the fistula had formed through
the lingual septum, where the tissue is relatively weak, due
to internal pressures associated with the swelling. Although
dermoid and epidermoid cysts often develop congenitally,
they are rarely discovered in early childhood unless they
become enlarged. Such cysts are commonly detected during
puberty, which may be related to the cysts slow growth,
and the increased functions and enlargement of sweat and
sebaceous glands during puberty. 9 Cysts are sometimes
discovered during this period due to exacerbation by inflammation. Caution is warranted for such patients, as respiratory
difficulties may also occur.10
Meyers classifications are generally used for the definitive diagnosis of these cysts.9 Meyer classified cysts into
3 histological types epidermoid, dermoid, and teratoid.
The lesion was diagnosed as a dermoid cyst in this patient
as histopathological analysis revealed a stratified squamous
epithelial lining and sebaceous glands in the cyst wall.
MRI, CT with contrast, and ultrasound are effective
diagnostic techniques.11 However, in patients with an accompanying fistula, dermoid cysts may be small during noninflammatory periods and therefore may not be observable

Asian J Oral Maxillofac Surg. Vol 21, Nos 1 & 2, 2009

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