Beruflich Dokumente
Kultur Dokumente
CASE REPORT
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
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.
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
Asian J Oral Maxillofac Surg. Vol 21, Nos 1 & 2, 2009
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).
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