Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 99101

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Asian Journal of Oral and Maxillofacial Surgery

journal homepage: www.elsevier.com/locate/ajoms

Case report

Large epidermoid cyst in lateral oor of mouth and submandibular region

Emiko Tanaka Isomura a,, Yudai Matsuoka b, Munehiro Hamaguchi c, Yumi Yamamoto d, Kouji Yonemitsu e
a

First Department of Oral and Maxillofacial Surgery, Osaka University Graduate School of Dentistry, 1-8 Yamadaoka, Suita City, Osaka, 565-0871, Japan
Department of Oral and Maxillofacial Surgery, Yao Municipal Hospital, 1-3-1 Ryugatyou, Yao City, Osaka, 581-0069, Japan
c
Department of Oral and Maxillofacial Surgery, Yao Municipal Hospital, 1-3-1 Ryugatyou, Yao City, Osaka, 581-0069, Japan
d
Department of Oral and Maxillofaial Surgery, Kojima Central Hospital, 3685 Ogawatyou, Kojima, Kurashiki City, Okayama, 711-0912, Japan
e
Department of Oral and Maxillofaial Surgery, Kojima Central Hospital, 3685 Ogawatyou, Kojima, Kurashiki City, Okayama, 711-0912, Japan
b

a r t i c l e

i n f o

Article history:
Received 25 October 2010
Received in revised form
28 December 2010
Accepted 20 January 2011
Available online 5 March 2011

a b s t r a c t
Epidermoid cysts are infrequently found in the oral and neck region. Among the cases reported, several
noted large epidermoid cysts are located in the oor of the mouth, with a maximum size of about 8 cm.
Herein, we present a case of a large epidermoid cyst sized 11 cm 9 cm 9 cm, which was located in the
oor of the mouth and reached the submandibular area across the mylohyoid muscle.
2011 Asian Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights
reserved.

Keywords:
Epidermoid cyst
Dermoid cyst
Floor of mouth
Submandibular region

1. Introduction
Epidermoid cysts or dermoid cysts, which arise as a result of
failure of the surface ectoderm to separate from the underlying
structures, are lled with a keratinous sebum-like material with
the evidence of skin derivatives. They are rarely found in the oral
and neck region. Typical characteristics of epidermoid cysts or dermoid cysts include slow growth, presentation in early adult life as
asymptomatic swelling that may occasionally cause elevation of
the tongue, interference with speech, and double-chin appearance.
There are several reports of cases of large epidermoid cysts or
dermoid cysts located in the oor of the mouth, with a maximum
size of about 8 cm [13]. Herein, we report a patient with a large
epidermoid cyst located in the oor of the mouth that reached to
the submandibular area.
2. Case report
A 77-year-old Japanese female was referred to Yao Municipal
Hospital for painless swelling in the oor of the mouth and left submandibular area on May 18, 2009. The patient had noticed swelling
in the oor of the mouth about 10 years previously, but did not

Corresponding author. Tel.: +81 6 6879 2936; fax: +81 6 6976 5298.
E-mail address: tanaemi@dent.osaka-u.ac.jp (E.T. Isomura).

seek medical attention. The lesion had gradually increased in size,

extending to the submandibular area, and she began to have difculties while speaking because of the elevation of tongue. We
observed a large submandibular swelling and a large mass in the
oor of the mouth, while the tongue elevation was also noticeable
(Figs. 1 and 2). The lesion was uctuant on palpation but not tender, and no lymphadenopathy was noted. The patient had no other
signicant medical history.
Computed tomography (CT) revealed a cystic lesion sized about
11 cm 9 cm 9 cm in the oor of the mouth that extended to the
submandibular area across the mylohyoid muscle (Fig. 3). An axial
CT section showed that the mass caused the airway to become narrowed, a coronal CT section showed that the mass swelled greatly,
and a sagittal showed that the mylohyoid muscle could be faintly
observed. No other abnormalities were noted in clinical examinations.
The cyst was removed through an extraoral approach under
general anesthesia on June 5, 2009. During the procedure, a sickleshaped skin incision was made two ngerbreadths inferior to the
angle of the left mandible, then the platysma was divided and the
cystic wall was located. Next, the inferior border of the cystic wall
was separated from surrounding tissue, then the anterior border
and posterior border of the wall were also separated. Once, dissection of the inferior pole was completed, cystic uid was aspirated to
reduce the mass of the lesion and facilitate control of the superior
pole of the cyst. The cystic brous attachment to the mylohyoideus

0915-6992/$ see front matter 2011 Asian Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.ajoms.2011.01.004

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E.T. Isomura et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 99101

Fig. 1. Pre-operative photograph showing a submental mass: (A) a view from patients front side; (B) a view from patients left side.

Fig. 2. Intraoral photograph of mass in left oral oor.

muscle was released, after which the cyst was completely freed and
removed. The inside of the cyst was lled with brown muddy uid
(Fig. 4).
Histological examination ndings revealed that the cyst consisted of stratied squamous cell epithelium with keratin piling

Fig. 4. Photograph of surgical specimen. The inside of the cyst is full with brown
muddy uid.

into the lumen, which were consistent with an epidermoid cyst.

The underlying connective tissue contained blood vessels, brous
tissue, and inammatory cells (Fig. 5).
Following the operation, difculty while speaking due to tongue
elevation disappeared. There was no evidence of recurrence after 1
year.

Fig. 3. Computed tomography (CT) revealed the cystic lesion whose size is 11 cm 9 cm 9 cm for both the oor of the mouth and the submandibular area across the
mylohyoid muscle: (A) an axial section at the level of the submandibular area; (B) a coronal section at the mandibular ramus (arrow shows the mylohyoid muscle); (C) a
sagittal section at the left side of oral oor.

E.T. Isomura et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 99101

Fig. 5. Histological nding. The cyst is consisted of stratied squamous cell epithelium with pilling of keratim into the lumen. The underlying connective tissue
contains blood vessels, brous tissue and inammatory cells (HE, 400).

3. Discussion
Among the cases reported, several noted large epidermoid cysts
that exist at midline such as submental or sublingual space, but
a lateral large epidermoid cyst like our case was not reported. In
a lateral case, it is considered that it is easy to be discovered at
the early stage before it becomes large because the face becomes
asymmetry. In the present case, the patient had noticed the mass
previously, but did not seek medical attention. Over the time, the
mass enlarged, and nally made speaking difcult and narrowed
the airway. A previous case study reported a female who noted a
cystic mass over a period of 9 years, however, its size was only
4 cm 5 cm [4]. The present patient had noticed the swelling about
10 years prior. When considering its nal size, we thought that she
should have noticed it earlier.
Classication of cysts is commonly performed based on histological ndings or location [5,6]. Historically, the generic term
dermoid cyst has been used to describe 3 histologic varieties, epidermoid cyst (lined with simple squamous epithelium
with a brous wall and no adnexal structures), true dermoid
cyst (an epithelial-lined cavity with keratinization and with skin
appendages), and teratoid cyst (lined with a range of epithelia, from
simple squamous epithelium to ciliated respiratory type, containing derivatives of ectoderm, mesoderm, and endoderm). However,
Teszler et al. proposed a comprehensive anatomo-surgical classication: suprageniohyoid, infrageniohyoid, and sublingual, which
are 3 types of supramylohioid cysts (intraoral or sublingual),
submental and submandibular, which are 2 types of inframy-

101

lohyoid cysts (cervical), submental transmylohyoid, and lateral

peri-mylohyoid and transmylohyoid, which are 2 types of peri- and
transmylohyoid cysts (dual intraoral and cervical) [6].
Recently, intraoral approaches have been commonly performed
to remove a dermoid cyst, while there is also a report of a median
glossotomy technique that was found useful for cases of suprageniohyoid cysts [2]. However, several studies have noted that an
extraoral incision is necessary when the cyst is under the geniohyoid muscle or mylohyoid muscle [2,5]. The present case was a
lateral perimylohyoid or transmylohyoid cyst whose origin might
have been the sublingual space, and the operation was performed
under an extraoral approach. We also reduced the mass by aspiration of the cystic uid following dissection of the cyst wall, due
to its large size. This technique was reported by Di Francesco et al.
and found to make removal of such cysts easier [7].
In this case, the patient did not complain of dyspnea but CT section showed clearly that the mass caused the airway to become
narrowed. It is necessary to remove before a cyst provokes an
episode of airway narrowing, while an extreme case like this is
rare. Additionally, epidermoid cysts and dermoid cysts are common benign lesions, though a few reports have found that they can
transform into malignant tumors [812]. Thus, it is important to
remove a cyst before the opportunity for malignancy.
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