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Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 99–101

Contents lists available at ScienceDirect

Asian Journal of Oral and Maxillofacial Surgery

 j o u r n a l h o m e p a g e :   w w w . e l s e v i e r . c o m / l o c a t e / a j o m s

Case report

Large epidermoid cyst in lateral floor of mouth and submandibular region

Emiko Tanaka Isomura a,∗, Yudai Matsuokab, Munehiro Hamaguchic, Yumi Yamamoto d, Kouji Yonemitsue

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

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

Keywords:

Epidermoid cyst

Dermoid cyst

Floor of mouth

Submandibular region

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 floor of the mouth, with a maximum size of about 8 cm.

Herein, we present a case of a large epidermoid cyst sized 11cm × 9 cm×9 cm, which was located in the

floor 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.

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 filled 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 der-

moid 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 floor of the mouth, with a maximum

size of about 8 cm [1–3]. Herein, we report a patient with a large

epidermoid cyst located in the floor of the mouth that reached to

the submandibular area.

2. Case report

A 77-year-old Japanese female was referred to Yao Municipal

Hospital forpainlessswelling in thefloor of themouthand left sub-

mandibular area on May18, 2009. The patient hadnoticed swelling

in the floor 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 dif-

ficulties while speaking because of the elevation of tongue. We

observed a large submandibular swelling and a large mass in the

floor of the mouth, while the tongue elevation was also noticeable

(Figs. 1 and 2). The lesion was fluctuant on palpation but not ten-

der, and no lymphadenopathy was noted. The patient had no other

significant medical history.

Computed tomography (CT) revealed a cystic lesion sized about

11cm× 9 cm ×9 cm in the floor 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 nar-

rowed, 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 examina-

tions.

The cyst was removed through an extraoral approach under

general anesthesia on June 5, 2009. During the procedure, a sickle-

shaped skin incision was made two fingerbreadths 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, dissec-

tion of theinferiorpole wascompleted, cysticfluid wasaspirated to

reduce the mass of the lesion and facilitate control of the superior

pole of the cyst. The cystic fibrous 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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100   E.T. Isomura et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 99–101

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

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

musclewas released,afterwhichthe cyst wascompletely freed and

removed. The inside of the cyst was filled with brown muddy fluid

(Fig. 4).

Histological examination findings revealed that the cyst con-

sisted of stratified squamous cell epithelium with keratin piling

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

muddy fluid.

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

The underlying connective tissue contained blood vessels, fibrous

tissue, and inflammatory cells (Fig. 5).

Following the operation,difficulty 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 floor 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 floor.

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E.T. Isomura et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 99–101   101

Fig. 5.   Histological finding. The cyst is consisted of stratified squamous cell epithe-

lium with pilling of keratim into the lumen. The underlying connective tissue

contains blood vessels, fibrous tissue and inflammatory 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 finally made speaking difficult 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 final size, we thought that she

should have noticed it earlier.

Classification of cysts is commonly performed based on his-tological findings or location  [5,6].   Historically, the generic term

“dermoid cyst” has been used to describe 3 histologic vari-

eties, epidermoid cyst (lined with simple squamous epithelium

with a fibrous wall and no adnexal structures), true dermoid

cyst (an epithelial-lined cavity with keratinization and with skin

appendages),and teratoid cyst (linedwith a range of epithelia, from

simple squamous epithelium to ciliated respiratory type, contain-

ing derivatives of ectoderm, mesoderm, and endoderm). However,

Teszler et al. proposed a comprehensive anatomo-surgical classi-

fication: suprageniohyoid, infrageniohyoid, and sublingual, which

are 3 types of supramylohioid cysts (intraoral or sublingual),

submental and submandibular, which are 2 types of inframy-

lohyoid cysts (cervical), submental transmylohyoid, and lateral

peri-mylohyoid andtransmylohyoid, which are2 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 suprage-

niohyoid cysts  [2].  However, several studies have noted that an

extraoral incision is necessary when the cyst is under the genio-

hyoid muscle or mylohyoid muscle [2,5]. The present case was alateral 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 aspi-

ration of the cystic fluid 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 sec-

tion 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 com-

mon benign lesions, though a few reports have found that they can

transform into malignant tumors  [8–12].  Thus, it is important to

remove a cyst before the opportunity for malignancy.

References

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[3] Yilmaz I, Yilmazer C, Yavuz H, Bal N, Ozluglu LN. Giant submandibular epider-moid cyst: a report of two cases. J Laryngol Otol 2006;20:E19.

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