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Case Reports in Otolaryngology
Volume 2012, Article ID 634949, 4 pages
ACase of SublingualDermoidCyst: Extendingthe Limitsof
Nobuo Ohta, Tomoo Watanabe,TsukasaIto, Toshinori Kubota,YusukeSuzuki,
Department of Otolaryngology, Head and Neck Surgery, Yamagata University Faculty of Medicine, 2-2-2 Iida-nishi,
Yamagata 990-9585, Japan
Correspondence should be addressed to Nobuo Ohta, firstname.lastname@example.org
Received 17 May 2012; Accepted 3 September 2012
Academic Editors: K. Tabuchi and H.-W. Wang
Copyright © 2012 Nobuo Ohta et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
We present the case of a dermoid cyst with an oral and a submental component in a 21-year-old Japanese woman who presented
with complaints of a mass in the oral cavity and difficulty in chewing and swallowing solid foods for about 2 years. MRI shows a
55×65mm well-circumscribed cystic mass extending from the sublingual area to the mylohyoid muscle. Under general anesthesia
and with nasotracheal intubation, the patient underwent surgical removal of the mass. Although the cyst was large and extending
from the surrounding tissues with appropriate traction and countertraction and successfully removed without extraoral incision.
Oral approach in surgical enucleation is useful procedure to avoid cosmetic problems in large and extending mylohyoid muscle
Sublingual epidermoid and dermoid cysts are benign lesions
encountered throughout the body, with 7% occurring in the
head and neck area and 1.6% within the oral cavity [1–
5]. They represent less than 0.01% of all oral cavity cysts
[6–9]. The pathogenesis of midline cysts of the floor of
the mouth is not well established, and dysontogenetic and
thyroglossal anomaly theories have been suggested [1–9]. In
fact, dermoid cysts occur primarily in the oral cavity, and
the most common location in the head and neck is the
external third of the eyebrow [1–9]. Dermoid cysts generally
present with slow and progressive growth, and even if they
of life [7–9]. The treatment of dermoid cysts of the floor of
the mouth is surgical; the approach can be either intraoral
or extraoral, depending on the localization and size of the
mass. Cysts are classified into three types by localization:
(1) sublingual, (2) submental, and (3) submandibular cysts.
Oral approach is usually applied for small sublingual cyst.
The extraoral incision is preferred in submental and large
sublingualcysts.Dermoid cystsusuallypresent earlyin lifeas
bone [1–9]. Such a swelling on the floor of the mouth can
occasionally cause serious problems with swallowing and
speaking [1–9]. Here, we outline a case of giant sublingual
dermoid cyst in a 21-year-old woman that was successfully
removed by oral approach without extraoral incision.
A 21-year-old Japanese woman was referred to our otolaryn-
gology department with the chief complaint of a swelling
below the tongue producing difficulty in chewing and
swallowing of solid foods for about 2 years. Examination
revealed the presence of a solitary midline swelling in the
sublingual region measuring 6 × 5cm. It was nontender,
fluctuant, soft, and nonmobile, and the overlying mucosa
showed no secondary changes. There were no inflammatory
signs or lymphadenopathy associated with the swelling
(Figure 1). Axial and sagittal magnetic resonance imaging
showed that tumor had low signal intensity on the T1-
weighted image and high signal intensity on the T2-weighted
2 Case Reports in Otolaryngology
Figure 1: Clinical pre-operative presentation of a submental swelling.
Figure 2: (a) Axial T1-weighted magnetic resonance imaging (MRI) shows a sharply circumscribed cystic mass. (b) Sagittal-T1 weighted
MRI shows an 55 × 65-mm well-circumscribed cystic mass extending from the sublingual area to the mylohyoid muscle. (c) Sagittal T2-
weighted MRI shows an high-intensity mass extending inferiomedially to the genioglossus muscle and inferiorly to the mylohyoid muscle.
image (Figure 2). Aspiration cytology was performed and
revealed a cheesy material containing numerous non-
nucleated epithelial cells. The aspiration cytology was very
helpful for making a diagnosis of epidermoid or dermoid
with nasotracheal intubation. An intraoral midline incision
from the base of the tongue to the floor of the mouth was
used to access the lesion (Figure 3). Special attention was
paid to the Wharton’s ducts to prevent injury bilaterally.
The cyst was completely exposed, and on evaluation partial
caudal herniation through the mylohyoid muscle was seen.
A combination of sharp and blunt dissection was used to
free the cyst with traction and couter-traction, and it was
delivered intact per os (Figure 3). The wound was closed
in layers and a non-vacuum drain was kept in situ for
24h. Examination with hematoxylin-eosin staining revealed
a cystic lesion with a stratified squamous epithelium lining
and a fibrovascular connective tissue capsule covering the
cystic lumen (Figure 4). These findings are consistent with
a dermoid cyst.
Epidermoid and dermoid cysts of the oral cavity represent
less than 0.01% of all oral cavity cysts [1–9]. Histologically,
this distinction of the cysts in the floor of the mouth was
presented by Meyer in 1955 . The cyst is described
as epidermoid when the lining presents only epithelium,
dermoid when skin adnexa are found, and teratoid when
other tissues such as muscle, cartilage, or bone are present
within the cyst . Dermoid cysts of the floor of the mouth
are disembryogenetic lesions derived from the entrapment
and subsequent growth of epithelial cells during midline
fusion between the first and second branchial arches in the
third and fourth embryonic weeks . Acquired forms
are derived from either iatrogenic or traumatic inclusion of
Case Reports in Otolaryngology3
removal, the specimen measured approximately 5.5 ×5.6 ×4.5cm. (c) Photograph of the dissected cyst shows cheesy, solid material.
Figure 4: Pathological examination showed orthokeratinized strat-
ified squamous epithelium with a flat epithelial-connective tissue
interface lining the cystic cavity, sebaceous glands, and hair, along
with copious sebaceous material. (Hematoxylin and eosin staining,
original magnification ×200.)
epithelium and skin appendages [8–10]. Dermoid cysts are
generally diagnosed in the second and third decades of life
but can present at any age. Congenital cysts of ectodermal
origin are uncommon in the oral cavity (1.6%), and
of the mouth are painless lesions that swell from the anterior
portion of this region. Because these cysts can displace the
tongue, patients usually present with dysphagia, dysphonia,
and dyspnea; in the case of lower localization they present
with a characteristic double chin [1–9]. Anatomically, three
genio-glossal (sublingual), median geniohyoid (submental),
and lateral, according to the anatomic relationship between
the cyst and the muscles of the floor of the mouth [1–10].
The floor of the mouth is the second most common site for
dermoid cysts in the head and neck region after the lateral
The differential diagnosis of sublingual lesions includes
infectious process, ranula, lymphatic malformation, hetero-
topic gastrointestinal cyst, and duplication foregut cyst [1–
9]. For this reason, bimanual conventional radiography is
not always sufficient for making a differential diagnosis.
Computed tomography and magnetic resonance imaging
allow more precise localization of the lesion in relationship
to the geniohyoid and mylohyoid muscles, and they also
enable the surgeon to choose the most appropriate surgical
approach, especially in the case of very large lesions [1–9].
Treatment is by enucleation via an intraoral or extraoral
approach. An intraoral approach is recommended by most
authorsfor sublingual cystsof small or moderate dimensions
(less than 6cm) above the mylohyoid muscle, whereas an
extraoral approach is preferred for larger sublingual cysts
(more than 6cm) [11, 12]. In present case, the cyst was
large (6.5cm) and extending mylohyoid muscle, however, an
intraoral approach was applied to avoid cosmetic problems.
Appropriate traction and countertraction of the cyst might
help to remove the cyst from mylohyoid muscle without
extraoral incision. El-Hakim and Alyamani used an intraoral
approach for large, deep-seated uninfected lesions, obtain-
ing good aesthetics and function [11, 12]. The extraoral
approach is used for very large dermoid cysts affecting
the submandibular and submental spaces and in cases of
infection that could compromise the patient’s airway [11,
Recurrence is very rare with complete excision of the
lesion, but a 5% rate of malignant transformation of oral
4 Case Reports in Otolaryngology Download full-text
dermoid cysts into the teratoid type has been reported in
Appropriate imaging techniques are necessary in the pre-
operative diagnosis of dermoid cysts of the mouth. Oral
approach in surgical enucleation is useful procedure to avoid
cosmetic problems in large and entending the mylohyoid
Written informed consent in Japanese was obtained from
the patient for publication of this paper and accompanying
images. A copy of the written consent is available for review
by the Editor-in-Chief of this journal.
Conflict of Interests
The authors declare that they have no conflict of interests.
N. Ohta used all the data available and wrote majority of
this report. T. Watanabe was the main consultant surgeon
involved in the management of this patient. T. Ito and S.
Kakehata supplied the principles of surgical information
in this article. Y. Suzuki and T. Kubota saw the patient
in hospital and contributed the case history notes used in
this report. A. Ishida reported and provided us with the
histopathological findings and slides. All authors read and
approved the final paper.
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