Dermoid Cysts of Floor of the Mouth:

Report of Four Cases

Rephael Zeltser, DMD, Dip Odont (Pretoria), Isaac Milhem, DMD,
Badri Azaz, DMD, and Oscar Hasson, DDS

(E&toria] Comment: The authors have nicely

illustrated their approach to this unusual problem.
Many physicians would use computed tomography
to image these lesions and may opt for an external
approach rather than the transoral approach.)
D e r m o i d cysts, u n u s u a l lesions of u n c l e a r
etiology, are c o n s i d e r e d to be d e v e l o p m e n t a l
anomalies. T h e y m a y be f o u n d at various sites,
but not u s u a l l y on the floor of the m o u t h . F o u r
n e w cases are p r e s e n t e d in this paper. The
clinical, radiographic, histological a n d scanning electron m i c r o s c o p y (SEM) features of
d e r m o i d cyst are described. The a d v o c a t e d
surgical a p p r o a c h is an intraoral t e c h n i q u e
t h r o u g h the lingual f r e n u l u m .
D e r m o i d cysts are p r i m a r i l y f o u n d in the
posterior anal region and ovaries. T h e y o c c u r
less f r e q u e n t l y in the h e a d a n d n e c k region,
a n d the floor of the m o u t h is not a c o m m o n
site for this cyst. A 25-year r e v i e w b y N e w a n d
Erich 1 s h o w e d that, of 1,495 cases, 6.9% (103
cases) of d e r m o i d cysts o c c u r r e d in the h e a d
a n d n e c k region, but o n l y 1.6% (24 cases) w e r e
in the floor of the m o u t h . 1 T h e lesion has also
b e e n f o u n d in several rare s i t e s - - t h e u v u l a 2
a n d the bone. 3
Several theories a t t e m p t e d to explain the
origin of these cysts. Several researchers postulated that d e r m o i d cysts of the floor of the
m o u t h d e r i v e d from e n c l a v e m e n t of epithelial
cells or debris in the m i d l i n e during closure of
the m a n d i b u l a r and h y o i d b r a n c h i a l arches. 4-6
T h e traumatic i m p l a n t a t i o n t h e o r y has b e e n
less a d v o c a t e d because significant injury to
the s u b m e n t a l area is u s u a l l y absent.

From the Department of Oral and Maxlllofaclal Surgery, Hebrew Un=verslty, Faculty of Dental Medicine,
Hadassah Medical Center, Jerusalem, Israel
Address reprint requests to R. Zeltser, DMD, Oral and
Maxllofaclal Surgery, Hadassah Medical Center, POB
12272, Jerusalem, Israel
Copyright 2000 by W.B Saunders Company
0196-0709/2101-0010510.00/0

D e r m o i d cysts of the floor of the m o u t h

u s u a l l y o c c u r in y o u n g adults, especially during the 2nd and 3rd decade, w i t h no sex
p r e d i l e c t i o n 1,5,6,7 but are rarely f o u n d in neonates or infants. 6,8 Clinically, t h e y p r e s e n t as a
slow-growing, painless swelling in the floor of
the m o u t h , u s u a l l y of d o u g h y consistency,
that m a y cause difficulty in eating, speaking,
and, in extreme cases, breathing. 9 Swelling in
the s u b m e n t a l area occurs w h e n the cyst lies
inferior to the g e n i o h y o i d muscle, giving the
patient a d o u b l e - c h i n appearance. Surgical
excision b y an intraoral a p p r o a c h is the treatm e n t of choice. Malignant t r a n s f o r m a t i o n is
unusual. 1 Recurrences, although rare, m a y
o c c u r if the cyst is i n a d e q u a t e l y r e m o v e d . 11

MATERIALS AND METHODS

Four cases of dermoid cyst on the floor of the

mouth were treated at the OMFS Department-Hadassah School of Dental Medicine, Jerusalem,
Israel, during an 18-year period between 1974 and
1992 (Table 1). The average age of patients was
19.75 years, ranging from 19 to 32 years, and the
male:female ratio was 1:3.
The main complaint was a painless swelling on
the floor of the mouth and mildly disturbed speech
function and swallowing, over periods of 9 months
and 16 months, respectively. Three cysts were
located in the midline area of the floor of the mouth
(Fig 1A) and one unilaterally on the left side. In all
cases, the overlying mucosa and salivary flow from
submandibular and sublingual glands were normal.
Extraoral examination showed that all cases presented a soft-tissue distention of the submental
region the so-called "double chin" (Fig 1B, C). The
general medical history was noncontributory, and
regional lymphadenopathy was not present.
Radiographic examination was performed after
aspiration, and a contrast medium was injected into
the submental swelling, showing the extent and the
exact limits of the lesion (Fig 2). The differential
diagnosis was dermoid cyst or plunging ranula.
Under general anesthesia, an intraoral incision was
carried out through the sublingual frenulum and
extended up to the orifices of Wharton's ducts. The

Amencan Journal of Otolaryngology,Vo121, No 1 (January-February),2000 pp 55-60

55

56

ZELTSER ET AL

TABLE1,

Dermold Cysts of the Floor of the Mouth

Cases

Sex

Age
(yr)

Location

Duration
(me)

32

Median

16

Median

19

Lateral

12

20

Median

12

cysts were enucleated completely without rupture

by blunt dissection (Fig 3).
Samples of cyst wall were processed for histological examination with hematoxylin and eosin (H&E)
stains, after fixation in 4% buffered formaldehyde,
and embedded in paraffin. Similar samples were

Signs and Symptoms

Treatment

Painless, mild speech d~sturbance,

double-chin appearance, normal
overlying mucosa, normal sahvary
flow
Painless, d~fflcultym speech, doublechin appearance, normal overlying
mucosa, normal salivary flow
Painless, double-chin appearance,
normal overlying mucosa, normal
sahvary flow
Painless, difficulty in swallowing and
speech, double-chrn appearance,
normal overlying mucosa, normal
sahvary flow

Intraoral approach
General anesthesia

Intraoral approach
General anesthesia
Intraoral approach
General anesthesia
Intraoral approach
General anesthesia

processed for scanning electron microscopy (SEM)

examination as follows: Samples were fixed in 2%
glutaraldehyde for 1 hour, washed in cacody]ate
buffer (0.1 tool/L, PH 7.2), and postfixed in a 1%
osmium teroxide solution in 0.1 mol/L cacodylate
buffer for i hour. This was followed by dehydration

Fig 1. (A) Dermoid cyst located in the anterior floor of

the mouth, while the tongue is displaced upward posteriorly against the palate. Clinical photographs of 2 patients
showing (B) submental mass and (C) "double chin"
profile.

DERMOID CYSTS OF FLOOR OF THE MOUTH

57

through a graded series of Ferron 113 solutions in

absolute ethanol. After triple rinsing in 100% freon,
~' the samples were vigorously shaken in the air for a
few seconds. Under these conditions, the liquid
phase of the freon evaporated rapidly, leaving the

Fig 3. (A) Operative photograph showing delivery of

the cyst through sublingual midline incision. (B) Cystic
specimen delivered intact.

specimens satisfactorily dried. 12 The stubs were

coated with gold using a Polaron ESIO0 sputter
coater (VG Microtech, West Sussex, England). The
specimens were examined with a Philips 505 SEM

Fig 2. (A) Posteroanterior cyst radiograph showing a

"figure of eight" radiopaque lesion, located in the sublingual and submandibular spaces partially separated by
the geniohyoid muscles. (B) Occlusal view cystogram
showing that the lesion is limited to the left side of the
floor of the mouth.

Fig 4. Light microscopy shows cyst wall consisting

of keratinized stratified squamous epithelium, with the
presence of sebaceous gland (arrows) and hair follicle
(arrowhead) (H&E, original magnification x 28).

58

ZELTSER ET AL

(Eindhoven, the Netherlands) at an accelerating

voltage of 20 kV.
Light microscopy showed a cystic structure
bounded by a corrugated, keratinized stratified
squamous epithelium with the lumen packed with
keratin squames. The cystic wall consisted of loose
fibrovascular stroma, scattered infiltrated lymphocytes, and islands of dermal appendages--sebaceous glands and occasional hair follicles and
shafts. Histological examination confirmed the clinical diagnosis of dermoid cyst (Fig 4). The SEM

Fig 6. One-year follow-up of the case in Fig l C

shows normal appearance of the chin-neck profile.

studies indicated the presence of epithelial cells in

an unusual arrangement. The cells are either
rounded or oblong and arranged in clumps. The
cellular surface is either smooth or rough. The
rough epithelial cell surface is characterized by a
ruffle-like structure associated with microvillar rudiments. The cyst lumen contains not only keratin,
but also fiber-like structures in an irregular configuration, possibly collagen fibers.
Below the epithelial cells, there are also the same
fiber-like collagen structures in which a hair shaft
can be clearly identified (Fig 5). Recovery was
usually uneventful, and follow-up for at least one
year showed good restoration of appearance and
function (Fig 6).
DISCUSSION

Fig 5. SEM pictures of the dermoid cyst wall consisting of epithelial cells arranged in clumps (A); rounded or
oblong with smooth or rough surface (ruffleqike structures probably microvillar rudiments (arrowhead) (B);
skin appendage-like hair shafts (C). (Bar = 0.1 mm).

The literature usually classifies dermoid

cysts according to anatomic site and histological characteristics. 5.t3
The anatomic classification is based on the
relation between the cyst and the muscles of
the floor of the mouth. There are basically 3
types. In the median genioglossal or sublingual type, the cyst lies above the geniohyoid
muscle, displacing the tongue superiorly. In
the median geniohyoid or submental type, the
cyst is between the geniohyoid and mylohyoid
muscle, producing a submental bulge. In a
large dermoid cyst, a portion of the cyst may
be located superiorly to the geniohyoid and
the rest inferiorly, giving the lesion a lobutated
appearance (dumbell shape) (Fig 7). The 3rd
type, which is rare, is a lateral cyst that lies
under the mandible.
Histologically, dermoid cysts can be divided
into 3 types: the epidermoid type with the cyst
wall lined by simple stratified squamous epithelium that may be partially keratinized; the

DERMOID CYSTS OF FLOOR OF THE MOUTH

59

I-

Fig 7. Dermoid cyst (DC) located under (A), above (B), and under and above the geniohyoid muscle (C). (1)
Geniohyoid muscle. (2) Mylohyoid muscle. (3) Hyoid bone. (Partly taken from CJT Howell: Oral Surg Oral Med Oral
Patho159"578-580, 1985).

dermoid type, consisting of an epithelial lining with skin appendages such as sebaceous,
sweat glands, and hair follicles in the capsule;
and the teratoid type, with an epithelial lining
and a fibrous capsule containing skin appendages, connective tissue, and derivates such as
bone, muscle, and respiratory and gastrointestinal mucosa.5
SEM studies were initiated to further investigate the unique configuration of the dermoid
cyst wall. The epithelial cells are not arranged
homogenously in a symmetrical ultrastrucrural configuration, unlike the epithelium of
some other odontogenic cysts. ~4 The rufflelike structures associated with microvillar rudiments on the epithelial surface are not found
in odontogenic cyst epithelium.
Swelling of the submental area may occur in
various conditions, such as dental infection;
l y m p h a d e n o p a t h y (tuberculosis, cat-scratch
disease, sarcoidosis, and infectious mononucleosis); neoplastic lesions; benign and malignant salivary gland tumor; cystic hygroma;
lymphomas; and lymphangioma and cystic
lesions (such as plunging ranula, brachial cyst
and thyroglossal duct cyst); and dermoid cyst.
Needle aspiration may be useful in the
diagnosis of the above-mentioned lesions. The

dermoid cyst, in particular, shows a typical

thick, creamy content.
The injection of contrast m e d i u m into the
lesion to determine its anatomic limits is of
value in the diagnosis of cystic type lesions.
Infection of the lesion after injection of the
contrast m e d i u m did not occur in the 4 cases
reported in this article.
The treatment of choice is undoubtedly total
enucleation of the lesion. In our view, the
intraoral approach is the best, giving a good
view of the cyst, easy access, and aesthetic
results, even when the cyst is under the geniohyoid muscle.
ACKNOWLEDGMENT

We wish to thank Prof. J. Lustmann and Dr E

Rahamim for their professional assistance in processing the tissue material for SEM evaluation.
REFERENCES

1. New GB, Erich IB: Dermoid cysts of the head and

neck. Surg GynecolObstet 65.48-55, 1937
2. YoshinaryM, NagayamaM: Epldermoid cyst of the
uvula: Report of a case. J Oral MaxfllofacSurg 44.828-829,
1986
3. LindhC, LarssonA' Unusual jaw-bone cysts. J Oral
MaxillofacSnrg 48'258-263, 1990

60

4 Colp R. Dermoid cysts of the floor of the mouth. Surg

Gynecol Obstet 40:183-195, 1925
5 Meyer I: Dermoid cysts (dermoids) of the floor of
the m o u t h Oral Surg Oral Med Oral Pathol 8.1149-1164,
1955
6 Shafer WG, Hme MK, Levy BM (eds): A Textbook of
Oral Pathology (ed 4). Philadelphia, PA, Saunders, 1983,
pp 78-79
7 Triantafillidou E, Karakasis D, Laskin J: Sweflmg of
the floor of the mouth. J Oral Maxillofac Surg 47:733-736,
1989
8. Gibson WS, Jr, Fenton NA' Congenital subhngual
dermmd cyst. Arch Otolaryngol 108.745-748, 1982
9 Zachariades N, Skoura-Kafoussia C: A life threaten-

ZELTSER ET AL

mg epidermoid cyst of the floor of the mouth' Report of a

case. ] Oral Maxillofac Surg 48:400-403, 1990
10. Tiecke RW. Oral Pathology. New York, NY, McGrawHill, 1965, pp 196-197
11. Blenkmsopp PT, Rowe NL: Recurrent dermoid cyst
of the floor of the mouth Br ] Oral Surg 18.34-39, 1980
12. Bachrach U, Ash I, Rahamim E: Effect of mmroinjected amine and diamine exodases on the ultrastructure
of eukaryotic cultured cells Tissue Cell 19.39-50, 1987
13. Seward GR: Dermoid cyst of the floor of the mouth.
Br J Oral Surg 3.36-47, 1965
14. Philipsen HP, Chan LSC, Reichart PA, et al: Scanrang electron microscopy of odontogenic cyst epithelium.
JDASA 47:219-223, 1992