International Journal of Pediatric Otorhinolaryngology (2008) 72, 12751279

www.elsevier.com/locate/ijporl

CASE REPORT

A floor of mouth teratoid cyst with tract in a

newbornCase report and English literature
review unraveling erroneous quotes and citations
Kenton Gan b,*, Elaine Fung b, Halliday Idikio a,1, Hamdy El-Hakim b
a

Laboratory Medicine & Pathology, Division of Anatomical Pathology, University of Alberta,

5B4.11 Walter C Mackenzie Health Sciences Centre, 8440 112 St., Edmonton, AB, Canada, T6G 2R7
b
Pediatric Otolaryngology, Divisions of Pediatric Surgery & Otolaryngology, University of Alberta, Stollery
Childrens Hospital, 2C3.57 Walter C Mackenzie Health Sciences Centre, 8440 112 St., Edmonton, AB,
Canada, T6G 2R7
Received 5 March 2008; received in revised form 29 April 2008; accepted 30 April 2008
Available online 12 June 2008

KEYWORDS
Dermoid;
Teratoid cyst;
Floor of mouth;
Tract;
Dysontogenetic

Summary Dysontogenetic cysts are thought to fall into one of three classes:
epidermoids, dermoids or teratoids. Floor of mouth teratoid cysts are the least
common presentation reported. Over the last 70 years, fewer than 20 histologically
proven cases have been described in the English literature. We report an infant
presenting with this lesion in association with a midline tract. The cyst was identified
at birth and interfered with feeding. It was surgically excised with no recurrence at 10
month point of follow-up. A literature search revealed that confusing terminology and
indirect quotation disseminated false beliefs regarding the epidemiology. Contrary to
most reports, floor of mouth teratoid cysts are most commonly encountered in
childhood with only a handful of cases in older age groups.
# 2008 Elsevier Ireland Ltd. All rights reserved.

1. Introduction
Dysontogenetic cysts (DC), commonly referred to as
dermoids, are hamartomas which may contain
various derivatives of endoderm, mesoderm and
* Corresponding author at: Division of Pediatric Surgery, 2C3.57
Walter C Mackenzie Health Sciences Centre, 8440 112 St., Edmonton AB, Canada, T6G 2R7. Fax: +1 780 407 2004.
E-mail addresses: kdgan@ualberta.ca (K. Gan),
ewfung@ualberta.ca (E. Fung), hallidayidikio@capitalhealth.ca
(H. Idikio), haelhakim@cha.ab.ca (H. El-Hakim).
1
Fax: +1 780 407 3009.

ectoderm. These lesions most often present in

the testes and ovaries, but are also found in the
head and neck region [1]. They are occasionally
accompanied by sinus tracts or fistulae that are
claimed to develop as a result of secondary infection [2]. DC fall into three classes: epidermoids,
dermoids and teratoids. The latter consist of derivatives of all three tissue types [3] and are the least
encountered of all. Any class of DC can occur at
numerous locations; although rare, the teratoids
have been well documented in the floor of the
mouth location.

0165-5876/$ see front matter # 2008 Elsevier Ireland Ltd. All rights reserved.
doi:10.1016/j.ijporl.2008.04.014

1276
Interestingly, nearly all past reports of floor of
mouth teratoid cysts (FMTC) have perpetuated the
claim that the rule was finding them in adult patients
[38]. Curiously, identifying actual reports, other
than in children, on PubMed proved to be an elusive
task!
We report the youngest case of a FMTC with
accompanying sinus tract in the English literature.
There are another 11 FMTC documented over the
last 50 years, all of which were in children and
four of them associated with sinus tracts [2,911].
An accompanying literature search helps to dispel
a widely disseminated belief regarding its epidemiology.

2. Case report
A 2.5-month-old, ex-premature infant with a large
sublingual cystic lesion was referred to the pediatric
otolaryngology service by his family physician. This
had been noted at birth and caused the tongue to be
displaced superiorly and interfered with feeding.
His physician and another otolaryngologist performed at least four aspirations of thick, mucoid
material only for the cyst to refill time and again.
Clinical examination revealed a mid-line white
cyst in the floor of the mouth posterior to the
submandibular duct openings (Fig. 1a). It did not
extend into the root of the tongue, nor was there
any component in the neck. Nasolaryngoscopy was
performed and showed no airway compromise.
The patient underwent a trans-oral excisional
biopsy under general anesthetic. Using a transverse
mucosal incision, the cyst was dissected and a tract

K. Gan et al.
was identified running inferiorly, in the midline,
between the geniohyoid muscles towards the body
of the hyoid. The cyst was excised completely along
with the tract up to its point of contact with the
body of the hyoid bone. The cyst was opened to
reveal clear gelatinous fluid. The postoperative
period was uneventful and the patient was discharged home the following day. Follow-up till 10
months later demonstrated no recurrence.

3. Pathology
The gross examination revealed a cyst-like structure
measuring 2 cm  1.5 cm  0.5 cm with a wall
thickness of 0.2 cm. The external surface was pink
and smooth, and the internal surface was white and
wrinkled. Microscopy revealed both cystic and keratotic areas lined by squamous mucosa, respiratory
epithelium, and gastric mucosa with an abundant
smooth muscle component and some goblet cell
component, squamous mucosa with attached hair
follicles and skin adnexae. There was inflammatory
infiltrate including neutrophils and a possible microabscess corresponding to the sinus tract (Fig. 1b).
The identified changes were consistent with those of
a mature teratoid cyst.

4. Literature search
A number of PubMed reviews of the English literature were performed to evaluate the number of
cases of the FMTC subclass of DC in both the pediatric and adult populations.

Fig. 1 (a) partially excised midline teratoid cyst in floor of mouth and (b) histology of microabscess corresponding to
tract showing inflammatory infiltrate including neutrophils.

A floor of mouth teratoid cyst with tract in a newborn

1. Searches for teratoid and cyst and mouth
(a) limited to All Child (018): 11 relevant
reports
(b) limited to All Adult (19+): 3 relevant reports
2. Searches for teratoid and mouth and sinus
tract
(a) limited to All Child (018): 1 relevant report
(b) limited to All Adult (19+): no relevant reports
3. Searches for teratoid and mouth and fistula
(a) limited to All Child (018): 2 relevant
reports
(b) limited to All Adult (19+): no relevant reports

5. Discussion
The term dermoid is often utilized to refer to all
types of dysontogenetic known otherwise as developmental cysts [4,12]. However, for clarity and
consistency, the term dysontogenetic cyst (DC)
will be used in this article. It has been hypothesized
that DC of the head and neck form when epithelial
debris is trapped during fusion of the first (mandibular) and second branchial (hyoid) arches [3,12], or
perhaps when epithelial cells are implanted during
trauma [3]. Since cells trapped during embryogenesis may be pluripotent, they may represent any of
the three germ layers [3,4] and resulting lesions
have been described to contain a variable mixture
of their derived mature tissues (skin, hair follicles,
respiratory/gastric epithelium, bones, cartilage,
nervous tissue, blood vessels, and collagen fibres
etc.) [14,6,912].
Meyer [12] proposed the widely used histological
classification for DC based on lining epithelium and
luminal contents: (1) epidermoids epithelial-lined
cavity surrounded by a capsule but without skin
adnexae, (2) dermoids epithelial-lined cavity with
skin adnexae (hair, hair follicles, sebaceous glands,
sweat glands, etc.) present in underlying connective
tissue, and (3) teratoids epithelial-lined cavity
with skin adnexae, connective tissue derivatives
(e.g. fibers, bone, muscle, blood vessels etc.) and
respiratory and/or gastroinstestinal tissues present
in the capsule. Of the three categories, epidermoids
are the most common, and the least being teratoids
[4]. Most DC occur in the testes and ovaries, whereas
only 7% present in the head and neck region [1]. Of
the latter, most are located in the lateral third of
the eyebrow [3], whereas sublingual lesions account
for only 1.6% [1].
FMTC have been documented equally in males
and females. Repeatedly they have been claimed to
affect most commonly those in their second or third
decade of life [38]. It has also been asserted that
FMTC are especially rare in infancy, but we venture
that the reverse is actually true.

1277
Clinically, patients with FMTC may present with a
variety of symptoms, including difficulty feeding or
breathing due to displacement of the tongue [36].
There is often no regional lymphadenopathy or sign
of infection [36], although sinus tracts or fistulas
are argued to be the result of secondary infection.
This is further seen as a rare event [8], as only a
handful of described cases have involved the presence of sinus tracts, most often running towards
the mandible or hyoid regions and all of them in
children [2,911].
When cysts become infected or when waiting
until surgical excision is possible, fine-needle aspiration or incision and drainage may be useful for
temporary relief of symptoms [9], but as rightly
stated in the literature, they will invariably refill
incessantly [57,9]. They may also recur if incompletely excised [8], particularly in cases involving an
accompanying sinus tract or fistula [12]. Therefore,
the definitive treatment for symptomatic FMTC is
complete surgical excision.

6. Review of literature
FMTC are rare in the pediatric population relative to
other pediatric DC; our literature search only found
11 relevant cases, and only five of these were in the
first year of life [47,12]. However, when we
searched for the adult population lesions, we were
surprised to find even fewer relevant results, in
direct contradiction of the common argument that
FMTC are rare in infancy and most common in the
second and third decades of life. A detailed review
of the literature determined that this opinion was
propagated through a series of misquotes of three
key publications by New and Erich [1], Meyer [12],
and Katz and co-workers [13,14].
The first paper by New and Erich [1], in referring
to dermoid cysts of the head and neck, found that
the ages of the patients at the time of operation
varied from 14 months to 72 years, about 60 percent
being between the ages of 15 and 35 years. This
statement was paraphrased by Meyer [12] some 18
years later: there is great variation in reported
age of occurrence of dermoids of the floor of the
mouth; the average lies between 15 and 35 years of
age. In addition, while New and Erich followed
their original statement with the words this, however, does not correspond to any degree to the ages
of the patients at the time when the dermoid first
became noticeable. As may be noted in Table I, 37.2
per cent of the tumors were present at birth,
whereas 62.7 per cent were observed by the fifth
year, Meyers review omitted this important qualification.

1278

K. Gan et al.

Table 1 Case reports describing sublingual teratoid

cysts in children (those associated with sinuses or
tracts are indicated in italics)
Publication
Lima et al. [8]
Kitagawa et al. [2]
Ohishi et al. [11]
Go
l et al. [3]
Faerber et al. [10]
Harada et al. [9]
Babuccu et al. [6]
Nagar et al. [7]
Present report
Bonilla et al. [4]
Meyer et al. [12]
Modolo et al. [5]

Sinus/tract

S
S
S
T

Age at
presentation
12 years
8 years
5 years
4 years
2 years
15 months
7 months
4 months
2.5 months
2 months
Newborn
Newborn

referenced Katz in this manner as well [6], it would

have never occurred to us that the incorrectly
spelled name would in fact lead us to the PubMed
article!
We are content that FMTC are most commonly
found in the infant and child (Table 1). Moreover, as
the number of FMTC in children actually exceeds
that of adults in the literature, the presence of
accompanying tracts no longer appears to be as rare
an event as previously thought. It is our feeling that
blind and second hand quotation is a practice to be
strongly discouraged if we are to avoid the dissemination of wrong information.

Conflict of interest statement

No conflict of interest declared.

Unfortunately, publications by Katz and co-workers [13,14] perpetrated what Meyer had done!
Katzs assertion that most of the patients (60
percent) are under 35 years of age [13] continued
to be quoted further, along with the papers by New
and Erich and Meyer, either independently or
together in various permutations by most recent
publications [38]. This propagated the false notion
that not head and neck DC, but rather FMTC specifically, are most common in the second and third
decades of life, and hence extremely rare in childhood.
King et al. [15] produced a key review in 1994 in
support of our opinion and alluding to the failures of
using non-histological diagnoses. This group
reviewed 141 histologically confirmed cases of floor
of the mouth DC of all three classes. They found that
the greatest incidence (27.6%) of sublingual DC was
in fact in the newborn to 5-year-old range, not
adulthood; they also found that 48.1% (68) of
patients were 15 years or younger, whereas only
38.3% (54) of the cases were within the 16 to 30 year
age period. When it came to FMTC in particular, King
et al. found only 7 histologically verified cases;
together with 8 additional cases which we found
in our literature search, it is likely that there are
fewer than 20 cases of FMTC in the English literature.
Inaccuracies of citation and quotation are not
uncommon in literature. Fenton et al. have estimated that major misquites and citation errors
occur in up to 11.1% and 11.9 % (respectively) of
otolaryngological publications [16]. Not only do
these errors confuse and mislead the reader, they
can also make it difficult to identify particular
articles; for instance, locating Katzs 1969 publication on PubMed was especially difficult because his
name was entered as Ktz. Had another paper not

Acknowledgements
No study sponsors and no sources of funding.

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