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Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2013) xxx–xxx

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Journal of Oral and Maxillofacial Surgery,

Medicine, and Pathology
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Case report

Dentigerous cyst associated with a supernumerary tooth in the nasal cavity:

A case report夽
Honoka Kiso a,∗ , Ryokoh Ando b
a
Department of Oral and Maxillofacial Surgery, Graduate School of Medicine, Kyoto University, Shogoin-Kawahara-cho 54, Sakyo-ku, Kyoto 606-8507, Japan
b
Department of Oral and Maxillofacial Surgery, Otsu Red Cross Hospital, Japan

a r t i c l e i n f o a b s t r a c t

Article history: There have been some reports concerning supernumerary teeth located in the maxilla; however, eruption
Received 5 February 2013 into the nasal cavity leading to a dentigerous cyst is quite rare. The authors report a 6-year-old boy
Received in revised form 25 May 2013 who was noticed to have a foreign body in the right nasal cavity during a school physical examination.
Accepted 18 June 2013
There was no subjective nasal symptom, such as bleeding or a feeling of rhinostenosis. On radiological
examination, a tooth-like radiopaque object was noted beneath the surface of the right nasal floor, and
Keywords:
part of a supernumerary tooth in the left maxillary incisor area was also seen. Ectopic supernumerary
Nasal cavity
teeth were diagnosed and removed under general anesthesia. Histopathological examination identified
Supernumerary tooth
Ectopic eruption
the tooth-like structure as a mature tooth, and the isolated tissue in the nasal cavity was coated by soft
Dentigerous cyst tissue, confirmed as a feature of a cyst.
Three years after surgery, the patient was doing well and had healed uneventfully.
© 2013 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.

1. Introduction 2. Case report

The ectopic eruption of teeth occurs in a variety of locations in
the oral cavity. Erupting into the maxillofacial sinus, mandibular
condyle, coronoid process and palate has been reported. Eruption
of teeth into the nasal cavity is uncommon. The first case of inverted
teeth in the nasal cavity was reported by Albinus in 1754 [1].
The majority of cases show significant morbidity such as epistaxis,
paranasal sinusitis, nasal abscess, and nasal oral fistula.
Most cases of intranasal teeth involve a single tooth in the uni-
lateral nose. Inverted teeth in the nasal cavity may originate from
an aberration of the regular dentition or can be supernumerary.
Intranasal teeth are supernumerary, deciduous, or permanent.
An intranasal tooth which forms a dentigerous cyst is quite rare.
This paper reports a case of double supernumerary teeth, one super-
numerary in the right nasal cavity and surrounded by a dentigerous
cyst, and a supernumerary tooth located in the left premaxilla
incisor region.
夽 AsianAOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian
Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathol-
ogy; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese
Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants.
∗ Corresponding author. Tel.: +81 75 751 3404; fax: +81 75 761 9732.
E-mail address: honoka k@kuhp.kyoto-u.ac.jp (H. Kiso).
A 6-year-old boy was introduced to the Department of Otolaryn-
gology of Otsu Red Cross Hospital Japan for intensive examination
because he was noticed to have a foreign body in the right
nasal cavity at the time of physical examination in his elemen-
tary school. Endoscopic examination was performed, and a hard,
tooth-like object was found slightly above the right nasal floor
(Fig. 1A). He was referred to our department for specific treat-
ment.
There was no subjective nasal symptom such as bleeding or rhi-
nostenosis. On inspection, a tooth-like structure covered with the
nasal mucosa and protruding into the right nasal floor was visible.
The surface of the surrounding mucous membrane was smooth,
and no abnormal findings were seen. On intra-oral examination,
Hellman’s Dental Stage II A was present, and neither tooth avulsion
nor loss of a milk tooth was revealed.
On upper occlusal and panoramic radiography, a tooth-like
radiopaque object situated in the vicinity of the right nasal floor was
demonstrated (Fig. 1A and C). Furthermore, a tooth-like radiopaque
object in the left premaxilla incisor region was also shown. There
was no lack of the permanent tooth germ. The left incisor was
slightly lower in comparison with the right incisor. Computed
tomography (CT) scan confirmed an about 10 mm × 12 mm tooth-
like radiopaque object in the right nasal floor and a 7 mm × 12 mm
incisor in the left palate (Fig. 2A and B). It also showed on about

2212-5558/$ – see front matter © 2013 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.ajoms.2013.06.002

Please cite this article in press as: Kiso H, Ando R. Dentigerous cyst associated with a supernumerary tooth in the nasal cavity: A case report. J
Oral Maxillofac Surg Med Pathol (2013), http://dx.doi.org/10.1016/j.ajoms.2013.06.002
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Fig. 1. (A) Endoscopic photography showing a tooth-like structure covered with the nasal mucosa in the right nasal floor. The surface properties of the surrounding mucous
membrane were smooth. (B) Maxillary occlusal radiograph showing two radiopaque tooth-like structures. (C) Panoramic dental radiograph clearly demonstrating tooth-like
radiopaque objects in the nasal cavity on the palatal side of that left incisor.

Fig. 2. (A) Coronal computed tomography (CT) of the paranasal sinuses showing a tooth-like radiopaque object in the right nasal floor. (B) Axial CT with a tooth-like radiopaque
object in the right nasal cavity showing the mass surrounded by a cystic region. (C) Coronal CT of the anterior teeth region showing a tooth-like radiopaque object on the
palatal side of the left incisor. (D) Axial CT with a tooth-like radiopaque object on the palatal side of the left incisor.

Please cite this article in press as: Kiso H, Ando R. Dentigerous cyst associated with a supernumerary tooth in the nasal cavity: A case report. J
Oral Maxillofac Surg Med Pathol (2013), http://dx.doi.org/10.1016/j.ajoms.2013.06.002
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Fig. 3. (A) Three-dimensional CT (3D-T) image showing tooth-like high density in the right nasal floor. It seems to erupt from the maxilla. (B) Multi-planar reconstruction
(MPR) axial reconstruction showing a tooth-like high density in the right nasal floor. (C) An MPR sagittal reconstruction showing a tooth-like structure in the right nasal
cavity.

20 mm × 16 mm well-defined cystic mass surrounding the tooth-
like radiopaque object in the vicinity of the right-side nasal floor
(Fig. 2C and D). For surgical planning, a three-dimensional CT
(3D-T) image was obtained, and we identified that the tooth-
like radiopaque object was approximately 30 mm from the nostril
(Fig. 3A–C).
From the previous examination, we planned to extract the
supernumerary tooth in the right nasal cavity through the right
nostril, and the impacted supernumerary tooth in the palate of
the left incisor area could be extracted with a routine palatal
approach under general anesthesia with oropharyngeal intuba-
tion. In order to prevent isolated hard tissue falling into the
rhinopharynx, a rear nostil balloon was inserted from the left
nostril before the operation. A mucosal elevator was inserted
from the nostril behind the object prominence and we fixed the
object with the elevator avoid falling back. A small incision in
the nasal mucosa overlying the mass was performed, and we
exposed the hard object using a mosquito pean. The hard object
was extracted with a curved mosquito pean through the ante-
rior naris, and the surrounding soft mass was entirely enucleated
(Fig. 4A and B). Then, granulation tissue on the bone of the nasal
floor was removed carefully using a curette. Following surgery,
chitin-coated gauze packing was applied to the right nasal cavity
for hemostasis.
The left supernumerary impacted tooth (mesiodens) of the pre-
maxilla region was extracted without difficulty through a palatal
approach simultaneously.
Three days after surgery, chitin-coated gauze in the right nasal
cavity was removed, and then the patient was discharged. Over
3 years of follow-up, the patient remained asymptomatic and no
nasal mucosal abnormalities were observed.
The supernumerary tooth in the right nasal cavity was an
inverted tuberculate type tooth, 12 mm in length and 9 mm in
diameter, and the supernumerary impacted tooth in the left incisor
region was a non-inverted conical type tooth, 17 mm in length and
7 mm in diameter (Fig. 4C and D).
On histopathological examination of the isolated tissue, both of
the tooth-like structures were identified as mature teeth (Fig. 5A).
In addition, pathological examination of a granulation mass showed
a cyst lined with stratified squamous epithelium. The wall was com-
posed of hyperplasic fibrous tissue with mainly plasma cells and
chronic inflammatory cells (Fig. 5B).
3. Discussion
The eruption of supernumerary teeth into the nasal cavity with
an associated dentigerous cyst is quite rare. A review of the lit-
erature since 1900 disclosed 32 reported cases of dentigerous cyst
associated with supernumerary teeth [2]. Ectopic and supernumer-
ary teeth may present in a variety of locations in and around the
oral cavity. The most common location of supernumerary teeth
is the upper incisor area, known as the mesiodens [3]. Supernu-
merary teeth have been reported to erupt into the maxillary sinus,
mandibular condyle, coronoid process, orbit, palate, nasal cavity,
and through the skin [4]. Single supernumerary teeth occur in 77%
of cases, double supernumerary teeth in 18.4% of cases, and three
or more supernumerary teeth in less than 4.6% of cases [5]. The

Please cite this article in press as: Kiso H, Ando R. Dentigerous cyst associated with a supernumerary tooth in the nasal cavity: A case report. J
Oral Maxillofac Surg Med Pathol (2013), http://dx.doi.org/10.1016/j.ajoms.2013.06.002
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Fig. 4. (A) Photograph of surgical specimen, removed from the right nasal floor. (B) Intranasal tooth with surrounding soft tissue. (C) After removal of the soft tissue
surrounding the tooth. A supernumerary tooth in the right nasal cavity (left) is an inverted tuberculate-type tooth, 12 mm in length, and 9 mm in diameter, and an impacted
supernumerary tooth on the palatal side of the left incisor (right) is a non-inverted conical type tooth, 17 mm in length and 7 mm in diameter. (D) Radiograph of supernumerary
teeth, showing dental pulp cavities.

Fig. 5. (A) Histological section obtained from the supernumerary nasal tooth. (B) Microphotograph showing the cyst wall; parts seemed to be stratified columnar epithelium,
and other parts were covered in stratified squamous epithelium (HE, 40×).

present case involved 2 supernumerary teeth, one had erupted into
the nasal cavity, while the other was in the upper incisor area.
The cause of supernumerary teeth remains unclear, but several
theories are considered. First, a reversion to the dentition of extinct
primates, which had three pairs of incisors [4]. Second, dichotomy
of the tooth primordia has been suggested as a possible etiologi-
cal factor in the development of supernumerary teeth [6]. Third,
it has been suggested that supernumerary teeth are formed as a
result of local, independent, conditioned hyperactivity of the dental
lamina [7]. Recently, there was a report of USAG-1-deficient mice

Please cite this article in press as: Kiso H, Ando R. Dentigerous cyst associated with a supernumerary tooth in the nasal cavity: A case report. J
Oral Maxillofac Surg Med Pathol (2013), http://dx.doi.org/10.1016/j.ajoms.2013.06.002
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with a supernumerary maxillary incisor, which appears to form as
a result of the successive development of the rudimentary upper
incisor tooth [8]. Genetic causes may be considered in supernumer-
ary maxillary incisors. The exact mechanism of tooth eruption into
the nasal cavity is still unclear. Vele et al. [9] reported a case of cal-
cified mucocele of the nasal cavity in 1996, and stated that cystic
lesions in the nasal cavity can originate in the mucosa of the nose
or from adjacent structures such as dental tissues. In most cases,
aberrations of regular deciduous or permanent teeth into the nasal
cavity are related to previous trauma, surgery, cysts, a cleft palate
[10], and anatomical malformation [5,11]. In rare cases, osteomyeli-
tis [12] or childhood infection with syphilis may be associated with
teeth in the nasal cavity [13]. Ray et al. reported an anomalous tooth
projecting from the posterior margin of the right inferior nasal con-
cha, and the posterior margin of the hard palate, and described the
possibility of a developmental origin as there was no evidence of
trauma evident in the bones around the nasal cavity [14]. In the
present case, the patient did not have such a history or anatomical
malformation. There were also no abnormal findings in the denti-
tion or occlusion. This suggests that it is probably developmental
in origin. A defect in migration of the frontonasal neural crest cells
before the end of the fifth embryonic week might cause ectopic
development of tooth germs in the area of the premaxilla, and the
most plausible theory of an inverted tooth is that, during the early
stages of formation of the palate and teeth, the tooth was translo-
cated into the incisive suture and migrated along the suture, an area
of minimal resistance, to emerge in the nasal cavity [15,16]. In the
present case, one supernumerary tooth was found on the mesiolin-
gual side of the left central incisor in the normal direction, and the
other supernumerary tooth was inverted and extended to the nasal
cavity.
Supernumerary teeth are mostly found around the age of 10
years old, being considered that it is the time which corresponds
with permanent-tooth eruption and the start of complaints of
subjective symptoms, and also school physical examination con-
tributes to early detection [17]. The present case did not show any
symptom and was found by chance on a school physical examina-
tion.
Clinical symptoms associated with inverted nasal teeth include
feelings of nasal obstruction, nasal discharge, nasal bleeding, foul
odor, and a foreign body sensation in the nasal cavity [11].
Simultaneously, there are conditions, such as a dull headache
and tinnitus as a rhinogenous reflex symptom [4,18]. Secondary
infection due to foreign bodies in the nasal cavity and fistula for-
mation between the nasal and oral cavities has also been reported
[19,20]. In the present case, there were initially no subjective symp-
toms.
Clinically, an intranasal tooth presents as a white mass with-
out covering by the nasal mucosa or as a protruding reddish mass,
which is completely or incompletely embedded in the nasal mucosa
[10].
Please cite this article in press as: Kiso H, Ando R. Dentigerous cyst associated with a supernumerary tooth in the nasal cavity: A case report. J
Oral Maxillofac Surg Med Pathol (2013), http://dx.doi.org/10.1016/j.ajoms.2013.06.002
5
Intranasal masses are identified clinically, and the differential
diagnosis should include nasal foreign bodies, a rhinolith, bony
sequestra, neoplasm, and exostoses [3,4]. Panoramic X-rays and
CT examinations were mainly used for the differential diagnosis
and confirming the existence of inverted teeth clinically. Computed
tomography provides detailed three-dimensional information on
palatal malformation and a soft tissue mass, and considered more
useful for surgical assessment. Histopathological examination is
necessary for ruling out carcinoma which is described in association
with dentigerous cysts [9].
Hyperplasia of the granulation tissue around a supernumerary
tooth inverted into the nasal cavity may cause marked morbidity of
nose bleeds and infection. Early extraction is recommended even if
there is no symptom.
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