p e d i a t r i c d e n t a l j o u r n a l x x x ( 2 0 1 7 ) 1 e6

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Pediatric Dental Journal

journal homepage: www.elsevier.com/locate/pdj

Case Report

Odontoma associated with unerupted primary

tooth in primary dentition e Three cases

Yukiko Takashima a, Yuko Morikawa a, Atsushi Takagi a, Yuki Matsumi a,

Tatsushi Matsumura b, Seiji Iida b, Shuhei Naka a,
Michiyo Matsumoto-Nakano a,*
a
Department of Pediatric Dentistry, Okayama University Graduate School of Medicine, Dentistry and
Pharmaceutical Sciences, Japan
b
Department of Oral and Maxillofacial Reconstructive Surgery, Okayama University Graduate School of Medicine,
Dentistry and Pharmaceutical Sciences, Japan

article info abstract

Article history: Odontomas are generally encountered in permanent dentition and rarely seen in primary
Received 22 August 2017 dentition. These lesions are regarded as essentially benign, though they often cause dis-
Received in revised form turbances in eruption of the associated tooth. We present clinical findings and manage-
29 September 2017 ment of 3 cases of odontomas that occurred in primary dentition, including a 3-year-old
Accepted 3 October 2017 girl referred for examination of an unerupted left upper primary canine, a 6-year-old boy
Available online xxx with an unerupted left lower second primary molar, and a 5-year-old girl who was pre-
sented with an unerupted right upper second primary molar. In all cases, periapical and
Keywords: panoramic radiography revealed a radio-opaque mass surrounded by a narrow radiolucent
Odontoma space located above the crown of the unerupted tooth. Each was diagnosed as a compound
Primary dentition odontoma interfering with eruption of a primary tooth and surgical removal was per-
Primary teeth formed under general anesthesia for all patients, with the final diagnosis based on histo-
logical findings. Following surgery, the primary canine in Case 1 spontaneously erupted,
while a space maintenance appliance was applied in Case 2 and 3. It is important to
recognize that an odontoma can appear in primary dentition, though the incidence is quite
low. Early detection and removal of an odontoma associated with an impacted primary
tooth is necessary for optimal dentition.
© 2017 Published by Elsevier Ltd on behalf of Japanese Society of Pediatric Dentistry.

and cementum [1,2]. Traditionally, odontomas have been

1. Introduction classified as benign odontogenic tumors, which are divided
into compound and complex type based on histological find-
An odontoma is a type of teratoma that develops from tooth ings [3,4]. Compound odontomas consist of multiple
germ dysplasia and is mixed variously with enamel, dentin,

* Corresponding author. Department of Pediatric Dentistry, Okayama University, Graduate School of Medicine, Dentistry and
Pharmaceutical Science, 2-5-1 Shikata-cho, Kita-ku, Okayama, 700-8558, Japan.
E-mail address: mnakano@cc.okayama-u.ac.jp (M. Matsumoto-Nakano).
https://doi.org/10.1016/j.pdj.2017.10.001
0917-2394/© 2017 Published by Elsevier Ltd on behalf of Japanese Society of Pediatric Dentistry.

Please cite this article in press as: Takashima Y, et al., Odontoma associated with unerupted primary tooth in primary dentition e Three
cases, Pediatric Dental Journal (2017), https://doi.org/10.1016/j.pdj.2017.10.001
2 p e d i a t r i c d e n t a l j o u r n a l x x x ( 2 0 1 7 ) 1 e6
miniature or rudimentary teeth, and the complex type ap-
pears as an amorphous and disorderly pattern of calcified
dental tissues [4]. Common sites for the compound type are
related to anterior teeth, while the complex type often appear
in the upper and lower molar tooth areas [5,6].
Odontogenic tumors are quite rare, with an absolute inci-
dence ranging from as low as 0.002%e0.1% [5,7]. In general,
odontomas that develop in younger patients are generally
seen in permanent dentition and rarely in primary dentition
[7]. Although initially asymptomatic, as an odontoma grows,
an eruption anomaly of the affected tooth occurs and the jaw
bone shows bulging, at which point the patient typically visits
a dental clinic and the odontoma is revealed by X-ray exam-
ination [8]. The basic treatment method is surgical removal
[9,10]. Although these lesions are regarded as essentially
benign, an odontoma may occasionally cause disturbances
not only in regard to tooth eruption, such as impaction, or
delayed eruption or retention of primary teeth [11,12], but can
also lead to eruption of the associated tooth [5]. In the present
study, we present clinical findings and management of
odontomas occurring in primary dentition of 3 child patients.
2. Case reports
Case 1. A 3-year-old girl was referred to Okayama University
Hospital for examination of an unerupted left upper primary
canine. At the first intraoral examination, all primary teeth
had erupted except for the left upper primary canine, while
the upper primary front teeth and molars, and lower primary
molars showed decay (Fig. 1). An extraoral examination
revealed no abnormal signs in the gingiva, buccal tissue, or
alveolar bone of the left maxilla. In the panoramic radiograph
images, all permanent tooth germs were present and the
successor tooth germ of the impacted primary canine was
identified (Fig. 2A). Furthermore, periapical and CT imaging
revealed a single radio-opaque structure present above the
crown of the unerupted upper left canine (Fig. 2B). Root for-
mation of the impacted primary tooth was complete at that
time. Therefore, on the basis of the radiographic results, we
diagnosed a compound odontoma interfering with eruption of
a primary tooth. Surgical removal of the tumor was performed
Fig. 1 e Case 1. Intraoral photograph of Case 1 obtained at
first visit. White arrowhead Indicates non-erupted left
primary canine.
Please cite this article in press as: Takashima Y, et al., Odontoma associated with unerupted primary tooth in primary dentition e Three
cases, Pediatric Dental Journal (2017), https://doi.org/10.1016/j.pdj.2017.10.001
under general anesthesia. Thereafter, the patient was fol-
lowed in regular dental appointments and the impacted pri-
mary teeth was kept under observation. At 1 month after the
operation, the impacted upper left primary canine spontane-
ously erupted. Histopathology findings showed a well-defined
tumor mass with the appearance of a supernumerary tooth, in
which dentin and pulp tissue arrangements were the same as
seen in a normal tooth. The histological diagnosis was com-
pound type (Fig. 3).
Case 2. A 6-year-old boy was referred to us with an uner-
upted left lower second primary molar. At the first intraoral
examination, all primary teeth except for the left lower second
primary molar had erupted. Extraoral examination findings
revealed no abnormal signs in the gingiva or alveolar bone of
the left mandibula. In panoramic radiograph and CT images, a
single radio-opaque structure was shown between the crown
of the unerupted lower left second primary molar and the first
molar, while the left lower second primary molar was inclined
in a centrifugal direction in the mandible (Fig. 4A and B). The
germ of the left permanent second premolar was evident in
those images. The first molar was present, though at a posi-
tion lower as compared to the right first molar. Surgical
removal was performed and the left lower primary second
molar was extracted under general anesthesia, with exposure
of the crown of the first molar performed at the same time.
Histologically results of the soft tissue section showed sparse
fibrous connective tissue containing collagen hyperplasia and
a few islands of orthodontic epithelial cells. The mass of hard
tissue was confirmed to be a complex odontoma with the
decalcified section showing dentin and also included dentinal
tubules, enamel matrix, and reduced enamel epithelium
(Fig. 5). Following the operation, a denture was fixed to
maintain the space for the second primary molar.
Case 3. A 5-year-old girl was referred to our hospital with
an unerupted right upper second primary molar. At the first
intraoral examination, the right upper second primary molar
was found to be not erupted (Fig. 6). Extraoral examination
findings revealed no abnormal signs in the gingiva or alveolar
bone of the right maxilla (Fig. 6). In panoramic radiograph
image, there was no germ of the right upper permanent sec-
ond premolar noted, though the results were not clear
(Fig. 7A). In CT images, multiple radio-opaque structures were
observed on the buccal side of the unerupted primary second
molar, while the germ of the right upper permanent second
premolar was clearly observed on the palatal side of the right
upper permanent second premolar (Fig. 7B). Surgical removal
was performed under general anesthesia and exposure of the
crown of the unerupted primary second molar was also per-
formed at that time. Histological results showed irregular
occurrence of enamel, dentin, and pulp tissues in the fibrous
connecting tissues, and led to a diagnosis of complex type
(Fig. 8). At 3 years after the operation, the right upper second
primary tooth spontaneously erupted. Unfortunately, the
germ of the successor tooth was not in a normal position.
3. Discussion
Tooth impaction commonly occurs in permanent dentition,
while that is rarely seen in primary dentition [13,14]. Here, we
 p e d i a t r i c d e n t a l j o u r n a l x x x ( 2 0 1 7 ) 1 e6 3

Fig. 2 e Case 1. A. Orthopantomograph obtained at age of 4 years 3 months. White arrowhead indicates non-erupted
mandibular left second primary molar and a single opaque structure on the upper second primary molar. B. Cone beam
computed tomography image obtained at the age of 4 years 10 months. White arrowhead indicates high-density structure
visible on the palatal side of the crown of the upper left canine.

describe 3 cases of an odontoma in primary dentition associ-

Fig. 3 e Case 1. Histopathological appearance of decalcified
section shown by hematoxylin-eosin staining. Upper
panel shows low magnification, while lower panels show
higher magnification of the area outlined in the low
magnification images.*Pulp-like structure D, dentin-like
structure.
ated with an unerupted primary tooth, with location in both
anterior and posterior teeth. The optimal treatment for an
odontoma occurring in either primary or permanent dentition
is surgical removal. Early diagnosis is important, as sponta-
neous eruption of the impacted tooth can be expected. Several
previous case reports have noted impacted primary molars
that were induced to erupt into alignment in the dental arch
using an orthodontic method [15e17]. In the present Case 1
and 2, the impacted primary tooth erupted spontaneously
after surgical removal of the odontoma was performed.
However, if an impacted primary tooth is diagnosed at a later
stage, dislocation or impaction of the permanent successor
may occur. If so, a combination of surgical and orthodontic
treatments will be necessary. In our Case 3, exposure of the
first molar was performed during the surgical procedure to
remove the odontoma, since the first molar may have been
accelerated to erupt. If the root of the impacted tooth is
halfway to two-thirds formed, the tooth will begin to erupt
and eruption will occur normally, though once root formation
is complete the tooth will lose its potential to erupt [18].
We performed surgical removal under general anesthesia
for each of the present patients, with the final diagnosis based
on histological findings. Early detection and removal of an
odontoma associated with an impacted primary tooth is
necessary for optimal dentition. Generally, adjacent uner-
upted permanent teeth should be preserved, if possible.
However, if those are extremely small, either preservation or
extraction is selected as the patient matures. In addition,
extraction of the adjacent tooth germ is performed extracted
when separation of the dental sac from the odontoma is
difficult.
Fortunately, removal is relatively easy, as they are sepa-
rated from adjacent teeth by the septum bone [19]. Although
odontomas have limited growth potential, they should be
removed due to the presence of various tooth formulations
that can predispose to cystic change, interfere with eruption
of permanent teeth, and cause considerable bone destruction
[20]. When surgical removal is performed close to eruption
time of the first molars, space maintenance is usually
required. In Case 1, the primary canine spontaneously erupted

Please cite this article in press as: Takashima Y, et al., Odontoma associated with unerupted primary tooth in primary dentition e Three
cases, Pediatric Dental Journal (2017), https://doi.org/10.1016/j.pdj.2017.10.001
4 p e d i a t r i c d e n t a l j o u r n a l x x x ( 2 0 1 7 ) 1 e6

Fig. 4 e Case 2. A. Orthopantomograph obtained at the age of 6 years 3 months. White arrowhead indicates non-erupted
mandibular left second primary molar. B. Cone beam computed tomography image obtained at the age of 6 years 3 months.
White arrowhead indicates a low-density structure visible between the mandibular left second primary molar and first
molar.

after surgery and we followed the patient, while a space
maintenance appliance was applied in Case 2 and 3. As for
Case 2, we installed a mandibular denture for space mainte-
nance after 2 months, then after eruption of the first molars,
we attached a lingual arch type space maintainer. In contrast,
we used a band loop space maintainer at 1 year after surgery
for Case 3 to prevent inclination of the first molars. All
patients received regular follow-up examinations to monitor
eruption of the adjacent tooth, including annual panoramic
imaging. Each was followed for more than 2 years.
Histopathological findings of an odontoma show normal
appearing enamel or enamel matrix, dentin, pulp tissue, and
cementum, which may or may not exhibit normal relation-
ship with each other. Complex odontomas consist of a loose
connective tissue capsule consisting of strands or islands of
odontogenic epithelium, while a compound odontoma con-
sists of a number of tooth-like structures enclosed in a fibrous
capsule. Of the cases presented in this study, 2 were complex
and 1 was compound.
In radiographic images, complex odontomas appear as a
spherical, ovoid, or irregular area of radiopacity with a fine
radiating periphery surrounded by a radiolucent zone, while
compound odontomas appear as a collection of tooth-like
structures with variable number surrounded by a radiolu-
cent zone [21]. In each of the present patients, periapical and
panoramic radiographs revealed a radio-opaque mass sur-
rounded by a narrow radiolucent space located above the
crown of the unerupted tooth. An odontoma is a dental con-
dition that often proceeds unrecognized until the occurrence
of clinical symptoms such as delayed eruption. Therefore, its
initial detection is likely accidental when examining radio-
logical findings and the need for routine radiographic exami-
nations must be emphasized.

Fig. 5 e Case 2. Histopathological appearance of decalcified

section by hematoxylin-eosin staining. Upper panel shows
low magnification, while lower panels show higher
magnification of the area outlined in the low magnification
images. Black arrowheads indicate enamel matrix. D:
dentin-like structure.
Fig. 6 e Case 3. Intraoral photograph obtained at first visit.
White arrowhead indicates non-erupted right second
primary molar.

Please cite this article in press as: Takashima Y, et al., Odontoma associated with unerupted primary tooth in primary dentition e Three
cases, Pediatric Dental Journal (2017), https://doi.org/10.1016/j.pdj.2017.10.001
 p e d i a t r i c d e n t a l j o u r n a l x x x ( 2 0 1 7 ) 1 e6 5

Fig. 7 e Case 3. A. Orthopantomograph obtained at the age of 6 years 3 months. White arrowhead indicates non-erupted
maxillary right second primary molar and a single opaque structure on the upper second primary molar can be also seen.
B. Cone beam computed tomography image obtained at the age of 6 years 4 months. White arrowhead indicates a high-
density structure on the labial side of the crown of the upper second primary molar.

In conclusion, it is possible to prevent malocclusion and

induce normal permanent tooth eruption by surgical removal Conflict of interest
of an odontoma occurring in primary dentition. Taken
together, our results show that early diagnosis of an odon- All authors declare no conflict of interest.
toma in primary dentition is essential to prevent later com-
plications, such as impaction or tooth eruption failure. references

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cases, Pediatric Dental Journal (2017), https://doi.org/10.1016/j.pdj.2017.10.001
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