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Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2015) xxx–xxx

Contents lists available at ScienceDirect

Journal of Oral and Maxillofacial Surgery,


Medicine, and Pathology
journal homepage: www.elsevier.com/locate/jomsmp

Case Report

Concurrent central odontogenic fibroma (WHO type) and odontoma:


Report of a rare and unusual entity
Jahanshah Salehinejad a , Narges Ghazi a,∗ , Farzin Heravi b , Elham Ghazi c
a
Dental Research Center, Department of Oral and Maxillofacial Pathology, School of Dentistry, Mashhad University of Medical Sciences, Mashhad, Iran
b
Dental Material Research Center, Department of Orthodontics, School of Dentistry, Mashhad University of Medical Sciences, Mashhad, Iran
c
Health Center, Sabzevar University of Medical Sciences, Sabzevar, Iran

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

Article history: Central odontogenic fibroma (COF) has been described as a rare, slow-growing tumor of the jaw with
Received 7 January 2015 odontogenic ectomesenchymal origin. Odontomas are hamartomatous developmental malformations.
Received in revised form 5 April 2015 The occurrence of complex odontomas is rare. These two separate odontogenic tumors have not been
Accepted 10 April 2015
reported previously as occurring simultaneously. Here, we present an unusual case of coincidental co-
Available online xxx
occurrence of COF (WHO type) and two complex odontomas in a 10-year-old boy.

Keywords: © 2015 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.夽
Central odontogenic fibroma
Odontoma
Odontogenic tumor

1. Introduction it might present mixed radiolucent and radiopaque features and


undefined borders. Root resorption and displacement have been
Central odontogenic fibroma (COF) has been described as a rare, reported in cases of more severe lesions [3,4].
slow-growing tumor of the jaw with odontogenic ectomesenchy- According to the WHO classification, COF is classified into
mal origin [1]. Histologically it is characterized by the presence two types: epithelium-poor and epithelium-rich. Odontomas are
of variable amounts of inactive looking odontogenic epithelium known as mixed odontogenic tumors with both epithelial and
scattered within a collagenous fibrous connective tissue [2]. Clini- ectomesenchymal components [5].
cally, the central odontogenic fibroma appears as an asymptomatic The term odontoma was first used by Paul Broca in 1867. In 1946,
expansion of the buccal or lingual cortical plate of the mandible Thoma and Goldman formulated a classification for odontomas [6].
and maxilla with equal frequency. In the maxilla the lesion appears According to World Health Organization (WHO), odontomas are
frequently to involve the anterior region, while the most prevalent classified into compound and complex. In a compound odontoma
area of presentation in the mandible is posterior area, involving the dental tissues are in an orderly pattern resembling tooth like struc-
premolar and molar areas [3]. COF can appear in a wide age group, tures, whereas complex odontoma consists of calcified dental tissue
although it usually develops between the second and fourth decade with irregular pattern [7].
of life. Tumor incidence rate is three times higher in female patients Although the etiology of odontoma is unknown, several the-
than in male patients [4]. ories have been proposed including local trauma during primary
COF radiologically has been described as a uni- or multilocu- dentition, infectious process and genetic mutation anomalies like
lar radiolucencies with well-defined borders. In some rare cases, Gardner syndrome. The remnants of lamina may also be an impor-
tant factor in the etiology of complex or compound odontomas
[6].
Odontomas are commonly asymptomatic. The radiological
夽 Asian AOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian
appearance of odontoma is a well-defined radiopacity situated in
Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathol-
ogy; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese
bone, but with a density that is greater than bone and equal to or
Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants. greater than that of a tooth [8].
∗ Corresponding author at: Dental Research Center, Department of Oral and Max- These two separate odontogenic tumors have not been reported
illofacial Pathology, School of Dentistry, Mashhad University of Medical Sciences, previously as occurring simultaneously. Here, we present an
Vakilalbad Blv., P.O. Box 911735-984, Mashhad, Iran. Tel.: +98 513 8829501;
unusual case of coincidental co-occurrence of COF (WHO type) and
fax: +98 513 8829500.
E-mail address: Ghazin@mums.ac.ir (N. Ghazi). odontoma in a young patient.

http://dx.doi.org/10.1016/j.ajoms.2015.04.002
2212-5558/© 2015 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.夽

Please cite this article in press as: Salehinejad J, et al. Concurrent central odontogenic fibroma (WHO type) and odontoma: Report of a
rare and unusual entity. J Oral Maxillofac Surg Med Pathol (2015), http://dx.doi.org/10.1016/j.ajoms.2015.04.002
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JOMSMP-397; No. of Pages 5 ARTICLE IN PRESS
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2. Case report

A 10-year-old boy presented with a chief complaint of asymme-


try, painless swelling in the right lower face and slow growth of it
during the last year, on October 2013. The patient had no history
of trauma. The patient had visited the physician in the hospital of
his town 1 year before attending our department with a chief com-
plaint of recurring strep throat infections, obstructive sleep apnea
and mild swelling of inferior border of mandible. Tonsillectomy
had been performed for the patient with the diagnosis of recurring
tonsillitis.
Extraoral examination revealed a painless swelling, present on
the right side of mandible extending from midway of the body to Fig. 1. Panoramic radiograph showing opaque lesions in the body and a lucent lesion
the ramus. Overlying skin was normal without any color change in the ramus of the right mandible.
and ulceration.
Intraoral examination revealed a nontender, bony hard swelling fibrous tissue with collagen fibers arranged in interlacing bundles.
of right mandible causing buccolingual expansion extending from Scanty strands and nests of odontogenic epithelium were observed.
right canine to posterior region. Overlying mucosa was normal in The histopathological findings were congruent with a diagnosis of
color and shape. Visible pigmentation, ulcerations or sinus opening central odontogenic fibroma (WHO type) (Fig. 4).
was not observed. Regarding the extension of the lesion and histopathologic evalu-
Preoperative panoramic radiograph demonstrated two opaque ation hemimandibulectomy was performed. The gross examination
lesions in the body and a lucent lesion in the ramus of the right demonstrated buccolingual expansion with hard bony consistency
mandible resulting in bone expansion (Fig. 1). and cortical thinning especially lingual cortex of ramus. Soft tis-
Radioopaque lesions showed irregular masses of calcified mate- sue with elastic consistency in the angle and ramus of mandible
rial with a well-defined margin surrounded by thin radiolucent was seen. Histopathologic evaluation of the lesions in body of the
area. Radiolucent lesion demonstrated large diffuse unilocular area mandible confirmed the diagnosis of complex odontoma (Fig. 5).
with a well-defined corticated border. Computed tomography (CT) The final histopathological diagnosis of two complex odontomas
images showed similar findings to those of the panoramic radio- in the body and a central odontogenic fibroma (WHO type) in the
graph (Figs. 2 and 3). A provisional diagnosis of complex odontoma ramus of right mandible was made.
was made for radioopaque lesions, whereas it was difficult to decide After surgery intraoral and radiographic examination were per-
whether radiolucent lesion was a cystic lesion or tumor. formed (Fig. 6). Maxillomandibular relationship of the patient was
Under local anesthesia, a subperiosteal flap with releasing inci- skeletal Class II.
sion is made at the crest of the alveolar ridge in retromolar region of Hybrid Activator Device is designed to help guide the devi-
right mandible. After removal of cortical bone, an incisional biopsy ated mandibular segment into a more satisfactory relationship
was performed. The specimen was submitted to the University of with the maxilla until the reconstructive phase and to minimize
Mashhad, Department of Oral and Maxillofacial Pathology for histo- mandibular deviation resulting from scar contracture. Follow-up
logic examination. Histopathological examination revealed cellular every 3 months was performed for the patient. Fig. 6 shows the

Fig. 2. (A) CT image. Axial images showing two opaque lesions in the body and a lucent lesion in the ramus of the right mandible (arrows) resulting in bone expansion. (B)
Coronal images showing opaque lesions and a lucent lesion (arrows).

Please cite this article in press as: Salehinejad J, et al. Concurrent central odontogenic fibroma (WHO type) and odontoma: Report of a
rare and unusual entity. J Oral Maxillofac Surg Med Pathol (2015), http://dx.doi.org/10.1016/j.ajoms.2015.04.002
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J. Salehinejad et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2015) xxx–xxx 3

Fig. 3. (A) Nests of odontogenic epithelium with round or cuboidal epithelial cells with bland nuclei (hematoxylin–eosin stain, 100×) and (B) serpentine strands of inactive
odontogenic epithelium surrounded by fibrous tissue (hematoxylin–eosin stain, 100×).

last patient’s follow-up on December 2014. The patient was given in diameter [14]. In the present case each of the odontomas measure
advice that the graft reconstruction will be done after puberty. 3 cm × 3 cm, which is very rare.
The origin of complex odontomas is unknown. Complex odon-
3. Discussion tomas occur in the posterior mandibular region with predilection
for the right side of the jaw [15].
COF is a rare benign neoplasm and somewhat controversial As in the present case, the majority of odontomas are found in
lesion with regard to its inception and definition [9]. It accounts the second decade with equal gender distribution. Although most
for 0–5.5% of all odontogenic tumors [10]. Although OFs can appear of the odontomas are asymptomatic, pain, expansion of the cortical
at any age, it is generally diagnosed in patients in the second and bone, tooth displacement, retention of deciduous teeth, nonerup-
third decades of their life [11]. tion of permanent teeth and swelling in the affected areas may be
The majority of former studies reported a predilection of COF seen [16].
in the female sex [12]; however, equal distribution between males In the present case, the patient presented with swelling and
and females has been also reported. Here, we present a COF in a expansion of buccolingual plate. The radiological appearance of
10-year-old boy. Odontogenic fibromas most commonly occur in complex odontoma is a radiopaque mass which has no tooth struc-
the anterior region of the maxilla and the posterior region of the ture [17].
mandible, similar like the present case in posterior mandible [12]. The occurrence of odontogenic tumors in the jaws can be colli-
Clinically, COFs show asymptomatic slow growth with corti- sion or combined/hybrid. Collision tumors composed of two tumors
cal expansion, similar like our case. Radiographically, conventional of different histogenic source occur at the same location. Hybrid
radiographs and CT images of COF demonstrate uni- or multiloc- tumors have rarely been reported in the jaws. Unlike collision
ular radiolucent areas with well-defined margins [13]. The lesions tumors, they consist of more than one histologic type with an iden-
are associated with the crown of an unerupted incisior, premolar tical origin in the same area [18].
or molar tooth [12]. The great variability of clinical and radiolog- The exact cause for hybrid tumor occurrence is unknown.
ical characteristics that COF can present obliges us to incorporate Odontogenic tumors are derived from odontogenic epithelium
numerous entities in the differential diagnosis including odonto- which has histomorphodifferentiation potential. Therefore, it can
genic cysts and tumors. be explained that pluripotential odontogenic epithelium at the
Odontomas are hamartomatous developmental malformations. same time can induce formation of hybrid tumors [18].
The occurrence of complex odontomas is less common compared The synchronous occurrence of central odontogenic fibroma
to the compound type with a prevalence of 5–30%. Most of the (COF) and central giant cell lesion (CGCL) has been reported [19]. It
complex odontomas reported in previous studies measure 1–2 cm is suggested that this kinship between these two lesions represents

Fig. 4. (A) Hemimandibulectomy was performed for the patient. (B) Photomicrograph showing irregular arrangement of dental tissues. Mature tubular dentin encloses clefts
or circular structures of removed enamel (hematoxylin–eosin stain, 100×).

Please cite this article in press as: Salehinejad J, et al. Concurrent central odontogenic fibroma (WHO type) and odontoma: Report of a
rare and unusual entity. J Oral Maxillofac Surg Med Pathol (2015), http://dx.doi.org/10.1016/j.ajoms.2015.04.002
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Fig. 5. Postoperative (A) radiographic, (B) extraoral and (C) intraoral images.

Fig. 6. Hybrid Activator Device is designed to help guide the deviated mandibular segment (last patient’s follow-up).

a “collision” tumor as a result of synchronous occurrence of COF in In the present case, central odontogenic fibroma and odontoma
the area of giant cell granuloma. It has been also explained that are occurring simultaneously as a hybrid tumor in the same patient.
these unusual lesions can represent either (1) collision tumors, (2) This was an unusual case of co-occurrence of these two lesions
COF with reactive CGCL component or (3) CGCL with an induced which is rare and has not been reported in literature.
COF component [19]. Central odontogenic fibroma (COF) and trau-
matic bone cyst have been also reported by Pushpanshu et al. as a
collision tumor [20–22]. Acknowledgement
The most commonly reported hybrid tumor is calcifying cystic
odontogenic tumor (CCOT) with odontoma followed by calcifying The authors thank Dr. Mohammad Ali Gerayeli for his co-
epithelial odontogenic tumor (CEOT) with adenomatoid odonto- operation.
genic tumor (AOT). Odontoma with AOT has been also documented
as hybrid odontogenic tumor, whereas to the best of our knowl-
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rare and unusual entity. J Oral Maxillofac Surg Med Pathol (2015), http://dx.doi.org/10.1016/j.ajoms.2015.04.002
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Please cite this article in press as: Salehinejad J, et al. Concurrent central odontogenic fibroma (WHO type) and odontoma: Report of a
rare and unusual entity. J Oral Maxillofac Surg Med Pathol (2015), http://dx.doi.org/10.1016/j.ajoms.2015.04.002

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