Professional Documents
Culture Documents
Case Report
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
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
G Model
JOMSMP-397; No. of Pages 5 ARTICLE IN PRESS
2 J. Salehinejad et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2015) xxx–xxx
2. Case report
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
G Model
JOMSMP-397; No. of Pages 5 ARTICLE IN PRESS
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
G Model
JOMSMP-397; No. of Pages 5 ARTICLE IN PRESS
4 J. Salehinejad et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2015) xxx–xxx
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-
References
edge the concurrent occurrence of odontoma with COF has not
been reported up till now in the English-language literature [1] Eversole LR. Odontogenic fibroma, including amyloid and ossifying variants.
[23,24]. Head Neck Pathol 2011;5:335–43.
Interestingly, according to documented cases of hybrid/ [2] Allen CM, Hammond HL, Stimson PG. Central odontogenic fibroma, WHO type:
a report of three cases with an unusual associated giant cell reaction. Oral Surg
combined odontogenic tumors in the English-language literature Oral Med Oral Pathol 1992;73:62–6.
CCOT and ameloblastoma tend to be combined with various types [3] Covani U, Crespi R, Perrini N, Barone A. Central odontogenic fibroma: a case
of odontogenic tumors. report. Med Oral Pathol Oral Cir Bucal 2005;10(Suppl. 2):154–7.
[4] Daskala I, Kalyvas D, Kolokoudias M, Vlachodimitropoulos D, Alexandridis
The combination of CCOT with different odontogenic tumors
C. Central odontogenic fibroma of the mandible: a case report. J Oral Sci
including ameloblastoma, ameloblastic fibroma (AF), ameloblastic 2009;51:457–61.
fibro-odontoma (AFO), odonto-ameloblastoma (OA) and AOT has [5] Regezi JA, Sciubba JJ, Jordan RC. Odontogenic tumors. In: Rudolf P, editor. Oral
been reported. In two of these case reports of CCOT, three distinct pathology: clinical pathologic correlations. 5th ed. St. Louis: W.B. Saunders;
2008. p. 286–90.
histologic types were observed [25,26]. [6] Raval N, Mehta D, Vachhrajani K, Nimavat A. Erupted odontoma: a case report.
The combination of ameloblastoma with calcifying odontogenic Diagn Res 2014;8:10–1.
cyst (COC), glandular odontogenic cyst (GOC), odontogenic kera- [7] Cohen DM, Bhattacharyya I. Ameloblastic fibroma, ameloblastic fibro-
odontoma and odontoma. Oral Maxillofac Surg Clin North Am 2004;16:375–84.
tocyst (OKC), AF, AOT and CEOT was also documented as hybrid [8] Vengal M, Arora H, Ghosh S, Pai KM. Large erupting complex odontoma: a case
tumors in literature [18,23]. report. J Can Dent Assoc 2007;73:169–73.
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
G Model
JOMSMP-397; No. of Pages 5 ARTICLE IN PRESS
J. Salehinejad et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2015) xxx–xxx 5
[9] Brannon RB. Central odontogenic fibroma, myxoma (odontogenic myxoma, including an example in a patient with cherubism, and hypotheses on the
fibromyxoma), and central odontogenic granular cell tumor. Oral Maxillofac pathogenesis. Head Neck Pathol 2008;2:333–8.
Surg Clin North Am 2004;16:359–74. [20] Scheifele C. Radiography as an important tool in diagnosing odontogenic
[10] Adalberto MT, Guillermo MM, Roman CB, Pablo AV, Victor TR, Ana María tumors. In: Reichart PA, Philipsen HP, editors. Odontogenic tumors and
CV. Central odontogenic fibroma: new findings and report of a multicen- allied lesions. 1st ed. United Kingdom: Quintessence Publication; 2004. p.
tric collaborative study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 179–88.
2011;112:349–58. [21] Mosqueda Taylor A, Bermúdez Flores V, Díaz Franco MA. Combined central
[11] Bueno S, Berini L, Gay C. Central odontogenic fibroma: a review of the literature odontogenic fibroma and giant cell granuloma like lesion of the mandible:
and report of a new case. Med Oral 1999;4:422–34. report of a case and review of the literature. J Oral Maxillofac Surg
[12] Veeravarmal V, Madhavan RN, Nassar MM, Amsaveni R. Central odontogenic 1999;57:1258–62.
fibroma of the maxilla. J Oral Maxillofac Pathol 2013;17:319. [22] Pushpanshu K, Kaushik R, Punyani SR, Jasuja V, Raj V, Seshadri A. Concurrent
[13] Hara M, Matsuzaki H, Katase N, Yanagi Y, Unetsubo T, Asaumi J, et al. Central central odontogenic fibroma (WHO Type) and traumatic bone cyst: report of a
odontogenic fibroma of the jawbone: 2 case reports describing its imaging fea- rare case. Quant Imaging Med Surg 2013;3:341–6.
tures and an analysis of its DCE-MRI findings. Oral Surg Oral Med Oral Pathol [23] Neuman AN, Montague L, Cohen D, Islam N, Bhattacharyya I. Report of two cases
Oral Radiol 2012;113:51–8. of combined odontogenic tumors: ameloblastoma with odontogenic keratocyst
[14] D’Cruz AM, Hegde S, Shetty UA. Large complex odontoma: a report of a rare and ameloblastic fibroma with calcifying odontogenic cyst. Head Neck Pathol
entity. Sultan Qaboos Univ Med J 2013;13:342–5. 2015 [Epub ahead of print].
[15] Magur VS, Prabhadevi C, Sharma R. Odontoma. A brief overview. J Dent Sci Res [24] Yamazaki M, Maruyama S, Abé T, Babkair H, Fujita H, Takagi R, et al. Hybrid
2011;2:59–61. ameloblastoma and adenomatoid odontogenic tumor: report of a case and
[16] Preetha A, Balikai BS, Sujatha D, Pai A, Ganapathy KS. Complex odontoma. Gen review of hybrid variations in the literature. Oral Surg Oral Med Oral Pathol
Dent 2010;58:e100–2. Oral Radiol 2014;118:12–8.
[17] Reddy GS, Reddy GV, Sidhartha B, Sriharsha K, Koshy J, Sultana R. Large complex [25] Phillips MD, Closmann JJ, Baus MR, Torske KR, Williams SB. Hybrid odontogenic
odontoma of mandible in a young boy: a rare and unusual case report. Case Rep tumor with features of ameloblastic fibro-odontoma, calcifying odontogenic
Dent 2014 [Epub ahead of print]. cyst, and adenomatoid odontogenic tumor: a case report and review of the
[18] Choudhari SK, Gadbail AR. Hybrid odontogenic tumors: a controversy. Pathol literature. J Oral Maxillofac Surg 2010:68470–4.
Oncol Res 2014 [Epub ahead of print]. [26] Zhang W, Chen Y, Geng N, Bao D, Yang M. A case report of a hybrid odontogenic
[19] Tosios KI, Gopalakrishnan R, Koutlas IG. So-called hybrid central odontogenic tumour: ameloblastoma and adenomatoid odontogenic tumour in calcifying
fibroma/central giant cell lesion of the jaws. A report on seven additional cases, odontogenic tumour. Oral Oncol Extra 2006;42:287–90.
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