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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 33  |  Issue : 4  |  Page : 472-475

Hybrid tumour of central ossifying fibroma and complex odontoma in the mandible – A rare case with review of literature


Department of Oral Medicine and Radiology, DY Patil University, School of Dentistry, Navi Mumbai, Maharashtra, India

Date of Submission18-Jun-2021
Date of Decision24-Aug-2021
Date of Acceptance09-Oct-2021
Date of Web Publication27-Dec-2021

Correspondence Address:
Dr. Vasavi K Santosh
Department of Oral Medicine and Radiology, DY Patil University, School of Dentistry, Sector 7, Nerul, Navi Mumbai, Maharashtra - 400705
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_175_21

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   Abstract 


Central ossifying fibroma is a rare, benign fibro osseous lesion that arises from the periodontal ligament. Odontoma is one of the most common odontogenic tumors containing enamel, dentin, cementum, and pulpal tissue. A 15-year-old boy came to our department with the chief complaint of missing teeth and malocclusion. On complete clinical and radiological investigations, we suspected a fibro osseous lesion and an odontoma occurring in the same site in the mandible, which was confirmed histopathologically to be complex odontoma and central ossifying fibroma. The patient was treated with en bloc resection and iliac crest graft. Such hybrid tumors where there is a synchronous occurrence of two tumors have been rarely reported in literature. There has been no established correlation in such cases but reporting more such cases may help in the establishment of a relationship.

Keywords: Case report, central ossifying fibroma, complex odontoma, hybrid tumour, maxillofacial imaging


How to cite this article:
Santosh VK, Hinduja S, Debnath AK, Waghmare M. Hybrid tumour of central ossifying fibroma and complex odontoma in the mandible – A rare case with review of literature. J Indian Acad Oral Med Radiol 2021;33:472-5

How to cite this URL:
Santosh VK, Hinduja S, Debnath AK, Waghmare M. Hybrid tumour of central ossifying fibroma and complex odontoma in the mandible – A rare case with review of literature. J Indian Acad Oral Med Radiol [serial online] 2021 [cited 2022 Jan 23];33:472-5. Available from: https://www.jiaomr.in/text.asp?2021/33/4/472/333864




   Introduction Top


Odontomas are common odontogenic tumors (hamartomas) with a high prevalence in the second decade of life. Complex odontoma (CO) is common in the posterior region of the mandible as an amorphous hyperdense mass, not resembling teeth, with surrounding radiolucent rim. The treatment of choice is usually surgical excision.[1]

Central ossifying fibromas (COF) are benign fibro osseous lesions arising in the skull and facial skeleton, with 10% arising in jaws in the third or fourth decade of life. They have a predilection to occur in females. COFs appear as delineated, unilocular, expansile lesion with variable amounts of radio densities and corticated borders on a radiograph.[2],[3],[4] COFs have tendency for recurrence and malignant transformation; hence, en bloc resection of jaw is generally preferred.

This is a case presentation of a 15-year-old boy with concomitant occurrence of CO and COF in the mandible which is a very rare presentation. The article discusses radiographic features and importance of good diagnostic workup to formulate the best treatment plan.


   Case Report Top


Clinical findings

A 15-year-old male patient reported with chief complaint of malaligned teeth, prominent lower jaw and congenitally missing teeth (36,37). Diffuse non-tender facial swelling was seen on the left lower third of the face without any associated pain or paresthesia. Upon intra oral examination, the edentulous space of 36,37 exhibited a 5 × 2 cm uniformly hard, non-tender swelling, extending from 35 to the pterygomandibular region, causing both buccal and lingual expansion [Figure 1].
Figure 1: (a) Extraoral picture showing diffuse swelling on left lower third of face, (b) intraoral picture showing bucco-lingual expansion with missing 36,37

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Diagnostic assessment

Panoramic image revealed a large, well-defined mixed expansile lesion extending from 35 to the ramus posteriorly, sparing the angle of mandible. Anteriorly, a well-defined hyperdense lesion with an impacted displaced molar was seen. Posteriorly, the lesion exhibited an amorphous mixed lesion, causing expansion of the lower border with another tooth-like structure displaced into the ramus. Right inferior border showed a fracture line apical to 48 probably related to the history of trauma given by the patient [Figure 2]a.
Figure 2: (a) showing Panoramic image, (b) CBCT axial section, (c) CT coronal section, (d) CT axial section

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Cone beam computed tomography (CBCT) of axial section revealed an aggressive lesion with effacement of lingual cortex and computed tomography (CT) scan revealed similar findings [Figure 2]b such as areas of multilocularity in the ramus with no soft tissue changes [Figure 2]c, [Figure 2]d.

Clinico-radiologic correlation affirmed that the lesion had a heterogenous appearance with complex odontoma in the anterior region and characteristics of a fibro osseous lesion in the posterior.

Biopsy 1 from anterior region revealed enamel spaces and dentine interspersed with connective tissue suggestive of complex odontoma. Biopsy 2 from posterior end revealed numerous cementicles, ossicles with osteoblastic rimming, and bony trabeculae in a dense fibrocellular stroma, confirming the diagnosis of ossifying fibroma [Figure 3].
Figure 3: Histopathological features [H and E 10X] (a) showing irregular deposition of dentin, immature enamel interspersed with enamel spaces and scanty connective tissue; (b) showing cementicles, ossicles lined by osteoblasts, and fibrocellular connective tissue with plump spindle cells

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Therapeutic intervention and follow up

En bloc resection with iliac crest graft was carried out under general anesthesia [Figure 4]. After six months of follow-up, patient showed no pain or disturbance in speech and mastication, and no recurrence of the lesion was noticed.
Figure 4: Post -op panoramic radiograph showing en bloc resection and iliac crest graft placement

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   Discussion Top


Literature exhibits several cases of multiple radiopaque and hyperdense lesions of the jaw, especially in Gardner's Syndrome.[4] However occurrence of multiple benign radiopaque lesions outside a syndrome are extremely rare.

COF typically occurs in premolar and molar regions of the mandible in females. Radiographically, they appear as a solitary, well-defined, unilocular, focally expansile lesion with sharp margins, ground-glass attenuation, and low-attenuation halo. Areas of soft-tissue enhancement may be seen, and tooth displacement and erosion are common.[5]

Histologically, COF shows the presence of mixture of woven and lamellar bone and cementum–like materials in a cellular fibrous connective tissue stroma, sometimes with osteoblastic rimming.[6]

COF are seen commonly in the posterior regions of the jaw with impacted teeth and can grow to large sizes. On radiographs, they appear as radiopaque mass consisting of enamel, dentine, and pulp not resembling the morphology of a tooth and are surrounded by a radiolucent zone.[1],[5]

Although odontomas are common benign lesions of the jaws, its co-existence with other odontogenic or nonodontogenic neoplasms is very rare.

[Table 1] depicts the details of cases reported in literature which describe concomitant appearance of odontomas and COF. Most of these cases[1],[4],[5],[7],[8],[9] showed discrete but concomitant appearance of multiple radiopaque lesions which were all seen in the mandible across the age range of 3 yrs to 50 yrs. Some of the cases were also associated with incidence of other disorders like focal cemento osseous dysplasia, periapical cemento-osseous dysplasia and osteoma.[4],[5]
Table 1: Literature review of concommitant cases of odontomes and COF

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Interestingly, there was one case report, similar to the present case, by Bakhtiari et al.[10] where they described compound odontoma and COF in the anterior mandible in a 37-year-old female who presented with an asymptomatic anterior mandibular enlargement and missing canine.

In the present case, a 15-year-old male presented with an asymptomatic enlargement in the posterior mandible and missing molars which was diagnosed as CO and COF.

Though COF is common in females in 3rd or 4th decade of life, in the present case, it was seen in a male patient in the 2nd decade of life. In the case reported by Bakhtiari et al.[10] compound odontoma was seen in a 37-year-old female, much later than its prevalence in the 2nd decade. However, both cases presented with typical features of COF and odontomas radiologically and histologically.

The fact that odontomas and COF develop close to each other in the mandible suggests the possibility of a local environment permissive for growth of multiple odontogenic tumors. Under certain conditions, an immature element of an odontoma may develop into COF.[1]

In addition, COFs are common in jaws due to the large amount of mesenchymal cellular induction into bone and cementum required in odontogenesis. Therefore, when there is an error in the tissue induction process, a COF may form in the jaw bones. Furthermore it is also possible that COF occurs de novo, and due to its high growth potential, the tumor gets overlapped with the odontoma, as seen in the present case and the case reported by Bakhtiari et al.[10]

In this case, the patient reported a history of asymptomatic lesion on his jaw, with bony, hard consistency which suggests a nonaggressive intrabony lesion with simultaneous odontogenic and non-odontogenic lesions, described as combined lesions, or sometimes called hybrid lesions which is extremely uncommon and has been reported in only one paper.[10]

Hybrid tumors are exceptional entities which are composed of two different tumor types, each of which conforms to an exactly defined tumor category. The tumor entities of a hybrid tumor are not separated and have an identical origin within the same topographical area.

The management of both of these lesions is very contrasting: CO requires a simple excision whereas COF requires resection to prevent recurrence. Hence, it is very important to identify and diagnose such exceptions to execute the best treatment for the patient. CBCT and CT images clearly demonstrate the location, extent, and internal characteristics of jaw lesions, thus exemplifying the nature of the lesion, and help in decision making for better and successful outcomes.


   Conclusion Top


The importance of this case lies in its rarity to see the simultaneous occurrence of different tumors within the jaw, and to educate oral and dental surgeons that treatment should be applied to the tumor of the most aggressive type. The relationship between these lesions is not obvious and maybe coincidental. We did not carry out any gene mapping in the patient but it may be interesting to investigate the genetic background of such patients in order to throw light on the occurrence of such hybrid tumors.

Declaration of patient consent

The authors certify that they have obtained appropriate patient consent. The patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and while due effort will be made to conceal his identity, anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Matsuo K, Yamamoto N, Morimoto Y, Yamashita Y, Zhang M, Ishikawa A, et al. Multiple complex odontomas and subsequent occurrence of an ossifying fibroma at the same site as the removed odontoma. J Dent Sci 2013;8:189-95.  Back to cited text no. 1
    
2.
Ram R, Singhal A, Singhal P. Cemento-ossifying fibroma. Contemp Clin Dent 2012;3:83-5.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Mithra R, Baskaran P, Sathyakumar M. Imaging in the diagnosis of cemento-ossifying fibroma: A case series. J Clin Imaging Sci 2012;2:52.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Borghesi A, Tonni I, Pezzotti S, Maroldi R. Peripheral osteoma, compound odontoma, focal cemento-osseous dysplasia, and cemento-ossifying fibroma in the same hemimandible: CBCT findings of an unusual case. Radiol Case Rep 2017;12:756-9.  Back to cited text no. 4
    
5.
Hosseini FA, Moslemi E. Central ossifying fibroma, periapical cemento-osseous dysplasia and complex odon-toma occurring in the same jaw. Clin Pract 2011;1:e36.  Back to cited text no. 5
    
6.
Chang CC, Hung HY, Chang JY, Yu CH, Wang YP, Liu BY, et al. Central ossifying fibroma: A clinicopathologic study of 28 cases. J Formos Med Assoc 2008;107:288-94.  Back to cited text no. 6
    
7.
Ohtake K, Nagamini T, Nakajuma T, Fukushima M. A case of multiple odontomas associated with ossifying fibroma.(In Japanese) Jpn J Oral Maxillofac Surg 1993;39:53-4.  Back to cited text no. 7
    
8.
Lina Z, Ting S, Haoman N, Ning G, Yaling T, Yu C. Mandibular ossifying fibroma and compound odontoma: A case report. Hua Xi Kou Qiang Yi Xue Za Zhi 2016;34:100-3.  Back to cited text no. 8
    
9.
Srivastava P, Sawhney A, Agarwal S, Rathi VC, Dhar S. An unusual association of cemento-ossifying fibroma with an odontoma in mandible: A case report. Int J Oral Facial Surg 2020;1:22-4.  Back to cited text no. 9
    
10.
Bakhtiari S, Mashhadi Abbas F, Mohajerani SH, Mohammad Salehi A, Bakhshi M, Elmi Rankohi Z. Coincidence of compound odontoma and cemento ossifying fibroma; A rare case report. J Dent Sch 2016;34:123-8.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]
 
 
    Tables

  [Table 1]



 

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