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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 11  |  Issue : 1  |  Page : 60-63

Central odontogenic fibroma: A case report with histopathological differential diagnosis


Department of Oral Pathology and Microbiology, King George's Medical University, Lucknow, Uttar Pradesh, India

Date of Submission09-May-2020
Date of Decision05-Oct-2020
Date of Acceptance20-Apr-2021
Date of Web Publication9-Aug-2021

Correspondence Address:
Dr. Jaya Singh
Department of Oral Pathology and Microbiology, King George's Medical University, Lucknow, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohs.ijohs_14_20

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  Abstract 


Odontogenic fibromas are one of the infrequent neoplasms seen in the oral cavity with a prevalence rate of 0.1%. They are derived from the odontogenic ectomesenchyme with diverse histopathology. Found most commonly in the anterior maxillary region, these lesions have a female predilection. Odontogenic fibromas are treated by enucleation followed by extensive curettage. The cognizance of the unique clinical, radiographic, and histopathological features is very important to conclude a confirmatory diagnosis. This article reviews a case of central odontogenic fibroma which presented in our department and also elucidates the diagnostic criteria of the same.

Keywords: Calcification, ectomesenchyme, female, fibroma, odontogenic, World Health Organization


How to cite this article:
Singh J, Singh S, Chandra S. Central odontogenic fibroma: A case report with histopathological differential diagnosis. Int J Oral Health Sci 2021;11:60-3

How to cite this URL:
Singh J, Singh S, Chandra S. Central odontogenic fibroma: A case report with histopathological differential diagnosis. Int J Oral Health Sci [serial online] 2021 [cited 2021 Nov 28];11:60-3. Available from: https://www.ijohsjournal.org/text.asp?2021/11/1/60/323524




  Introduction Top


The World Health Organization (WHO), on the basis of biological characteristics has divided odontogenic tumors into two categories: malignant and benign.[1] Odontogenic fibroma has been classified under the category of benign odontogenic tumor of mesenchymal origin. In 1991, Handlers et al. characterized central odontogenic fibroma (COF) as an extremely rare lesion.[2] COF is an uncommon benign neoplasm composed by varying amounts of inactive-looking odontogenic epithelium embedded in a neoplastic mature and fibrous stroma.[3] COF has a female predilection, and around 45% of the cases are found in the maxilla.[4] It appears as an asymptomatic lesion causing expansion of the maxillary and mandibular cortical plates.[5],[6] Radiographically, they tend to be well-defined, unilocular, radiolucent lesions often associated with the peri-radicular area of erupted teeth. Larger cysts might become multilocular and around 12% of the COF may show radiopaque flecks.[4] The aim of this case report is to present a case of COF and to compare its clinical, radiographic, and histopathological features with the present literature; and thus, significantly add to the same.


  Case Report Top


A 32-year-old male reported with a chief complaint of a swelling in the left back tooth region since a year. The swelling gradually progressed to the current size. The patient had no significant family medical and habit history. There was no extraoral swelling seen neck region, and the lymph nodes were nonpalpable.

On intraoral examination, the unilateral swelling was extending from mandibular second molar ascending up to the ramus region. It was red in color, and the margins were indistinct. On palpation, the lesion was tender, and no discharge was seen on compression. The lesion was surgically excised and sent for the histopathological examination. The clinical differential diagnosis includes odontogenic keratocyst, ameloblastoma, odontogenic myxoma, ameloblastic fibroma, and central giant cell tumor.

The radiographic examination revealed an ill-defined radiolucency extending from the mandibular second molar to the ramus of the mandible. The third molar in the region was impacted. Owing to its location and radiographic picture, a provisional diagnosis of unicystic ameloblastoma or ameloblastic fibroma was given.

On gross examination, multiple soft- and hard-tissue specimens were received collectively measuring 5.5 cm × 3.5 cm × 2 cm in size, creamish to grayish white in color, irregular in shape, smooth surface, and firm in consistency [Figure 1]. The cut surface of the largest bit was glistening in appearance with the epithelium thrown into folds. Two hard tissue resembling like teeth with some tissue attached to the cervical part was also received. All the soft-tissue specimen were sectioned, and all the bits were kept for routine processing. The teeth specimen was preserved.
Figure 1: Showing the received gross specimen

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The histopathological examination revealed strands of odontogenic epithelium in a densely fibrous, moderately cellular, and focally myxoid connective tissue stroma [Figure 2]. Few odontogenic islands showed peripheral layer of cuboidal cells with hyperchromatic nuclei and reversal of polarity at focal areas [Figure 3]. The central angular-shaped cells of these odontogenic islands were loosely cohesive resembling stellate reticulum. The connective tissue stroma was moderately dense with plump, oval to spindle shaped cells in mature collagen tissue. Focal areas showed calcified cementum-like material along with normal bony trabeculae with osteoblastic lining and osteocytes within [Figure 4]. Few engorged blood vessels and inflammatory cells chiefly composed of neutrophils were seen too.
Figure 2: Strands of odontogenic epithelium in a densely fibrous, moderately cellular and focally myxoid connective tissue stroma along with two calcified foci of osteoid (H and E, ×10)

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Figure 3: Odontogenic islands showing peripheral cuboidal cells and central angular stellate reticulum like cells with matrix formation (H and E, ×40)

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Figure 4: Calcified cementum like material seen in the connective tissue stroma (H and E, ×10)

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  Discussion Top


COF is a very rare odontogenic neoplasm accounting for 0%–5.5% of odontogenic tumors.[7] It has a slow, persistent growth leading to asymptomatic buccal or lingual plate expansion.[8] It is a mesenchymal odontogenic tumor showing increased predilection for maxilla. It is seen more commonly in females. Smaller lesions have a unilocular well-defined radiographic presence, but large lesions may depict multilocularity. Earlier in 1980, COF was classified into simple type and WHO type by Gardner. The simple odontogenic fibroma has stellate fibroblasts arranged in a whorled pattern, odontogenic islands may or may not be present with foci of dystrophic calcifications seen occasionally.[4] The COF, WHO type, is complex with odontogenic epithelium seen in long strands or nests in a fairly cellular fibrous connective tissue matrix [Table 1]. The fibrous component may vary from myxoid to densely hyalinized. Calcifications composed of cementum-like material or dentinoid are seen too.[4] All these features are present in our case.
Table 1: Classification of odontogenic fibroma

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Later, the odontogenic fibroma was classified in 2005 by the WHO into WHO type and non-WHO type [Table 1].

Sometimes, a COF can also occur in concurrence with giant cell granuloma like lesion. These are termed as collision tumors. Variants of COF with ossification and with amyloid-like protein deposits have also been mentioned in the literature.[10] Recurrence of COF is relatively uncommon with a negligible tendency to undergo malignant transformation.[11]

The pathogenesis of COF is abstruse due to its low occurrence. Chandrashekar et al. found a case of COF with mast cells present, thereby highlighting the role of C-kit in the pathogenesis.[12] Earlier in 1984, Dunlap and Barker also had postulated that the development of COFs can be attributed to neural crest cells.[13] In the year 2016, Wang et al. derived a theory of how an active EMT may aid in the acquisition of epithelial stem-like properties for epithelial rests and contributes to the overgrowth of mesenchymal stromal tissues, and hence, the aggressive advancement of COF.[14]

Histopathological differential diagnosis of COF includes desmoplastic fibroma, ameloblastic fibroma, desmoplastic ameloblastoma, and odontogenic myxoma[15] [Table 2].
Table 2: Differential diagnostic characteristic[4]

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An absolute distinction is mandatory from desmoplastic fibroma and myxoma because they have a higher recurrence rate as compared to COF. Apart from its histopathological classification into simple and complex/WHO types, there are no clear clinical or radiographic diagnostic guidelines to reach a definitive diagnosis of COF.[16]

COF responds well to surgical enucleation[17] with no tendency to undergo malignant transformation.[18] Even though COFs are not invasive and have minimal tendency to recur, complete enucleation is advised. It usually has a good prognosis.


  Conclusion Top


Odontogenic fibromas are rare odontogenic tumors with an incidence rate of 0.1%–1.5% of all odontogenic tumors and 6.1% if odontoma is excluded.[19] Due to its rare occurrence, these lesions may often be misdiagnosed with a periapical or a malignant lesion. It characteristically presents as a palatal soft-tissue depression of the affected tooth.[8] COFs have peculiar histopathologic features which should be probed in depth before diagnosing any such neoplasm. Our case showed basophilic cementum-like calcifications and remnants of odontogenic epithelium in a highly cellular fibroblastic connective tissue background, this differentiated our case into COF separating it from other odontogenic lesions. Even though COFs have low recurrence and good survival rate, these lesions also demand apt diagnosis. This case report elucidates the importance of the histologic diagnosis of any neoplasm apart from their routine clinical and radiographic diagnosis and also how an oral pathologist could help better in diagnosing a lesion in the oral cavity with specific features.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Self-funded.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Soluk-Tekkeşin M, Wright JM. The World Health Organization Classification of Odontogenic Lesions: A Summary of the Changes of the 2017 (4th) Edition. Turk Patoloji Derg 2018;34. doi: 10.5146/tjpath.2017.01410. PMID: 28984343.  Back to cited text no. 1
    
2.
Hedge U, Rekh M. Central odontogenic fibroma of maxilla – A case report. Int J Appl Dent Sci 2015;1:05-7.  Back to cited text no. 2
    
3.
Brazão-Silva MT, Fernandes AV, Durighetto-Júnior AF, Cardoso SV, Loyola AM. Central odontogenic fibroma: A case report with long-term follow-up. Head Face Med 2010;6:20.  Back to cited text no. 3
    
4.
Neville BW, Damm DD, Allen CM, Bouquet JE. Oral and Maxillofacial Pathology. 2nd ed. Philadelphia: W.B. Saunders; 2001.  Back to cited text no. 4
    
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Khajeh Ahmadi S, Rahpeyma A. Central odontogenic fibroma. Iran J Pathol 2013;8:131-4.  Back to cited text no. 5
    
6.
Chhabra V, Chhabra A. Central odontogenic fibroma of the mandible. Contemp Clin Dent 2012;3:230-3.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Mosqueda-Taylor A, Martínez-Mata G, Carlos-Bregni R, Vargas PA, Toral-Rizo V, Cano-Valdéz AM, et al. Central odontogenic fibroma: New findings and report of a multicentric collaborative study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:349-58.  Back to cited text no. 7
    
8.
Seo YK, Kang JH, Lee SR, Choi YS, Hwang EH, Oh SH. Diagnostic considerations in central odontogenic fibroma of the maxilla: 2 case reports. Imaging Sci Dent 2019;49:229-34.  Back to cited text no. 8
    
9.
Eversole LR. Odontogenic fibroma, including amyloid and ossifying variants. Head Neck Pathol 2011;5:335-43.  Back to cited text no. 9
    
10.
Veeravarmal V, Madhavan RN, Nassar MM, Amsaveni R. Central odontogenic fibroma of the maxilla. J Oral Maxillofac Pathol 2013;17:319.  Back to cited text no. 10
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11.
Santoro A, Pannone G, Ramaglia L, Bufo P, Lo Muzio L, Saviano R. Central odontogenic fibroma of the mandible: A case report with diagnostic considerations. Ann Med Surg (Lond) 2016;5:14-8.  Back to cited text no. 11
    
12.
Chandrashekar C, Sen S, Narayanaswamy V, Radhakrishnan R. A curious case of central odontogenic fibroma: A novel perspective. J Oral Maxillofac Pathol 2018;22:S16-9.  Back to cited text no. 12
    
13.
Dunlap CL, Barker BF. Central odontogenic fibroma of the WHO type. Oral Surg Oral Med Oral Pathol 1984;57:390-4.  Back to cited text no. 13
    
14.
Wang S, Xu Q, Alawi F, Zhang Q, Carrasco LR. Central Odontogenic Fibroma of the Mandible-Revisiting Pathogenesis of Benign Tumor of the Jaw. Int J Stem Cell Res Ther 2016;3:043. 10.23937/2469-570X/1410043  Back to cited text no. 14
    
15.
Tandon A, Bordoloi B, Siddiqui S, Jaiswal R. Central odontogenic fibroma with unusual presenting symptoms. Cancer Transl Med 2018;4:167-9.  Back to cited text no. 15
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Zia M, Arshad A, Zaheer Z. Central odontogenic fibroma: A case report. Cureus 2018;10:e2556.  Back to cited text no. 16
    
17.
Venugopal S, Radhakrishna S, Raj A, Sawhney A. Central odontogenic fibroma. J Indian Soc Periodontol 2014;18:240-3.  Back to cited text no. 17
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18.
Whaits E. Essentials of Dental Radiography and Radiology. Edinburgh: Churchill Livingstone; 1992. p. 247-8.  Back to cited text no. 18
    
19.
Pippi R, Santoro M, Patini R. The central odontogenic fibroma: How difficult can be making a preliminary diagnosis. J Clin Exp Dent 2016;8:e223-5.  Back to cited text no. 19
    


    Figures

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

  [Table 1], [Table 2]



 

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