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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 31  |  Issue : 1  |  Page : 88-91

A diagnostic dilemma of progressive, unilateral mandibular swelling: A mirage unveiled


1 Department of Oral Medicine and Radiology, Darshan Dental College and Hospital, Loyara, Udaipur, Rajasthan, India
2 Department of Oral and Maxillofacial Surgery, Darshan Dental College and Hospital, Loyara, Udaipur, Rajasthan, India

Date of Submission28-Dec-2018
Date of Acceptance12-Feb-2019
Date of Web Publication23-Apr-2019

Correspondence Address:
Dr. Anwesha Bhattacharjee
Darshan Dental College and Hospital, Loyara, Udaipur, Rajasthan - 313 011
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_217_18

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   Abstract 


Ossifying fibroma is a rareentity among the other type of fibro-osseous lesions. It is a non-odontogenic, benign tumor of the jaw, which arises from the multipotential cells of the periodontal ligament. Previously in 1992, WHO classified this type of lesion into ossifying fibroma and cemento-ossifying fibroma. Now the term- “cemento-ossifying fibroma” is replaced with “ossifying fibroma” in recent WHO classification (2005). These tumors are slow growing, painless and occur more commonly in the mandible and in the third and fourth decades of life with slight female predilection. Hereby, in this article, an unusual case of central ossifying fibroma in an 80-year-old female patient with expansile swelling of the right side of mandible since 2 months is repotred. The diagnosis was further confirmed by Cone beam computed tomography (CBCT) and histopathological evaluation. The main objective of this article is to enlighten about the diagnostic dilemma of mandibular swellings and atypical clinical behavior of central ossifying fibroma.

Keywords: CBCT, fibro-osseous lesion, mandible, ossifying fibroma


How to cite this article:
Bhattacharjee A, Bhuvaneshwari S, Khan S, Bulgannawar BA. A diagnostic dilemma of progressive, unilateral mandibular swelling: A mirage unveiled. J Indian Acad Oral Med Radiol 2019;31:88-91

How to cite this URL:
Bhattacharjee A, Bhuvaneshwari S, Khan S, Bulgannawar BA. A diagnostic dilemma of progressive, unilateral mandibular swelling: A mirage unveiled. J Indian Acad Oral Med Radiol [serial online] 2019 [cited 2019 Jun 20];31:88-91. Available from: http://www.jiaomr.in/text.asp?2019/31/1/88/256902




   Introduction Top


Central ossifying fibroma is a benign, nonodontogenic tumor of the jaw bone, which is characterized by painless slow growth and proliferation of cellular fibrous tissue with deposition of osseous, cementum-like material. Despite the fact that it has been classified under fibro-osseous lesions (FOL), still it presumes like a benign neoplasm of bones. In 1972, WHO categorized it into two types – ossifying fibroma and cementifying fibromas; but in 1992, WHO contemplated it in one heading as cemento-ossifying fibroma (COF). As in extent, the term “cemento-ossifying fibroma” was replaced by “ossifying fibroma” in 2005 under the new WHO classification.[1] Branon and Fowler were the first oneto-use the term “ossifying fibroma” in place of “cemento-ossifying fibroma.”[2]

Theories include its origin from the multipotential cells of the periodontal ligament. It usually involves the premolar, molar region of the mandible in the third and fourth decades of life with slight female predilection.[3],[4] Radiographically, the internal structure of the lesion shows either completely radiolucent or radiopaque or mixed radiolucent and radiopaque appearance.[5] Conservative surgical curettage and surgical excision are needed to treat the lesion with good prognosis.[2],[3],[6]


   Case Report Top


An 80-year-old female patient named as Shanta Devi Mehta reported with swelling in the right side of the face since last 2 months. Her history of present illness revealed that the swelling had gradually increased in size with no pain and associated symptoms. The patient gave a history of self-exfoliation of lower right back teeth 5 years back. The past medical history, family history, and personal historywere not contributory. On examination, a diffuse, unilateral extra-oral swelling measuring approximately 6 × 6 cm in size was evident in the right lower one-third of the face. On palpation, the swelling was firm to hard and non-tender. The temperature of the overlying skin was normal like the surrounding skin. [Figure 1] and [Figure 2]. No lymphadenopathy was found. A diffuse swelling was evident intra-orally along right buccal mucosa and premolar, molar region, which was firm to hard in consistency and nontender in palpation. Vestibular obliteration was evident from right lower labial vestibule extending posteriorly till the ramus of mandible [Figure 3]. Expansion of buccal and lingual cortical plate was evident over that region. After correlating history and clinical examination, a provisional diagnosis of the residual cyst was given with differential of ossifying fibroma and ameloblastoma.
Figure 1: Frontal view

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Figure 2: Lateral view

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Figure 3: Intra-oral view

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IOPAR irt 45,46,47 region revealed a diffuse radiolucency in the edentulous region and the presence of a radiopaque mass in 45 region [Figure 4].
Figure 4: IOPAR

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Mandibular cross-sectional occlusal radiograph further revealed discontinuity of the lingual cortical plate [Figure 5].
Figure 5: Mandibular cross-sectional occlusal radiograph

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Orthopantomograph (OPG) showed an extensive mixed radiopaque and radiolucent lesion, involving the right side of the body, angle, and inferior border of the mandible. The internal structure of the lesion showed complete loss of bony architecture with periphery showing wispy and fine trabecular pattern forming loculation. Resorption of the right inferior border of the mandible was evident. A radiopaque area was present in the premolar region suggestive of root stump [Figure 6].
Figure 6: OPG

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In 3D construction view of cone beam-computed tomography, the right body, angle, and ramus of mandible showed hypodense area of an irregular shape. Thinning and resorption of buccal and lingual cortical plate were evident. Coronal view showed that the lesion was from the level of maxillary sinus up to the neck of the condyle. Axial view showed complete resorption of the alveolus, extending inferiorly with complete loss of inferior border of the mandible. A radiodense mass immediately belowthe ridge area in 45,46region was evident. These findings led to a radiographic diagnosis of central ossifying fibroma [Figure 7] and [Figure 8].
Figure 7: CBCT- 3D construction view

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Figure 8: CBCT- Axial view

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The incisional biopsy was performed, and histopathologically, the lesion showed highly cellular connective tissue stroma with many star-shaped and plump fibroblastsin it and also the presence of calcificationsin the form of multiple interconnecting trabeculae of bone. The histopathological findings confirmed the diagnosis of the lesion as ossifying fibroma [Figure 9] and [Figure 10].
Figure 9: Incisional biopsy

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Figure 10: Histopathology

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   Discussion Top


FOL refers to a collection of nonneoplastic intra-osseous lesions that replace normal bone by fibrous tissue, which contains a newly formed, mineralized product. Fibrous dysplasia (FD), cemento-ossifying dysplasia (COD), COF, and their subtypes are included in FOL. Various classifications were proposed to classify these lesions. Recently, the WHO classified COF as a “bone-forming tumor” of jaws. However, the controversy still exists regarding whether the COF, long-held to be a primary bone lesion, should be viewed as a primary odontogenic neoplasm, similar to COD.[2] The etiology and pathogenesis of ossifying fibroma are yet not clearly understood. Odontogenic, developmental, and traumatic causes have been suggested by some authors. The pluripotent mesenchymal cells of the periodontal ligament which have the capacity of producing cementum, osteoid-like materials, are considered as the etiological factors of ossifying fibroma.[3],[7] Mutation in tumor suppressor gene HRPT2, a protein product known as parafibronin which leads to tumor formation or chromosomal abnormalities such as translocation and interstitial deletion of codings in chromosome 2, may also lead to tumor formation.[1]

Review of the literature has revealed that ossifying fibroma presents as a painless, slow-growing mass with marked extra-oral facial asymmetry which was the same in our case.[3],[5] As their growth is relatively slow, the cortical plates of the bone are intact, but in this reported case, extensive resorption of the lingual cortical plate was evident.[3],[8] Ossifying fibroma generally occurs between second and fourth decades of life with female predilection. In our reported case, the patient was much older, an 80-year-old female patient. The premolar, molar region of the mandible is affected more commonly than maxilla, which was same in our case. Teeth displacement and root resorption are the frequent findings, but our patient was completely edentulous.[2],[3],[5]

Radiographically, MacDonald-Jankowski described three stages of Ossifying Fibroma(OF); an initial radiolucent stage, mixed stage, and eventually a sclerotic appearing stage. The lesion may be either unilocular or multilocular. The centrifugal growth pattern is an important diagnostic feature of ossifying fibroma. There are three different types of radiographic borders of ossifying fibroma: defined lesion without sclerotic border, defined lesion with sclerotic border, and lesion with ill-defined border. The last type is seen in rapidly growing tumor. In this reported case, the radiographic and tomographic images revealed the last type of radiographic border.[2],[9]

Histopathologically, OF presents well vascularized fibrocellular connective tissue with irregularly shaped calcifications, which was same in this reported case.[2],[3]

The differential diagnosis of OF may include FD, calcified epithelial odontogenic tumor (CEOT), adenomatoid odontogenic tumor, osteogenic sarcoma, cementoblastoma, and odontoma. In radiographic findings of FD, “ground-glass” appearance is seen. Ossifying fibroma, because of its centrifugal growth, alters bone morphology. FD causes minimum changes in bone morphology, and tooth resorption rarely occurs. In odontoma, the pattern of calcifications resembles tooth structures. Sarcomas do not have well-defined borders. In radiographic findings of CEOT, honeycomb appearance is seen. Adenomatoid odontogenic tumor (AOT) generally affects young individuals associated with an impacted tooth, which mostly occurs in maxilla.

Complete surgical resection and reconstruction are indicated in case of large lesions. Surgical curettage also may perform in case of small lesions, but there is a chance of recurrence (28%). However, a long-term follow-up is needed in this case.[2],[10]

Ossifying fibroma consists of different morphological features, and therefore, it can be mistaken for other FOL. Therefore, we can achieve a correct diagnosis only by correlating the clinical, radiographic, and pathological features of the lesion.[1]

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Swami A, Kale L, Mishra S, Choudhary S. Central ossifying fibroma of mandible: A case report and review of literature. J Indian Acad Oral Med Radiol 2015;27:131.  Back to cited text no. 1
  [Full text]  
2.
Bal Reddy P, Sridhar Reddy B, Prasad N, Kiran G, Patlolla K. Central ossifying fibroma of the mandible: An unusual case report. J Clin Diag Res 2012;6:539-41.  Back to cited text no. 2
    
3.
Algadi HH, Alqasi WM, Jamali OM. Central ossifying fibroma in old female: A case report and review of literature. J Indian Acad Oral Med Radiol 2018;30:315-7.  Back to cited text no. 3
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4.
Mohapatra M, Banushree CS, Nagarajan K, Pati D. Cemento-ossifying fibroma of mandible: An unusual case report and review of literature. J Oral Maxillofac Pathol 2015;19:405.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
da Silveira DT, Cardoso FO, Silva BJA, Alves Cardoso CA, Manzi FR. Ossifying fibroma: Report on a clinical case, with the imaging and histopathological diagnosis made and treatment administered. Rev Bras Ortop 2016;51:100-4.  Back to cited text no. 5
    
6.
Sciubba JJ, Younai F. Ossifying fibroma of the mandible an d maxilla: Review of 18 cases. J Oral Pathol Med 1989;18:315-21.  Back to cited text no. 6
    
7.
Carvalho B, Pontes M, Garcia H, Linhares P, Vaz R. Ossifying Fibromas of the Craniofacial Skeleton, Histopathology Enrique Poblet, IntechOpen. Fibromas of the Craniofacial Skeleton, Histopathology Enrique Poblet, IntechOpen. Available from: https://www.intechopen.com/books/histopathology-reviews-and-recent-advances/ossifying-fibromas-of-the-craniofacial-skeleton. [Last accessed on 2018 Jun 25].  Back to cited text no. 7
    
8.
Liu Y, Wang H, You M, Yang Z, Miao J, Shimizutani K, et al. Ossifying fibromas of the jaw bone: 20 cases. Dentomaxillofac Radiol 2010;39:57-63.  Back to cited text no. 8
    
9.
MacDonald-Jankowski DS. Ossifying fibroma: A systematic review. Dentomaxillofac Radiol 2009;38:495-513.  Back to cited text no. 9
    
10.
Silvestre-Rangil J, Silvestre FJ, Requeni-Bernal J. Cemento-ossifying fibroma of the mandible: Presentation of a case and review of the literature. J Clin Exp Dent 2011;3:e66-9.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10]



 

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