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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 3  |  Page : 327-330

Juvenile ossifying fibroma of the mandibular ramus in a five-year-old boy


1 Department of Oral Medicine and Radiology, Sri Hasanamba Dental College and Hospital, Hassan, Karnataka, India
2 Departments of Oral Medicine and Radiology, JSS Dental College and Hospital, JSS University, Mysore, Karnataka, India
3 Departments of Oral and Maxillofacial Surgery, JSS Dental College and Hospital, JSS University, Mysore, Karnataka, India

Date of Submission26-Aug-2014
Date of Acceptance15-Oct-2014
Date of Web Publication19-Nov-2014

Correspondence Address:
Srikanth Hanasoge Srivathsa
Department of Oral Medicine and Radiology, Sri Hasanamba Dental College and Hospital, Vidyanagar, Hassan, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.145020

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   Abstract 

Juvenile ossifying fibromas (JOF) are benign central lesions occurring in children. Two variants of this entity have been recognized - trabecular and psammomatoid. These lesions are usually asymptomatic and present only as a growing swelling. We hereby report a case of a trabecular variant of a juvenile ossifying fibroma in a five-year-old boy.

Keywords: Aggressive ossifying fibroma, juvenile ossifying fibroma, ossifying fibroma


How to cite this article:
Srivathsa SH, Puttaraju MK, Patil K, Degala S. Juvenile ossifying fibroma of the mandibular ramus in a five-year-old boy . J Indian Acad Oral Med Radiol 2014;26:327-30

How to cite this URL:
Srivathsa SH, Puttaraju MK, Patil K, Degala S. Juvenile ossifying fibroma of the mandibular ramus in a five-year-old boy . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2022 May 22];26:327-30. Available from: https://www.jiaomr.in/text.asp?2014/26/3/327/145020


   Introduction Top


Adiverse group of entities, characterized by replacement of normal bone with a fibrous connective tissue matrix, containing bone, osteoid or cementum have been termed as fibro-osseous lesions. The juvenile ossifying fibroma (JOF) is an atypical type of this group of lesions. [1]

Ossifying fibromas are benign lesions that can occur centrally or peripherally. The central ossifying fibroma is most common. This entity has been also referred to as ossifying fibroma, cemento-ossifying fibroma or cementifying fibroma. All these terminologies describe the same entity. [2] Juvenile aggressive ossifying fibroma, aggressive psammomatoid ossifying fibroma, and juvenile active ossifying fibroma are the other synonyms. [2]


   Case Report Top


A five-year-old male patient was brought to the Department of Oral Medicine and Radiology for evaluation of a swelling on the right side of his face, which was present since a year. There was no history of trauma preceding the onset of swelling. The swelling had gradually increased in size for about six months and remained static thereafter. There was no history of pain in the swelling or any other associated symptoms. The patient's parents had not sought any consultation previously. His medical and dental histories were noncontributory. On extraoral examination, no obvious swelling was detected on inspection, and only on palpation, a solitary, hard, nontender, non-mobile swelling, measuring about 2 2 cm in size, was detected on the right side of his face, at the level of the tragus of the ear, corresponding to the middle of the ramus of the mandible [Figure 1]. The swelling was nonreducible and no egg-shell crackling was noted. The regional lymph nodes were non-palpable. Intraoral examination revealed no visible swelling, and on palpation, a solitary, hard swelling on the right side of the ramus of the mandible was detected, with smooth margins. There was no obliteration of the buccal or lingual vestibules. The swelling was nontender and no decortications were noticed [Figure 2]. Differential diagnoses of an odontogenic cyst, idiopathic osteosclerosis, and osteoblastoma were considered.
Figure 1: Extraoral photograph of the patient

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Figure 2: Intraoral photograph of the lesion

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A panoramic radiograph showed a well-defined, multilocular radiolucency, on the right side of the mandibular ramus, measuring about 2.5 x 2 cm in size, having well-defined, corticated margins. There was no evidence of discontinuity of the posterior border of the ramus of the mandible. No internal radio-opacities were detected [Figure 3].
Figure 3: Digital panoramic radiograph showing a multilocular radiolucent lesion

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A computed tomographic scan was performed, which showed a solid, hypodense multilocular mass in the ramus of the mandible, with expansion of the buccal cortex, a picture similar to the panoramic radiograph, with no internal hyperdense areas and no enhancement on contrast agent administration [Figure 4], [Figure 5], [Figure 6].
Figure 4: Axial computed tomography (CT) image showing the lesion in the right ramus of the mandible

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Figure 5: Coronal CT image showing the expansile multilocular lesion in the ramus of the mandible

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Figure 6: 3D reconstruction of the lesion

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An excisional biopsy was performed under general anesthesia and the specimen was submitted for histopathological examination. Under Hematoxylin and Eosin staining, the sections showed a tumor composed of proliferating spindle cells with fine chromatin and trabeculae of bone and osteoid, with osteoblastic rimming. The histopathological findings suggested an ossifying fibroma [Figure 7].
Figure 7: Photomicrograph of the lesion (40× magnification)

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On the basis of the clinical, radiographical, and histopathological findings, a final diagnosis of juvenile ossifying fibroma- trabecular variant in the right ramus of mandible was made. The patient is under follow-up and six months postoperatively, no recurrence has been observed [Figure 8].
Figure 8: Six-month post operative digital panoramic radiograph

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   Discussion Top


Definition

The World Health Organization (WHO) describes juvenile ossifying fibroma (JOF) as an actively growing fibro-osseous neoplasm displaying a cell-rich fibrous stroma, containing bands of cellular osteoid without osteoblastic lining, along with trabeculae of typical woven bone. Foci of small giant cells may be present. The lesion is non-encapsulated, but well-demarcated from the surrounding bone. [1] The Ossifying Fibroma was first described in 1872, by Menzel. [3]

Classification

Slootweg et al., have classified JOF into two types: The JOF-WHO type and JOF-PO (Psammoma-like ossicles) type, based on the age of occurrence. El-Mofty has classified it into the trabecular and the psammomatoid types, based upon the histopathological appearance. [3]

Etiopathogenesis

It is not clear as to how this lesion originates. However, an origin from the periodontal ligament is proposed by many authors. [2] Others believe that it arises from the primitive mesenchymal cells or remnant cells after incomplete migration of the medial part of the nasal anlage. [4] Mutation of the tumor suppressor gene, HRPT 2, has also been identified in cases of JOF. [5]

Epidemiology

A juvenile ossifying fibroma commonly occurs between five and fifteen years of age. About 2% of oral tumors in children constitute JOFs. Nevertheless, there are reports with patient ages ranging from three months to 72 years. [3] According to one report, there is a conflict about the predilection of JOFs in the jaws. Although, it generally occurs in the premolar-molar region, it can also occur in the maxilla, paranasal sinuses, orbit, mandible, and frontoethmoid bones. No gender predilection has been reported in the literature. [2] The present case is atypical, in that it has occurred in the middle of the ramus of the mandible in a five-year-old boy.

Clinical features

Generally, JOFs are asymptomatic. Some tumors enlarge very rapidly and cause extensive bone destruction resulting in asymmetry of the face, local pain, loss of teeth, displacement of roots, malocclusion, cortical plate expansion, and perforation. Maxillary JOFs can cause maxillary nasal obstruction, sinusitis, and proptosis. [2],[6] The present case was also completely asymptomatic and swelling was the only complaint.

Imaging

On a conventional radiograph, such as a panoramic radiograph, the lesion can appear as a totally radiolucent, radiopaque or a mixed lesion, depending on the degree of calcification. There can be evidence of expansion or perforation of the cortex or cortices, displacement of teeth and root resorption. The lesion is usually well-corticated with a radiopaque border. Very rarely a ground-glass appearance can be noted. [6] The radiograph may sometimes show nonspecific features, such as a unilocular or multilocular radiolucency, having ill-defined borders and occasional central opacification. [2] In the present case, the radiographic feature was that of a multilocular lesion, with no internal radio-opacities.

Histopathological features

The histological features of JOFs include a cell-rich fibrous stroma containing a cellular osteoid without an osteoblastic lining, osteoid strands, and cementum particles. The trabecular JOF is characterized by the presence of trabeculae, fibrillar osteoid, and woven bone. The psammomatoid variant is characterized by the presence of small uniform spherical ossicles that resemble psammoma bodies. [3] In the present case as well, proliferating spindle cells with osteoid formation and osteoblastic rimming were noted.

Management

Many authors suggest conservative treatment, such as, enucleation or curettage, as the treatment of choice, whereas, others suggest a radical en bloc resection for larger lesions and curettage for smaller ones. [4]

Prognosis

A high recurrence rate of JOFs has been noted; as high as 30-58%. Recurrence is attributed to an incomplete treatment procedure. [2],[3] In our case, there has been no recurrence of the lesion six months postoperatively.


   Conclusion Top


Juvenile ossifying fibromas are rare and aggressive lesions. They are most often asymptomatic and the presence of a swelling may be the only presenting symptom, as in the present case. Thorough clinical, radiographical, and histopathological examinations are required to differentiate JOFs from other serious pathologies, such as osteosarcomas.

 
   References Top

1.Ravikumar R, Raghavendra K, Santhosh Kumar. Aggressive juvenile ossifying fibroma of the anterior mandible. J Dent Sci Res 2011;2:26-34.  Back to cited text no. 1
    
2.Tortorici S, Buzzanca ML, Burruano F, Difalco P. Juvenile central ossifying fibroma of the mandible: A case report. J Pediatr Surg 2010;45:e1-4.  Back to cited text no. 2
    
3.Ranadive PA, Deshpande MD, Ingole SN, Ankush C. Mandibular mass in a pediatric patient: A diagnostic dilemma. J Indian Soc Pedod Prev Dent 2012;30:179-82.  Back to cited text no. 3
[PUBMED]  Medknow Journal  
4.Smith SF, Newman L, Walker DM, Papadopoulos H. Juvenile aggressive psammomatoid ossifying fibroma: An interesting, challenging, and unusual case report and review of the literature. J Oral Maxillofac Surg 2009;67:200-6.  Back to cited text no. 4
    
5.Rao SC, Dandriyal R, Sapra G, Sharma H, Agarwal U, Pandit N. Psammomatoid juvenile aggressive ossifying fibroma of mandible. Natl J Maxillofac Surg 2012;3:47-50.  Back to cited text no. 5
[PUBMED]  Medknow Journal  
6.Thankappan S, Nair S, Thomas V, Sharafudeen KP. Psammomatoid and trabecular variants of juvenile ossifying fibroma - Two case reports. Indian J Radiol Imaging 2009;19:116-9.  Back to cited text no. 6
[PUBMED]  Medknow Journal  


    Figures

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



 

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