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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 8  |  Issue : 1  |  Page : 179-181  

Trabecular variant: A rare entity of Juvenile ossifying fibroma of the mandible


1 Department of Oral and Maxillofacial Surgery, National Institute of Medical Sciences Dental College and Hospital, Jaipur, Rajasthan, India
2 Department of Oral Medicine Diagnosis and Radiology, National Institute of Medical Sciences Dental College and Hospital, Jaipur, Rajasthan, India
3 Department of Oral Medicine and Radiology, National Institute of Medical Sciences Dental College and Hospital, Jaipur, Rajasthan, India

Date of Web Publication24-Apr-2017

Correspondence Address:
Pallavi Malaviya
Department of Oral and Maxillofacial Surgery, National Institute of Medical Sciences Dental College and Hospital, Jaipur, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0976-237X.205043

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   Abstract 


One of the rarest entities of fibro-osseous lesions that arise within the craniofacial bones is Juvenile ossifying fibroma (JOF). It is an intraosseous expansile lesion of the jaw that imitate odontogenic lesions. WHO has described two distinct histopathological variants of JOF; trabecular and psammomatoid. Histologically, they are characterized by the presence of fibrous connective tissue stroma along with osteoblastic and osteoclastic cells. Clinical, characteristics show an early age of onset, typical histological patterns, high rate of aggressive behavior and recurrence. This article presents a rare clinical case of the trabecular variant of JOF, its clinical, radiological, histological, and treatment aspects.

Keywords: Aggressive lesion, mandible, trabecular variant


How to cite this article:
Malaviya P, Choudhary S, Gupta S, Toshniwal O D. Trabecular variant: A rare entity of Juvenile ossifying fibroma of the mandible. Contemp Clin Dent 2017;8:179-81

How to cite this URL:
Malaviya P, Choudhary S, Gupta S, Toshniwal O D. Trabecular variant: A rare entity of Juvenile ossifying fibroma of the mandible. Contemp Clin Dent [serial online] 2017 [cited 2021 Jul 30];8:179-81. Available from: https://www.contempclindent.org/text.asp?2017/8/1/179/205043




   Introduction Top


Juvenile ossifying fibroma (JOF) is an actively growing aggressive lesion containing cells rich in the fibrous stroma containing cellular osteoid, trabeculae of woven bone without osteoblastic lining, with or without Clusters of giant cells. It is nonencapsulated and well demarcated from surrounding bone (WHO).[1] In 2002 El-Mofty first identified the two histopathological variants of JOF: Trabecular and psammomatoid.[2] Rare cases of mandibular JOF have been reported.[3] Clinical examination reveals it as asymptomatic slow growing swelling causing facial asymmetry, extending to a substantial size and behaving as an aggressive lesion.[4] Pain and paraesthesia are less commonly noted. Depending on the site of involvement, exophthalmos, and epistaxis is sometimes observed. It has higher male predominance with the mean age of occurrence ranging from 2 to 15 years.[4] The radiographic features show unilocular or multilocular radiolucency with well-delineated borders and few opacification in the center.[4] The suggestive features of the aggressive variant are the presence of the perforation, cortical thinning, tooth displacement, and root resorption. The advanced imaging studies show more invasive and destructive features apart from conventional radiographic features.[4] Histopathologically both the entities are nonencapsulated, well demarcated from the surrounding bone consisting of neoplastic cellular stroma formed by spindle shape fibroblast.[5] The trabecular variant shows irregular strands of cellular osteoid and irregular osteocytes. The osteoblast encloses the focal areas of the multinucleated giant cell. In contrast, the psammomatoid pattern forms concentrically laminated ossicles that vary in shape and having peripheral eosinophilic osteoid rims with basophilic centers typically.[5] The overall clinical features, histopathological features, and advanced imaging were indispensable to provide the diagnosis as JOF. A nonaggressive form of this lesion is managed by curettage and local excision whereas aggressive form is managed by complete surgical excision, en-bloc resection or hemimaxillectomy to prevent recurrences.[6] Reported here is a case of a trabecular variant of JOF in a 17-year-old female patient.


   Case Report Top


A 17-year-old female represented with a chief complaint of pain and slowly progressive swelling of the left side of face along with reduced mouth opening for the last 2 months. Initially, the swelling was smaller in size and increased progressively to attain final size causing facial asymmetry with reduced mouth opening. There was no relevant medical and dental history. Extraoral examination revealed an ill-defined, diffuse, and bony hard swelling of approximately measuring about 6 cm × 4 cm on the left side of the face, extending superior-inferiorly from left zygomatic arch till angle of the mandible and anteroposteriorly from nasolabial fold till tragus of the left ear with normal overlying skin [Figure 1]a and [Figure 1]b Intraorally, swelling was solitary, slightly tender with the expansion of buccal and lingual cortical plates with no pathological changes to the overlying mucosa. There was reduce mouth opening measuring about 15 mm in the greatest dimension [Figure 2]a. Orthopantomogram revealed a large well-bordered, well-defined unilocular, circular, mixed lesion (calcification specks) approximately, 2 cm in size involving the ramus, coronoid, and angle of the mandible [Figure 3] posteroanterior view of mandible showed a well-defined, mixed lesion involving the coronoid process, ramus and the angle of the left side of the mandible [Figure 4]. Axial and sagittal view of computed tomography confirmed the routine radiographic features and further showed a heterogeneously enhancing mixed lytic-sclerotic lesion with a sclerotic rim extending into adjacent soft tissue measuring 5.12 cm × 3.83 cm with exterior and superior extension into the left infratemporal fossa and medial extension up to pterygoid muscle [Figure 5]a and [Figure 5]b. Biopsy was taken from the involved site, and the H and E section showed the presence of trabeculae of fibrillar osteoid and woven bone. In few areas, multinucleated giant cells were also noted. Peripherally bony trabeculae lined by osteoblast were also seen [Figure 6]. Based on clinical, radiographic, advanced imaging studies and histopathological report provided the diagnosis of the trabecular variant of JOF affecting left mandible with differential diagnosis of cement-ossifying fibroma, osteoblastoma, and osteogenic sarcoma. The lesion was excised by segmental resection using transmandibular approach and reconstruction was done [Figure 7]a and [Figure 7]b. The closer was done using 3.0 vicryl sutures. Postoperative healing was uneventful; mouth opening was increased to the value of 30 mm [Figure 2]b. Postoperatively, the esthetics, oral function of the patient such as speaking, and chewing were intact and normal [Figure 8]a and [Figure 8]b. The patient is still under regular follow-up as the lesion has high recurrence rate.
Figure 1: (a and b) preoperative extraoral view of the patient showing swelling on the left side of face

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Figure 2: (a) Preoperative mouth opening, (b) postoperative mouth opening

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Figure 3: Preoperative panoramic view of the patient

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Figure 4: Preoperative posteroanterior view of the patient

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Figure 5: (a) Axial view of computed tomography, (b) sagittal view of computed tomography

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Figure 6: Histopathological section

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Figure 7: (a) Postoperative panoremic view of the patient, (b) postoperative posteroanterior view of the patient

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Figure 8: (a and b) Postoperative extraoral view of the patient with intact facial symmetry

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   Discussion Top


JOF mostly strikes children with slightly male predominance with a mean age of 11 years. Clinically, it is asymptomatic, however rarely it is aggressive and symptomatic.[7] In the present case, the patient was symptomatic showing its rare entity. According to Slootweg and Müller, both maxilla and mandible are affected with more prevalence for maxilla.[8] In the present case, the duration of swelling noted was 2 months. The characteristic radiographic features reveal a central opacification with well-defined unilocular to multilocular pattern giving it a ground glass appearance.[9] This radiographic appearance depends on duration, stage, and histology of the lesion. The 50% of cases of JOF are recorded in multiple sinuses with few occurring in the single sinus.[9] The duration, clinical and radiographic features and extension of the lesion into the adjacent soft tissue favors the aggressive type of JOF as previously reported.[3] The features like resorption and perforation of cortices, tooth displacement and aggressive growth of the lesion favor the diagnosis as an aggressive variety of JOF. In this case, except for tooth resorption all other features like perforation of the cortices and extension into the surrounding tissue was noted. Because of the high recurrence rate, there is no follow-up protocol mentioned in the literature and immediate reconstruction is not advised. The smaller lesions are treated by enucleation and curettage successfully.[10] Resection is considered where there is recurrence, invasion into surrounding tissues, or when preserving the inferior border is not feasible.[11]


   Conclusion Top


JOF is an uncommon clinical entity, because of its aggressiveness and high recurrence rate it is necessary to arrive at an early diagnosis, applying the appropriate clinical, diagnostic approaches, and appropriate treatment for effective results and long-standing follow-up.

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.
Williams HK, Mangham C, Speight PM. Juvenile ossifying fibroma. An analysis of eight cases and a comparison with other fibro-osseous lesions. J Oral Pathol Med 2000;29:13-8.  Back to cited text no. 1
    
2.
El-Mofty S. Psammomatoid and trabecular juvenile ossifying fibroma of the craniofacial skeleton: Two distinct clinicopathologic entities. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;93:296-304.  Back to cited text no. 2
    
3.
Leimola-Virtanen R, Vähätalo K, Syrjänen S. Juvenile active ossifying fibroma of the mandible: A report of 2 cases. J Oral Maxillofac Surg 2001;59:439-44.  Back to cited text no. 3
    
4.
Papadaki ME, Troulis MJ, Kaban LB. Advances in diagnosis and management of fibro-osseous lesions. Oral Maxillofac Surg Clin North Am 2005;17:415-34.  Back to cited text no. 4
    
5.
Neville BW, Damm D, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 3rd ed. Philadelphia: WB Saunders; 2005.  Back to cited text no. 5
    
6.
Regezi JA, Sciubba JJ. Oral Pathology: Clinical Pathologic Correlations, Genetic and Metabolic Diseases of the Jaws. Philadelphia: WB Saunders; 1989.  Back to cited text no. 6
    
7.
Walter JM Jr., Terry BC, Small EW, Matteson SR, Howell RM. Aggressive ossifying fibroma of the maxilla: Review of the literature and report of case. J Oral Surg 1979;37:276-86.  Back to cited text no. 7
    
8.
Slootweg PJ, Müller H. Juvenile ossifying fibroma. Report of four cases. J Craniomaxillofac Surg 1990;18:125-9.  Back to cited text no. 8
    
9.
Mintz S, Velez I. Central ossifying fibroma: An analysis of 20 cases and review of the literature. Quintessence Int 2007;38:221-7.  Back to cited text no. 9
    
10.
Rinaggio J, Land M, Cleveland DB. Juvenile ossifying fibroma of the mandible. J Pediatr Surg 2003;38:648-50.  Back to cited text no. 10
    
11.
Keles B, Duran M, Uyar Y, Azimov A, Demirkan A, Esen HH. Juvenile ossifying fibroma of the mandible: A case report. J Oral Maxillofac Res 2010;1:e5.  Back to cited text no. 11
    


    Figures

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



 

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