|Year : 2014 | Volume
| Issue : 2 | Page : 232-235
Juvenile trabecular ossifying fibroma
Nymphea Pandit1, Nitin Saini2, Shikha Kler2, Shaifi Jindal1
1 Department of Periodontology and Oral Implantology, D.A.V. (C), Dental College and Hospital, Yamunanagar, Haryana, India
2 Department of Oral and Maxillofacial Pathology, D.A.V. (C), Dental College and Hospital, Yamunanagar, Haryana, India
|Date of Submission||29-Mar-2012|
|Date of Acceptance||04-Oct-2013|
|Date of Web Publication||23-Apr-2014|
Departments of Periodontology and Oral Implantology, D.A.V. (C) Dental College and Hospital, Model Town, Yamunanagar, Haryana - 135 001
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Juvenile ossifying fibroma (JOF) is an uncommon fibro-osseous lesion with highly aggressive clinical behavior, higher incidence in young adults, and a strong tendency to recur. Two categories, trabecular JOF (TrJOF) and psammomatoid JOF (PsJOF), based on histologic criteria and a distinct predilection for specific age groups have been identified. Juvenile trabecular ossifying fibroma of peripheral variety is an uncommon clinical entity with aggressive local behavior and high recurrence rate. This article reports a case of aggressive JTOF in the anterior mandibular region in a 13-year-old boy with history of recurrence. Complete surgical resection of the growth was done along with involved periodontal ligament and periosteum to minimize the possibility of recurrence. The case was followed up for more than 3 months without recurrence.
Keywords: Histology, pathology-oral, prognosis, treatment planning
|How to cite this article:|
Pandit N, Saini N, Kler S, Jindal S. Juvenile trabecular ossifying fibroma. J Indian Soc Periodontol 2014;18:232-5
| Introduction|| |
Benign fibro-osseous lesions of the head and neck region are uncommon and constitute a wide range of tumors sharing some histopathological features. These include fibrous dysplasia (FD), ossifying fibroma (OF), and cemento-osseous dysplasia (COD).  Ossifying fibromas are rare benign, neoplasms arising from undifferentiated cells of the periodontal ligament tissues.  These have been described as demarcated or rarely encapsulated neoplasms consisting of fibrous tissue containing varying amounts of mineralized material resembling bone and/or cementum which is one of its principal characteristics.  In 1872, Menzel first described ossifying fibroma, but in 1972, Montgomery assigned terminology to it. It accounts for 3.1% of all oral tumors and for 9.6% of all gingival lesions. There are two types of ossifying ﬁbroma, central and peripheral. The central type arises from the endosteum or periodontal ligament adjacent to the root apex and expands from the medullary cavity of the bone. The peripheral type occurs solely on the soft tissue overlying the alveolar process.  It is common in young adults (2 nd and 3 rd decades) with a female predominance and is generally asymptomatic until the growth produces a noticeable swelling most often found in interdental gingiva, located anterior to molars and in the maxilla.  The suggested etiology although unknown has been associated with inflammatory hyperplasia of the periodontal ligament.  Trauma or local irritants are known to precipitate the development of this lesion.  When ossifying fibroma is diagnosed in young people the term "juvenile" is used. The accurate nature and classification of JOF has undergone considerable debate among pathologists, resulting in a confusing evolution of competing nomenclatures.  According to the WHO classification of odontogenic tumors 2005, JOF is further subdivided into two distinct clinic-pathological variant i.e., juvenile psammornatoid ossifying fibroma (JPOF) and juvenile trabecular ossifying fibroma (JTOF).  In this paper, a recurrent case of JTOF occurring peripheraly in a young patient is described.
| Case Report|| |
A 13-year-old boy reported to the Department of Periodontology and Oral Implantology, DAV (C) Dental College and Hospital, Yamunanagar, with a chief complaint of painless, swelling of the gums in left mandibular anterior region from past 20 days. The swelling was first noticed by the patient 2 months back as a painless, small lesion which increased gradually in size. The growth was removed 1 month back but it reappeared within 20 days and progressively increased to the present size. Intraoral examination revealed the diffuse swelling extending from distal aspect of 32 up to mesial aspect of 31, and an open contact area between 31 and 32 wherefrom it extended to the distal aspect of 31 labio-lingually [Figure 1]. The overlying gingiva was red and erythematous, soft in consistency with rolled out margins. Bleeding on probing was present with probing depth of 4-5 mm and grade I mobility in relation to 31, 32, 41, 42. Both the maxillary lateral incisors were congenitally missing. Apart from poor oral hygiene, the rest of the oral cavity showed no unusual features. There was no accompanying cervical lymphadenopathy and no relevant systemic history. Routine hemogram was found to be normal.
Intra- oral periapical radiograph (IOPA) of 31, 32, 41 revealed mild horizontal bone loss between 31 and 32, widening of periodontal ligament in relation to 31, and loss of lamina dura in relation to 32 [Figure 2]. Orthopantomograph (OPG) revealed no other significant findings.
|Figure 2: Intraoral periapical radiograph of 31, 32, 41 showing mild horizontal bone loss between 31 and 32, widening of periodontal ligament in relation to 31 and loss of lamina dura in relation to 32|
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The treatment plan included scaling and root planning (Phase I therapy). After a week, surgical excision was done. Following the administration of local anesthesia, the growth was excised completely and curettage was performed up to the bone level. It was made sure that all the soft tissue was removed particularly from the periodontal ligament region. Periodontal dressing was applied after suturing the flap. Excised tissue [Figure 3] was stored in the 10% formalin and sent to the department of oral pathology for histopathological examination.
The patient was advised to rinse with 0.2% chlorhexidine gluconate solution twice daily for 14 days for assistance in plaque control. He was prescribed a non-steroidal anti-inflammatory agent three times a day for 3 days to prevent post-operative discomfort and amoxicillin 250 mg three times a day for 5 days to prevent infection. Sutures were removed after 10 days.
Histopathology of the biopsied specimen reveals stratified squamous epithelium with underlying connective tissue [Figure 4] and [Figure 5]. Just beneath the epithelium irregular interconnecting trabeculae of immature bone tissue containing coarse lacunae with plump osteocytes is seen [Figure 6]. These trabeculae anastomose to form a lattice type structure [Figure 7]. Connective tissue is highly cellular consisting of numerous proliferating fibroblasts. Overall histopathological features are suggestive of JTOF.
|Figure 4: Photomicrograph showing epithelium and underlying connective tissue with calcifi cations.(Original magnifi cation, ×40)|
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|Figure 5: Photomicrograph showing stratifi ed squamous epithelium (blue arrow) and trabecular osteiod tissue (black arrow). (Original magnifi cation, ×100)|
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|Figure 6: Photomicrograph showing trabeculae anastomizing to form lattice type structure (blue arrow). (Original magnifi cation, ×100)|
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|Figure 7: Photomicrograph showing coarse lacunae with plump osteocytes. (Original magnifi cation, ×400)|
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No recurrence was seen even after 3 months post operatively and the gap between the teeth (31 and 32) seemed to be reduced eventually after the complete removal of the growth.
| Discussion|| |
The most characteristic feature of JOF, as the name suggests, is its aggressive clinical behavior and higher incidence in young adults. Among the many classification systems for this lesion, the most recent classification is by El-Mofty (2002) who identified two categories, trabecular JOF (TrJOF) and psammomatoid JOF (PsJOF), based on histologic criteria.  However, the two categories also have a distinct predilection for specific age groups. The average age of occurrence of TrJOF is 8-12 years, whereas that of PsJOF is 16-33 years. One clinical feature that helps differentiate TrJOF from PsJOF is the site of involvement, with PsJOF occurring mainly in the paranasal sinuses and TrJOF occurring mainly in the maxilla. Mandibular and extracranial involvement is rare. Cases affecting the mandible have been reported, the ramus-molar area being the most common site.  This case report highlights the documentation of a JOF occurring in the mandibular anterior region, thus making it a rare entity. Also, the present case represents the clinical as well as histopathological presentation as evident in JTOF. As the lesion recurred and grew rapidly in present case, a more aggressive behavior of juvenile ossifying fibroma can be confirmed which probably occurred due to incomplete initial removal or persistence of local irritants.  It is seen that incomplete resection causes recurrence in aggressive tumors and recurrence rates of 30% to 58% have been reported for juvenile ossifying fibromas. , However, the case reported here is followed up for more than 3 months without recurrence [Figure 8] suggesting that thorough surgical resection, rather than conservative curettage, is therefore the preferred line of treatment. The histopathological picture further confirmed the trabecular pattern of juvenile ossifying fibroma. Such aggressive pattern is seen mainly in young patients because high levels of periodontal ligament activity (e.g., formation and degradation) are more commonly seen in children, and the constant irritation associated with both primary tooth exfoliation and permanent tooth eruption can contribute for the increased prevalence of reactive lesions in younger patients.  Although the classic ossifying fibromas occurs two to four times more frequently in females than in males but no significant sexual predilection is noted in juvenile ossifying fibroma.  The lesion may cause a separation of the adjacent teeth, and occasionally minimal bone resorption can be seen beneath the lesion.  These findings are also evident in the radiograph in this case and interestingly the gap between the teeth (31 and 32) seemed to be reduced eventually after the complete removal of the growth. As the rapid growth rate often exhibited by these lesions can be quite alarming, the clinician must be aware of the aggressiveness and distinct histopathology of JTOF for adequate treatment planning to prevent their recurrence in future.
| Conclusion|| |
Juvenile trabecular ossifying fibroma of peripheral variety is an uncommon clinical entity. The aggressive local behavior and high recurrence rate calls for an early diagnosis, prompt treatment, and especially, long-term follow up of the patient. Elimination of etiological factors and complete surgical excision along with involved periodontal ligament and periosteum minimize the possibility of recurrence. The rarity of this condition makes it beyond the domain of many clinicians and the unpredictable recurrence at varied intervals makes the management difficult.
| References|| |
|1.||Sarode SC, Sarode GS, Waknis P, Patil A, Jashika M. Juvenile psammomatoid ossifying fibroma: A review. Oral Oncol 2011;47:1110-6. |
|2.||Godhi S, Goyal S, Giraddi G, Goyal S. Cementifying fibroma of the mandible: A case report. J Oral Health Comm Dent 2008;2:42-5. |
|3.||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. |
|4.||Yadav A, Mishra MB. Peripheral cemento- ossifying fibroma of mandible: A case report. Indian J Stomatol 2011;2:193-6.2 |
|5.||Shetty DC, Urs AB, Ahuja P, Sahu A, Manchanda A, Sirohi Y. Mineralized components and their interpretation in the histogenesis of peripheral ossifying fibroma. Indian J Dent Res 2011;22:56-61. |
|6.||Kenney JN, Kaugars GE, Abbey LM. Comparison between the peripheral ossifying fibroma and peripheral odontogenic fibroma. J Oral Maxillofac Surg 1989;47:378-82. |
|7.||Brannon RB, Fowler CB. Benign fibro-ossoeus lesions: A review of current concepts. Adv Anat Pathol 2001;8:126-43. |
|8.||Barnes L, Eveson JW, Reichart P, Sidransky D. (editors). World Health Organisation classification of tumours. Pathology and genetics of head and neck tumours. Lyon: IARC Press; 2005. p. 283-328. |
|9.||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. |
|10.||Ravikumar R, Raghavendra K, Kumar S. Aggressive juvenile ossifying fibroma of the anterior mandible: Case report. J Dent Sci Res 2011;2:26-34. |
|11.||Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 2 nd ed. Philadelphia: W. B. Saunders Company; 2004. p. 557. |
|12.||IPassos M, Azevedo R, Janini ME, Maia LC. Peripheral cemento-ossifying fibroma in a Child: A case report. J Clin Pediatr Dent 2007;32:57-9. |
|13.||Wood NK, Goaz PW. Differential Diagnosis of Oral and Maxillofacial Lesions. 5 th ed. St Louis: Mosby; 2006. p. 143-4. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]