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Aggressive Juvenile Ossifying Fibroma

Volume 2 Issue 1 February 2011

Case Report

Aggressive Juvenile Ossifying Fibroma of the Anterior Mandible


Dr.Ravikumar.R1, Dr.Raghavendra.K2, Dr.Santhosh Kumar3
1

Senior Lecturer, 2Reader Department of Oral Surgery, 3Senior Lecturer Departmant

of Periodontics, Sri Siddhartha Dental College, Tumkur, Karnataka.

Abstract: Juvenile ossifying fibroma (JOF) is an uncommon fibro-osseous lesion containing different amounts of calcified tissue scattered in a cellular fibroblastic stroma. bones. It is highly aggressive and has a strong tendency to recur. It has been recognized as a separate histopathological entity among the fibro-osseous group of lesions. The clinical behavior of Juvenile ossifying fibroma is variable and difficult to predict. This article reports a case of Aggressive JOF of the anterior mandible in a 10 year old girl. Key words: ossifying fibroma; fibro-osseous lesion; juvenile ossifying fibroma; psammomatoid; trabecular; active fibrous dysphasia.
Journal of Dental Sciences & Research 2:1: Pages 26-34

Introduction: The fibro osseous lesions of the jaws represent a diverse group of entities that are characterized by replacement of normal bone by a fibrous connective tissue matrix,

developmental lesions, reactive

(hamartomatous) or dysplastic

processes and neoplasms. One such rare or atypical fibro

osseous lesion is Juvenile ossifying Fibroma describes histopathological Psammomatoid and (JOF). The two term JOF

with in which varying amounts of osteoid, immature and mature bone and in some instances, cementumlike material are deposited1. Fibro osseous lesions of the jaws include

distinct variants: Trabecular

varieties2 .Psammomatoid JOF is an extra-gnathic lesion occurring

26 Journal of Dental Sciences and Research

Aggressive Juvenile Ossifying Fibroma

Volume 2 Issue 1 February 2011 Panoramic and intra-oral periapical radiographs revealed a radiolucent mass in the 31, symphyseal 32, 41 region 42

predominantly in the sinonasal and orbital bones. Trabecular JOF is a gnathic lesion affecting the jaws with a predilection for maxilla2, 3, 4. Cases affecting the mandible have been reported; the ramus- molar area being the most common site3. This presentation highlights the third

displacing

and

laterally and perforating both the cortical plates of the mandible

[Figure-3a, b]. CT scan revealed mass, an expansile

documentation of a JOF occurring in the mandibular anterior region; thus making it a rare entity. Case report: A 10-year-old girl presented to the department of oral and maxillofacial surgery with a rapidly enlarging

osteolytic

encompassing

symphysis of the mandible causing splaying of the incisors; with an intact lower mandibular border. Few bony spicules were seen with in the lesion [Figure-4 a,b]. Routine blood examination and

mass of 20days duration over the mandibular anterior region[Figure1]. On physical firm, examination, oral an

biochemical investigations were with in the normal limits. An incisional biopsy was performed, fixed in 10% formalin and sent for

ovoid,

intra

swelling,

measuring about 3x3 cm; in relation to 31, 32, 41 and 42, slightly

histopathological

examination.

Correlating the age, duration, clinical behavior, radiological pattern and microscopic appearance [Figure-

extending into the floor of the mouth with minimal tenderness on

palpation and no significant regional lymphadenopathy.There was Lateral displacement with gradeII mobility of permanent mandibular incisors and deciduous mandibular

5a,b,c],the lesion was diagnosised as Aggressive JOF(Trabecular general

variant).

Under

anaesthesia, an enbloc resection was carried out through a by trans-oral chemical

canines[Figure-2a,b].

approach

followed

27 Journal of Dental Sciences and Research

Aggressive Juvenile Ossifying Fibroma

Volume 2 Issue 1 February 2011 is by El-Mofty


[4]

cauterization acid[Figure-6 postoperative eventful

using a,b,c]. recovery a

carbolic The was un

who identified two

categories, trabecular JOF (TrJOF) and psammomatoid on JOF (PsJOF), criteria.

without

significant

based

histologic

problem[Figure-6c]. The patient was reviewed regularly and on a follow up of one year, showed no signs of recurrence[Figure-8&9]. Discussion: The most characteristic feature of JOF, as the name suggests, is its higher incidence in children and
4

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 1633 years.4 Although JOF can occur anywhere in the skeleton, its highest incidence is in the facial bones, most commonly the maxilla.3,4,5,8 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.4 Mandibular and extracranial involvement is rare.5 Gender predilection has been a

young adults.5,6 However, it can also occur in the


5

older

age-groups .

Johnson et al

have reported JOFs

occurring at any age between 3 months and 72 years. Among the many classification systems for this lesion, the classification by Slootweg et al
7

is noteworthy. They have JOF into two distinct

classified

groups, the JOF-WHO type and JOFPO (psammoma-like ossicles) type, based primarily on the difference in the age of occurrence: the mean age of occurrence of JOF-WHO is 11.8 years and that of JOF-PO is 22.6 years. The most recent classification
2

matter of controversy, with some authors claiming no predilection for either sex, whereas Johnson et al . found a higher incidence in females
3,5

and El-Mofty reported a male

predilection.4

28 Journal of Dental Sciences and Research

Aggressive Juvenile Ossifying Fibroma

Volume 2 Issue 1 February 2011 of the involved site and the first presentation will be a clinically

The histogenesis of this lesion is poorly understood.Johnson et

al.believe that

mandibular

obvious swelling. All these features were seen in our case. Radiographically the internal

lesions arise from the myxoid dental papilla of the developing tooth.

Virtanen et al. consider JOF as a neoplasm that develops from the undifferentiated cells of the periodontal ligament.4 JOF usually manifests bony-hard as an

structure can be radiolucent, mixed, or radiopaque, depending on the degree of calcification.3,4 is though The lesion common rare, can Root and can cause

displacement resorption, occur.4,8,9

asymptomatic

swelling,

the duration and extent of which may vary depending on the site and aggressiveness of the lesion;

expansion as well as perforation.4 The radiographic features of JOF can resemble that of other lesions, such as fibrous dysplasia and cementoossifying fibroma.
2,4

however, it does not demonstrate the chronic, long-standing evolution of some of the other fibro-osseous lesions. It can expand the involved bones, causing facial asymmetry.

JOF

is

not

capsulated but is separated from surrounding bone by a radiopaque border, 3,8and this finding can help in differentiating dysplasia.3,10 it A from fibrous

Depending on the site, symptoms such as pain, sinusitis, paresthesia, proptosis,

malocclusion, swelling.8,9 In general,

'ground-glass'

etc., can also occur due to the

appearance on radiographs has been reported.4 It usually has a concentric

JOF

has

more

or centrifugal growth pattern, which can lead to an erroneous clinical diagnosis fibroma.8,10 of JOF cemento-ossifying has also been

aggressive growth pattern than the adult variant of are ossifying usually

fibromas.They

asymptomatic, exhibit rapid growth

reported to be associated with other

29 Journal of Dental Sciences and Research

Aggressive Juvenile Ossifying Fibroma

Volume 2 Issue 1 February 2011 conservative curettage, is therefore the preferred line of treatment.6,8 In the present case et we als followed staged

bony lesions such as aneurysmal bone cyst.4 Aggressive lesions with marked structures destruction may of adjacent

radiographically

modified

Troulis

mimic osteogenic sarcoma. 8,9 The microscopic features of the

protocol for the treatment of the jaw tumours in children. Conclusion: Although JOF is an uncommon

lesion are distinctive and include a cell-rich fibrous stroma containing bands of cellular osteoid without osteoblastic lining, osteoid strands, and trabeculae of woven bone.4,7,10 PsJOF is slightly more cellular than TrJOF. Due to the resemblance of the psammoma-like ossicles seen in PsJOF to the cementicles in

clinical entity, its aggressive local behaviour and high recurrence rate calls for an early diagnosis, prompt treatment and especially, long term follow up of the patient.The rapid growth rate often exhibited by these lesions can be quite alarming and cause the

cemento-ossifying fibroma, it has been argued that PsJOF is a type of cemento-ossifying


2,4

clinician to suspect the presence of a malignancy.

fibroma.

However, the marked cellularity of

JOF is in sharp contrast to the usually stroma-rich appearance of the latter group of lesions. The aggressive nature of this entity, along with the reported high rates of recurrence (30-58%),
4,8

suggests
FIGURE-1: A 10-year-old girl presented with a rapidly enlarging mass of 20 days duration region. over the mandibular anterior

that JOF should be treated like a locally aggressive neoplasm, very much Surgical like an ameloblastoma. rather than

resection,

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Aggressive Juvenile Ossifying Fibroma

Volume 2 Issue 1 February 2011

Figure 2a:

Figure 3b:
FIGURE-3a, b: OPG and IOPA radiographs revealed a radiolucent mass in the

symphyseal region

displacing 31, 32, 41

and 42 laterally and perforating both the cortical plates of the mandible.

Figure 2b:
FIGURE-2a, b: Local examination reveals an ovoid, firm, intra oral swelling, measuring about 3x3 cm; in relation to 31,32, 41 and 42, slightly extending into the floor of the mouth and causing lateral displacement of the above mentioned teeth.

Figure-4a:

Figure 3a:

Figure-4b: FIGURE-4a, b: CT revealed a


well-defined radiolucent expansile mass encompassing symphysis of the mandible causing splaying of the incisors; with an intact lower mandibular border. Few bony spicules were seen with in the lesion.

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Aggressive Juvenile Ossifying Fibroma

Volume 2 Issue 1 February 2011

Figure 6a: Figure-5a

Figure-5b:

Figure-6 b:

Figure-5c:

FIGURE-5a,

and

c:

Figure-6 c:
FIGURE-6a, b and c: An enbloc resection was carried out through a trans oral

Histopathological

examination

revealed

highly cellular fibrous stroma consisting of spindle to polyhedral shaped fibroblast-like cells with vesiculated nuclei were seen that are arranged in a swirling pattern and a flowing whorled pattern. The highly cellular nature of the fibrous matrix and the woven bone reflected the aggressive behaviour of the tumor.Multi nucleated giant cells were also seen.

approach followed by chemical cauterization using carbolic acid.

32 Journal of Dental Sciences and Research

Aggressive Juvenile Ossifying Fibroma

Volume 2 Issue 1 February 2011

Figure-7 a

Post-operative
FIGURE-8a, b: Follow up of one year,

showed no signs of recurrence.(Extra-oral photograph)

Figure -9a,b:FOLLOW-UP

Figure-7b:

FIGURE-7a,

b:

The

macroscopic appearance of the lesion and the teeth involved by the mass.

Figure -8a,b:FOLLOW-UP

Pre-operative

Pre-operative

Post-operative
FIGURE-9a,b: Follow up of one year,

showed no signs of recurrence.(Intra-oral photograph)

33 Journal of Dental Sciences and Research

Aggressive Juvenile Ossifying Fibroma

Volume 2 Issue 1 February 2011 active massive mandible. ossifying fibroma of with the Surg

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Reconstr

2004;113:970-4. 7. Slootweg PJ, Panders AK,

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Ossifying

fibroma,

odontogenic

tumors and allied lesions. London: Quintessence 2004. 3. Saiz-Pardo-Pinos MV, AJ, OlmedoE, Publishing Co, Ltd;

histopathological

Pathol Med 1994;23:385-8. 8. Noffke CE. Juvenile ossifying

Gaya

Prados-Snchez M.

fibroma of the mandible: An 8 year radiological Dentomaxillofac 1998;27:363-6. 9. Offiah C, Hall E. The rapidly enlarging chin mass. Br J Radiol 2005;78:175-6. 10. Williams PM. HK, Mangham C, follow-up. Radiol

Vallecillo-Capilla

Juvenile

ossifying fibroma: A case study. Med Oral Patol Oral Cir Bucal

2004;9:454-8. 4. El-Mofty S. Psammomatoid and trabecular juvenile ossifying fibroma of the craniofacial skeleton: Two distinct clinicopathologic entities.

Speight

Juvenile

ossifying

Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;93:296-304. 5. Johnson LC, Yousefi M, Vinh TN, Heffner DK, Hyams VJ, Hartman KS. Juvenile active ossifying fibroma: Its nature, dynamics and origin. Acta Otolaryngol Suppl 1991;488:1-40. 6. Zama M, Gallo S, Santecchia L, Bertozzi E, De Stefano C. Juvenile

fibroma: An analysis of eight cases and a comparison with other fibroosseous lesions. J Oral Pathol Med 2000;29:13-8.

34 Journal of Dental Sciences and Research

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