JOURNAL OF ORAL & MAXILLOFACIAL RESEARCH Keles et al.

Juvenile Ossifying Fibroma of the Mandible: a Case Report

Bahar Keles1, Mutlu Duran1, Yavuz Uyar2, Ahmet Azimov1, Abdullah Demirkan1, Haci Hasan Esen3
1
Department of Otorhinolaryngology and Head & Neck Surgery, Meram Medical Faculty, Selçuk University, Konya, Turkey.
2
Department of Otorhinolaryngology and Head & Neck Surgery, SSK Okmeydani Education Hospital, Istanbul, Turkey.
3
Department of Pathology, Meram Medical Faculty, Selçuk University, Konya, Turkey.

Corresponding Author:
Bahar Keles
Selçuk Üniversitesi, Meram Tıp Fakültesi
Kulak Burun Boğaz Hastalıkları Servisi
42090 Meram, Konya
Turkey
Phone: +90 332 2236646
Fax: +90 332 3236181
E-mail: baharkeles@gmail.com

ABSTRACT

Background: Fibro-osseous lesions of the jaws, including juvenile ossifying fibroma, pose diagnostic and therapeutic
difficulties due to their clinical, radiological and histological variability. The aim of this study was to report the outcome of a
9 years old girl with diagnosed juvenile ossifying fibroma treatment.
Methods: A 9 years old girl presented with a 6 x 8 cm sized hard fixed tumour on right ramus and corpus of the mandible.
On the radiological examination tumour showed an irregular but well bordered, unilocular and expansive lesion on the right
corpus and ramus of the mandible. There was no teeth displacement or teeth root resorbtion. Microscopically, the tumour had
trabeculae, fibrillary osteoid and woven bone. After the clinical, radiological (panoramic radiography, computed tomography
and magnetic resonance imaging) and histologic analysis it was diagnosed juvenile ossifying fibroma. In the history of the
patient there has been an acute lymphocytic leukaemia in the remission for 3 years.
Results: Because of large size of mandibular tumour, resultant expansion and destruction of mandibular cortex, the patient
underwent right hemimandibulectomy using transmandibular approach. There was no recurrence or complications for two
years follow-up.
Conclusions: Although juvenile ossifying fibroma is an uncommon clinical entity, its aggressive local behaviour and high
recurrence rate means that it is important to make an early diagnosis, apply the appropriate treatment and, especially, follow-
up the patient over the long-term.

Keywords: mandibular diseases; mandibular neoplasms; fibroma, ossifying; surgery, oral; lymphocytic leukemia.

Accepted for publication: 7 March 2010

To cite this article:
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 (Apr-Jun);1(2):e5
URL: http://www.ejomr.org/JOMR/archives/2010/2/e5/e5ht.pdf
doi:10.5037/jomr.2010.1205

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JOURNAL OF ORAL & MAXILLOFACIAL RESEARCH Keles et al.

sclerotic shell of bone. It appears locally aggressive with

INTRODUCTION cortical disruption and involvement of many adjacent
anatomical structures. This lesion has predominating
Fibro-osseous lesions of the cranial and facial bones are soft tissue consistency with variable amounts of
usually benign and tend to grow slowly. Benign fibro- internal calcification and/or linear or irregular focal
osseous lesions have similar histopathological features bone [2]. It usually shows a low density mass due to
with fibrous dysplasia, ossifying fibroma, and cemento- cystic changes on computed tomography (CT) scans.
ossifiying dysplasia [1,2]. Following intravenous injection of iodinated contrast,
Ossifying fibroma, a rare tumour entity, is a well the lesion may show diffuse appearance enhancement
demarcated benign fibro-osseous tumour with capsule [2]. Magnetic resonance imaging (MRI) is important for
composed of metaplastic bone, fibrous tissue and the lesion extent evaluation, but is inadequate for bony
varying amounts of osteoid [3,4,5]. The ossifying components. It is isointense on T1-weighted images
fibromas are subdivided into conventional and juvenile and hypointense on T2-weighted images. Following
clinicopathologic subtypes [3]. Conventional ossifying gadolinium injection, there is homogeneous tumour
fibromas are usually slow growing and generally seen appearance enhancement [2].
in the third and forth decades of life [6,7]. They are Histologically, JOF is characterized by the presence of
treated with simple curettage and the recurrence is rare cellular fibrous strom, garland like bony strands and
[8]. It affects people of all ages, but in contrast to the cement particles [2,6,11,13]. The JOFs are classified
form seen at adults, the juvenile form is clinically more into two distinct clinicopathological entities: the
aggressive and tends to be recurrent [3]. trabecular and the psammomatoid types. Trabecular
According to the new edition of the classification of JOF is distinguished by the presence of trabeculae of
the World Health Organization [9], ossifying fibromas fibrillar osteoid and woven bone and psammomatoid
which appear as fast growing mass between 5 and JOF is characterised by the presence of small uniform
15 years of age, radiologically well bordered, and spherical ossicles that resemble psammoma bodies [15].
consistent with ossifying fibroma histologically, are Psammomatoid JOF is reported more commonly than
referred as juvenile (aggressive) ossifying fibroma. trabecular JOF [14,16]. Psammomatoid JOF occurs
Juvenile ossifying fibroma (JOF) appears at an early age predominantly in the sinonasal and orbital bones,
and in 79% of the patients are diagnosed before the age and trabecular JOF predominantly affects the jaws.
of 15 [2,3,10]. Males and females are equally affected Psammomatoid JOF has aggressive behaviour and it
[11]. JOF originates from periodontal ligament and has a very strong tendency to recur [15-17].
ranges 2% of oral tumours in children [13]. The JOF An accurate diagnosis of JOF is made by correlating the
is located mainly (85%) in facial bones, in some cases clinical, CT scan, MRI and histopathological findings
(12%) in calvarium and very seldom (3%) extracranially [2]. Authors herein presented a case of juvenile ossifying
[2]. Ninety percent of the lesions located in the face fibroma of the mandible which caused expansion and
region, involve the sinuses, mainly the maxillary antra destruction of mandibular cortex.
[2]. Mandibular lesions are seen in 10% of the cases
[2,14]. The tumour is well circumscribed by a tiny

Figure 1. Photograph of a 9 year old girl with JOF showing Figure 2. Photograph of mandibular ramus and corpus region
unilateral swelling extending from the right submandibular to the showing clear lingual expansion of the mansoble (arrow).
right mandibular ramus and corpus region.

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Figure 3. CT axial image shows a lesion involving submandibular Figure 4. MRI axial image shows a large tumour which causes
area and causing expansion and destruction in right ramus and destruction in the right ramus and corpus of the mandible (arrow).
corpus of the mandible (arrow). Pharyngeal air column displaced
to opposite site.

CASE DESCRIPTION AND RESULTS

A 9 year old girl applied to the Department of

Otorhinolaryngology and Head & Neck Surgery, Selçuk
University, Konya, Turkey, complaining of a swelling
on the right side of her lower jaw lasting three months.
She also felt a pain and inflammation in this area.
Medical history revealed acute lymphocytic leukaemia
presenting in remission for 3 years.
Physical examination revealed a hyperaemic swelling
about 6 x 8 cm in size, causing facial asymmetry in the
region of submandibular area including right corpus and
ramus of the mandibule (Figure 1). Assessment with
palpation showed a hard, nontender mass with smooth
surface adhered to the mandible. The mouth opening Figure 5. MRI coronal image shows contrast retention in the
of the patient was normal and there were no decayed tumour (arrow) after gadolinium injection. Pharyngeal air column is
displaced to opposite site.
teeth in the lesion area, but there was malocclusion.
There was clear lingual expansion of the right mandible
(Figure 2). Oral hygiene was good. The right palatine
tonsil was deviated to the left. There were no assessed
pathological changes of the mucous membrane in the
tumour region.
Panoramic radiograph showed an irregular but well
bordered, unilocular, expansive lesion of the right
corpus and ramus of mandible. There was no teeth
displacement or teeth root resorbtion. There were
registered deciduous right mandibular canine and
first and second premolars. However, there were no
deciduous teeth in the left side.
The CT scan of mandibular tumour showed a solid
hypodense mass that enlarged the submandibular area,
filled the pterygoid fossa and right masseter muscle Figure 6. Photomicrograph of tumour shows the presence of
region. The tumour was occupied and destructed the trabeculae of fibrillar osteoid and woven bone (hematoxylin and
right corpus and ramus of the mandible (Figure 3). eosin stain, original magnification x40).

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Figure 7. Intraoperative photograph showing exposed

tumour using a transmandibular approach.

Figure 10. Three-dimensional CT scan of the patient after

right hemimandibulectomy.

Figure 8. Intraoperative photograph showing right

hemimandibulectomy.

Figure 11. Three-dimensional CT scan shows good jaws

relationship and occlusion in the left side.

Figure 9. Photograph of gross surgical specimen of about

13 x 8.5 x 6.5 cm in size.

The heterogenic mass lesion caused destruction of

the right ramus of mandible was seen on MRI. It was
hypointense on T1-weighted images and hyperintense
on T2-weighted images (Figure 4). Pharyngeal air
column, hyoid bone and larynx were displaced to the
opposite side (Figure 5). There was clear contrast
retention following intravenous gadolinium injection.
The incisional biopsy was taken from the lesion and
the pathology process was reported as JOF. Diagnosis
was based on the presence of trabeculae of fibrillar
osteoid and woven bone fragments (Figure 6). Because
of large size of the mandibular tumour, the resultant Figure 12. Photograph showing patient’s postoperative
expansion and destruction of mandibular cortex, and appearance.

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the close adjacency to the temporomandibular joint, the bone lesion with sclerotic boundary, abnormal soft tissue
patient underwent a right hemimandibulectomy using mass and aggressive bone destruction is seen in the
transmandibular approach (Figures 7 - 9). No recurrence osteosarcoma, and cystic lesion connected to premolar
was observed during the 2 years of follow-up with three or molar teeth is seen in odontogenic tumours [3].
dimensional CT scan and MRI (Figures 10 and 11). Oral The JOF is characterized as expansive, having defined
functions of the patient (chewing, eating and speaking) sclerotic borders, locally aggressive and destructive
were appearing intact (Figure 12). at cortex on CT scan. This lesion is observed as a soft
tissue mass with internal calcification, linear or irregular
bone focuses [2,22]. An increase in diffuse contrast is
DISCUSSION seen after intravenous injection [22]. Contrast increase
is seen on adventitia on MRI [14]. While aggressive
Most benign fibro-osseous lesions of jaws are cortical changes are seen in juvenile form, sclerotic
asymptomatic and slowly progressing. Moreover, an changes are more common in adult form [2]. JOF is
unusual clinical presentation with apparent aggressive isointense in T1-weighted images and hypointense or
and destructive growth may be expected when the isointense in T2-weighted images. Cystic areas can
lesion is encountered in a younger patient, especially be identified. Following gadolinium injection, a slight
below the age of 15 years [24,26]. increase in contrasting is seen [2,22]. In present case,
The JOF is a fibro-osseous lesion that occurs in the the lesion was hypointense in T1 and hyperintense in
facial bones [1,2]. It is also called aggressive ossifying T2 on MRI and there was clear contrast retention after
fibroma due to its aggressiveness and the high tendency the injection of contrast agent. These findings suggest
to recur, unlike other fibro-osseous lesions, such as that there was acute lymphocytic leukaemia in the
cemento-ossifying fibroma, which may resemble patient’s history. However, after the incissional biopsy
radiographically [18]. Due to its distinct histological obtained from the lesion the final diagnosis of JOF was
features, JOF has been recognized as a separate recognized.
histopathological entity among the fibro-osseous group Ong and Siar [23] presented JOF as a progressively
of lesions [9]. growing lesion that can attain an enormous size with
JOF is a relatively rare fibro-osseous lesion of the jaws resultant deformity if left untreated. They presented a
characterized by the early age of onset i.e., under 15 case of large cemento-ossifying fibroma involving the
years of age, the location of tumour, and the radiological left mandible in a 15 year old male patient. The long
appearance and the tendency to recur [28]. lasting history of untreated JOF resulted to spontaneous
JOF affects both males and females equally without fracture of mandible. Furthermore, if JOF do not have
any significant gender predilection. However some adequate surgical treatment, it may have high rate of
researches showed that it is more common among recurrence [4,24]. The recurrences are generally seen
men [19]. In contrast, Johnson et al. [20] stated that at early stage and they are more aggressive when
mandibular tumours are more frequently common in compared to primary lesions [4].
girls between the age of 5 - 11 or during the second to There is no consensus on the treatment of JOF cases.
fourth decades of life [6]. In present paper 9 years old Radical resection, local excision conservatively or
girl was presented. enucleation with curettage are among the treatment
A few cases of facial trauma have been suggested as a alternatives [4,13,25]. Slootweg and Müller [10]
possible etiologic factor in the JOF development [10]. suggested that there were no differences between
There was no trauma in anamnesis of present patient, the cases that have limited surgical treatment and
but there was an abscess previously drained from this those with major surgery in terms of results, and they
area. recommended conservative surgery. On the other hand,
Noffke [21] after 8 year follow-up of a juvenile Waldron et al. [26] suggested that local excision and
ossifying fibroma in the left mandible of a 4 year old curettage should be a more preferable method and
boy demonstrated initial lack of radiological evidence added that local surgical excision can be applied for
of demarcation and subsequent eccentric enlargement, recurrent tumour treatment. However, rate of recurrence
selective tooth displacement and a multilocular after conservative treatment was reported in 30 - 58% of
appearance in areas of active growth. Additionally, an cases [4,27,28]. Incomplete resection causes recurrence
aneurysmal bone cyst and a decrease in the bone content in aggressive tumours. Therefore, some authors were
were presented in the excision specimen. Furthermore, recommended en block resection as an adequate
osteblastoma, osteosarcoma and odontogenic tumours treatment [12,28]. Curettage together with peripheral
should be considered in the differential diagnosis of JOF. osteotomy or sometimes segmental mandibular
While the osteoblastoma is radiologically seen as cystic resection and mandibular reconstruction are suggested in

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prevalent or recurrent cases [4]. Sarcomatous the patient was achieved. There was no recurrence or
degeneration is reported to develop in lesions that have complication during two years of follow-up period.
recurrence in long term [18]. In contrast, Espinosa et al.
[4] reported a case of unusual bone regeneration after
resection of JOF. Secondary mandibular reconstruction CONCLUSIONS
with autogenous grafts was delayed due to the rapid
bone formation. Although juvenile ossifying fibroma is an uncommon
Because the large size of the mandibular tumour, resultant clinical entity, its aggressive local behaviour and high
expansion and destruction of mandibular cortex, the recurrence rate mean that it is important to make an
patient underwent right hemimandibulectomy using early diagnosis, apply the appropriate treatment and,
transmandibular approach. Zama et al. [8] in a similar especially, follow-up the patient over the long-term.
case performed resection and reconstruction keeping the
mandibular tissue around the temporomandibular joint.
However, in present case, it was necessary to perform ACKNOWLEDGMENTS AND DISCLOSURE
right hemimandibulectomy due to close localisation of STATEMENTS
the tumour to the tempormandibular joint and absence
of adequate reliable surgical border. Despite of that The authors report no conflicts of interest related to this
the oral functions of the patient remained adequate. study.
Furthermore, cosmetically tolerable appearance of

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To cite this article:

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(2):e5
URL: http://www.ejomr.org/JOMR/archives/2010/2/e5/e5ht.pdf
doi:10.5037/jomr.2010.1205

Copyright © Keles B, Duran M, Uyar Y. Azimov A, Demirkan A, Esen HH. Accepted for publication in the JOURNAL OF
ORAL & MAXILLOFACIAL RESEARCH (http://www.ejomr.org), 7 March 2010.

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included.

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