Kor J Oral Maxillofac Pathol 2012;36(4):243-250

하악 전방부에 발생한 거대 중심성 골화성 섬유종

문성용1)*, 김수관1), 김학균2), 윤정훈3)**

조선대학교 치과대학 구강악안면외과학교실1), 충남대학교 의과대학 치과학교실 구강악안면외과2)

원광대학교 치과대학 대전치과병원 구강병리과3)

<Abstract>

Huge Central Ossifying Fibroma on the Anterior Mandible

: A Case Report

Seong Yong Moon1)*, Su Gwan Kim1), Hak Kyun Kim2), Jung Hoon Yoon3)**

Department of Oral and Maxillofacial Surgery, School of Dentistry, Chosun University Dental Hospital1),

Department of Oral and Maxillofacial Surgery, School of Medicine, Chungnam National University Hospital2),

Department of Oral & Maxillofacial Pathology, College of Dentistry, Daejeon Dental Hospital, Wonkwang University3)

The ossifying fibroma (OF), with the microscopic features of trabuclae or spherules of bone or cementum-like material in a cellular

fibrous connective tissue stroma, is one of the most common benign fibro-osseous lesions in the jaw bones. The OF often occurs in

patients from 20 to 40 years of age, which is a definite female predilection. The mandibles are involved far more often than the

maxillas, especially the pre-molar and molar regions. It is slow-growing, bone producing, asymptomatic and well-demarcated. The OF

is a disorder of odontogenesis or osteogenesis ascribed to bone marrow stroma cells (BMSCs) abnormality. However, the detailed

mechanisms of OF’s oncogenesis, cytodifferentiation, and tumor progression remain unknown. In this article, we reported a huge cen-

tral OF on the anterior mandible. The lesion was enucleated and peripheral ostectomy was done via intraoral approach and re-

constructed with vascularized iliac block bone graft. After 25 months of follow up, the tumor had not recurred. This case shows that

OF may be successfully treated by conservative surgical enucleation and peripheral ostectomy.

Key words：Ossifying fibroma, Mandible, Surgical enucleation

*Correspondence: Seong-Yong Moon, Department of Oral and Maxillofacial Surgery, Chosun University Dental Hospital, Philmoondaero 303, Seo-seok Dong, Dong-gu, Gwang-Ju, 501-759, South Korea. Tel: 82-62-220-3810, E-mail: msygood@hanmail.net

** Correspondence: Jung-Hoon Yoon, Department of Oral and Maxillofacial Pathology, Wonkwang University Daejeon Dental Hospital, Doonsan-Ro 77.Tel: 82-42-366-1146, E-mail: opathyoon@wku.ac.kr

본 연구는 2012년 조선대학교 치과병원 연구비 지원으로 이루어진 것임.

Received: Jun 13, 2012; Revised: Jun 18, 2012; Accepted: Jun 27, 2012

Ⅰ. Introduction

Ossifying fibroma (OF) is a relatively rare benign neo-

plasm of the jaws, composed of connective tissue of mixed

and variable cellularity, with a mineralized component that

consists of trabecular or woven bone1). OF is one of the be-

nign fibro-osseous lesions included in a heterogeneous

group of bone disorders affecting the craniofacial bones2).

OF was first described by Menzel in 1872 and the term

‘ossifying fibroma’ was subsequently coined by Montgomery

in 19273). This lesion was then thought to be a histological

variant of fibrous dysplasia, but it was later separated into

a distinct clinico-pathological entity.

The OF and cementifying fibroma have been grouped

under a single designation as cemento-ossifying fibroma in

the 1992 World Health Organization (WHO) classification4).

244

The tumor is defined as a demarcated, occasionally

encapsulated lesion consisting of fibrous tissue that contains

variable amounts of mineralized material resembling bone or

cementum4,5). It shows a female predilection, and most cases

are seen at the third and fourth decades of life. The

premolar and molar region of the mandible is the most

common site1). Most cases of active OF are asymptomatic,

as is reflected in the present case, and the first clinical

manifestation is a swelling of the mandibular cortical layer,

which produces a marked extra-oral facial asymmetry6). The

essential characteristics of this clinical entity are as follows:

the early age of onset, the bone pattern, the high tendency

to recurrence and the aggressive local behavior. Sometimes,

these tumors may reach a very large size7). Such cases may

require additional reconstructive surgery because of some

aesthetic and functional problems, especially when teeth are

removed8,9).

Radiographically, The lesions most often are well defined

and unilocular, and are either completely radiolucent or

mixed, depending on the amount of calcification, or are

completely radiopaque and surrounded by a radiolucent rim.

In each type, there is a sclerotic border around the lesion.

Multilocularity is rare. Root divergence and resorption are

not uncommon10)

.

The treatment of choice is always surgery. Small lesions

are treated with conservative excision and the circumscribed

nature of the lesion permits complete local enucleation or

curettage. Whereas larger lesions that have destroyed a

considerable amount of bone, especially those in the

maxilla, may show a more aggressive pattern and require

radical surgery as segmental resection11). Mandibular lesions

have a recurrence rate of 28% after curettage1). To avoid or

minimize the chance of recurrence, en bloc resection or

partial resection of the jaw is generally preferred1,8,12,13).

In this report, aggressive large ossifying fibroma on the

mandible shows that OF could be diagnosed by combination

of clinical and radiologic and histopathologic examination.

And OF cloud be treated by surgical enucleation and

peripheral ostectomy successfully, and immediate

reconstruction was performed.

Ⅱ. Case report

A 38-year-old woman taken the panorama in local clinic

and she referred to our department because of facial

swelling on the anterior mandibular area. In panoramic and

CT view, she had radiolucent-radiopaque mixed lesion

surrounded by radiolucent rim from #36 to #45 area, and

loss of lamina dura of from #33 to #43 teeth, and expansion

and thinning of buccal and lingual cortical bone (Fig 1). The

CT findings favor an OF (Fig 2). The patient was not aware

of its presence until an examination. The surrounding soft

tissue did not show any pathologic appearance.

The lesion was biopsied by sulcular and vertical releasing

incision on the mandibular symphyseal area; the results of

which showed an OF, that showed bony trabeculae with

osteoblastic rimming and fascicular proliferation of spindle

cell (Fig 3). In September 4th, 2008, under general

anesthesia, a subperiosteal surgical enucleation was

performed and peripheral ostectomy about 1 mm at the all

enucleated area with piezosurgery (Fig 4-6). The immediate

reconstruction was performed with a deep circumflex iliac

arterial osseous iliac crestal flap harvested from the left ilium

and positioned through intraoral approach (Fig 7). A small

incision of the skin at the left submandibular area for

exposing the facial vessel used for the anastomosis.

The subsequent histologic examination showed the lesion

to be characterized by a dense cellular proliferation of

polyhedral and spindle-shaped cells arranged in a whorled

pattern in which trabuculae of primary bone with occasional

osteoblastic rims and granules of cells of the osteclastic type

were also present. The overall picture led to the diagnosis

of an OF. 1 year and 2 year after the surgery, panoramic

245

Fig. 1. An initial lesion

of the panoramic radio-

graph.

Fig. 2. Conbeam and 3D

CT images showed the

lesion involving anterior

mandiblar area and caus-

ing expansion and de-

struction in mandible

symphysis area

246

Fig. 3. Photomicrographs

of tumour showed the

presence of trabeculae

of fibrillar osteoid, woven

bone and osteoblastic

rimming.

Fig. 4. Intraoperative

photograph showed ex-

posed huge tumor via in-

traoral approach.

Fig. 5. Huge tumor mass

including mandibular ante-

rior teeth was enucleated.

Fig. 6. This photograph

showed intraoperative

state after surgical enu-

cleation and peripheral os-

tectomy

Fig. 7. This intraopera-

tive photograph showed

reconstructed state of

mandible using DCIA

flap.

and CT view showed the absence of recurrence and

satisfactory reconstruction of the mandible (Fig 8-11). And

now we have a plan to install implant at the reconstructed

mandible for rehabilitation of the lower dental arch.

Ⅲ. Discussion

OF affected females more frequently, and mandible was

three times more prevalent than maxilla. The mean age at

first presentation was 31 years, the decade with greated

frequency was the fourth. Females were in the majority

except in the first decade. The main symptom was swelling

(66%). 31% were found incidentally. 84% of cases displayed

buccolingual expansion; half of the mandibular cases

exhibited downward displacement of the lower border of

the mandible and 90% of maxillary cases involved the

maxillary antrum. 12% of cases recurred or were

247

Fig. 8. This is panoramaic radiograph of 1 year after surgery.

There was no evidence of recurrence.

Fig. 9. This is panoramaic radiograph of 2 year after surgery.

There was no evidence of recurrence.

Fig. 10. These con-

ebeam and 3D CT images

showed reconstructed

mandible with DCIA flap

2 years after surgery.

248

Fig. 11. These intraoral

photographs showed sa-

tisfactory healed and

flexible denture adapted

state of the patient 2

years after surgery.

reactivated16,17). Radiographic features are non-specific and

typically consist of an unilocular or multilocular radiolucecy

having ill-defined borders and occasional central

opacification. Aggressive lesions may show cortical thinning

and perforation.

OFs share many pathological features with fibrous

dysplasia15). The normal bone architecture is replaced by

fibroblasts and collagen fibers containing variable amounts

of mineralized material. In an attempt to separate these

entites for prognosis and treatment, radiologic differentiation

was introduced16,17). The histopathological distinction

between fibrous dysplasia and OF of the craniomaxillofacial

bones is a best of debate19). The following parameters are

used to separate them: lesional circumscription, variability in

tissue composition within the lesion from one microscopic

field to another, presence of bone maturation from woven

to lamellar, prevelance of osteoblastic rimming around the

bone trabeculae, and the configuration of the lesional bone

trabeculae. However, differentiation of solitary lesions of OF

and fibrous dysplasia can be quite difficult on histologic

grounds alone, but the lesions generally can be distinguished

if radiographic and clinical criteria are used together with an

analysis of histopathology of a biopsy specimen from the

central part of the lesion. Fibrous dysplasia has a diffuse

margin radiographically; OF is an expansile process with a

clearly defined cortical margin (being a benign tumor)22).

In this patient, there were the similar appearance in

histopathologic features of fibrous dysplasia and OF. The

radiologic features, however, were in favor of an OF. OFs

are radiologically expansile lesions with sharp demarcation

from the adjacent bone. It usually shows larger nonossified

areas of fibrous tissue. Discrete areas of calcification and

ossification may be evident. Aggressive lesions may show

massive expansile growth17). This is in comparison with

fibrous dysplasia, which shows diffuse changes and margins.

OFs show varying degrees of radiographic density

depending on the amount of calcification and ossification.

Aggressive lesions tend to show less calcification18). On CT,

the proportion of soft or fibrous tissue and calcification and

ossification is variable. Expansile or aggressive lesions may

thinning the wall of the mandibular cortex.

OF is a slow growing, asymptomatic, neoplasm that can

reach a very large size. The treatment of choice is always

surgical intervention. Lesions have been reported as being

removed by radical resection or conservatively by local

excision or enucleation with curettage23-25). Small lesions

area treated with conservative excision, whereas larger

lesions have a recurrence rate of 38% after curettage1). To

avoid or minimize the chance of recurrence, en bloc

resection or partial resection of the jaw is generally

preferred12,26-28)

. The recurrent potential of the lesion,

application of a local fixative (Carnoy’s solution) was used

after curettage of the lesion, which has shown to be

successful in large OF cases13)

. OF favors conservative

249

surgery rather than en block resection in well demarcated

with radiolucent rim29). In these lesions are usually having

definite radiolucent rim, it means that it can be separated

easily from surrounding tissue and well encapsulated lesion.

In this case, the lesion was enucleated via intra-oral

approach with involved teeth, and peripheral ostectomy was

performed approximately 1 mm at entire surface of the

margin, and also shaped the rectangular form for

reconstruction with free vascularized iliac bone. The defect

was larger than 5cm, so free vascularized bone graft using

deep circumflex iliac arterial osseous flap (7*2 cm) was

performed immediately. The relationship between histologic

features and clinical behavior is not sufficiently30). We have

not been able to trace any documented report of a case of

aggressive OF transforming into osteosarcoma, a possibility

mentioned by Hoffman et al31).

Ⅳ. Conclusion

OF is a relatively rare benign neoplasm of the jaws,

composed of connective tissue of mixed and variable

cellularity with a mineralized component that consists of

trabecular or woven bone. The essential characteristics of

this clinical entity are as follows : the early age of onset, the

bone pattern, the high tendency to recurrence and the

aggressive local behavior. The diagnosis of OF is

radiologically expansile lesions with sharp demarcation from

the adjacent bone. Surgical excision is usually selected for

treatment to avoid recurrence, but peripheral ostectomy or

partial mandibulectomy of the jaw is sometimes preferred.

In this case, aggressive large OF on the anterior mandible

was treated by surgical enucleation and peripheral

ostectomy about 1mm using piezosurgery and simultaneous

reconstruction was done with iliac crestal osseous flap.

Successful result was obtained with no reccurrence during

2 years follow up. This study intend to report this case with

systemic reviews.

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