Vol. 114 No.

2 August 2012

Ossifying fibroma of the jaws: a clinical study of 14 cases and

review of the literature
Katherine Triantafillidou, MD, DDS,a Grigoris Venetis, MD, DDS,b Georgios Karakinaris, MD, DDS,c and
Fotis Iordanidis, MDd
Aristotle University of Thessaloniki and “G. Papanikolaou” General Hospital of Thessaloniki, Thessaloniki, Greece

Objective. The purpose of this study was to evaluate the clinical outcomes of ossifying fibroma (OF) in a group of 14 patients
treated in our clinic and discuss the management and the prognosis of this tumor.
Study Design. The study included 14 consecutive patients with OF of the jaws with ages ranging from 7 to 55 years. Primary
site distribution was maxilla (1), maxilla, maxillary sinus (1), and mandible (12). All of the patients treated by surgery
(curettage, enucleation, or radical surgery).
Results. The mean follow-up range was 2-18 years. All of the patients were alive and disease free.
Conclusions. OF is a benign slow-growing tumor of the jaws. Early tumors that are small or well demarcated are treated by
curettage or enucleation. For aggressive tumors that show rapid enlargement, radical resection (maxillectomy or segmental
mandibulectomy) is used. By either method of treatment, the recurrence rate is extremely low. (Oral Surg Oral Med Oral
Pathol Oral Radiol 2012;114:193-199)

Fibro-osseous lesions (FOLs) of the craniofacial bones molar region) is affected more often than the maxilla,
comprise a group of lesions composed of hypercellular whereas in the other cranial and facial bones, the peri-
fibrous elements and osseous elements, both of which orbital, frontal, ethmoid, sphenoid, and temporal bones
exhibit a wide spectrum of variations. The majority of are also familiar sites for the tumor.7,10-14
FOLs have overlapping histologic features, so the The clinical presentation of OF is usually a spherical
proper diagnosis requires good correlation of the his- or ovoid jawbone mass, painless and expansive, that
tory, clinical, and radiographic findings and histologic may displace the roots of adjacent teeth and sometimes
features. Among the more significant lesions of this cause root resorption. Large mandibular lesions may
group are fibrous dysplasia (FD) and ossifying fibroma cause a characteristic thinning and downward “bowing”
(OF).1,2 Ossifying fibroma is a fibro-osseous tumor of the inferior border as it is visible on radiographs and
affecting both jaws and composed of proliferating fi- computerized tomography (CT) scans.2-4,9 Early le-
broblasts and osseous products that include bone and sions are small and radiolucent. As they enlarge and
cementum-like material.1-4 The tumor is slow growing mature they become mixed radiolucent-radiopaque and
and well demarcated from adjacent bone. Some lesions finally completely radiopaque.2,4,15
may grow to massive size, causing considerable es- In the present study, we present the results from the
thetic and functional deformity. Because of the pres- management of 14 patients with OFs treated in our
ence of both bone and cementum-like products in OFs, clinic, with reference to the differential diagnosis and
these lesions are designated as ossifying fibroma, ce- prognosis of this tumor.
mento-ossifying fibroma, and cementifying fibroma. It
is agreed that these 3 terms describe the same lesion.4-7
METHODS
OFs occur in patients in the second to fourth decades
Fourteen consecutive cases with OF of the jaws were
of life, yet they may arise in children and adolescents as
retrospectively analyzed for age, gender, tumor loca-
well as in older adults.2,8,9 The mandible (especially the
tion, symptoms, and signs on occurrence, surgical treat-
ment, and postsurgical clinical course.
a
Associate Professor, Department of Oral and Maxillofacial Surgery, The female-to-male ratio was 9:5, and the age of the
Dental School, Aristotle University of Thessaloniki. patients ranged from 7 to 55 years. The location of the
b
Assistant Professor, Department of Oral and Maxillofacial Surgery,
Dental School, Aristotle University of Thessaloniki.
tumor was maxilla (1), maxilla and maxillary sinus (1),
c
Oral and Maxillofacial Surgeon, Department of Oral and Maxillo- and mandible (12; Table I).
facial Surgery, Dental School, Aristotle University of Thessaloniki. Diagnostic evaluation included a history and physi-
d
Pathologist, “G. Papanikolaou” General Hospital of Thessaloniki. cal examination for all patients and diagnostic imaging
Received for publication Jan. 30, 2011; returned for revision Jul. 12, of the primary lesion with plain films and CT scans.
2011; accepted for publication Jul. 18, 2011.
© 2012 Mosby, Inc. All rights reserved.
Bone swelling or expansion or cortical plate perforation
2212-4403/$ - see front matter were the most common clinical and radiologic signs
doi:10.1016/j.tripleo.2011.07.033 (Figs. 1 and 2).

193
ORAL AND MAXILLOFACIAL SURGERY OOOO
194 Triantafillidou et al. August 2012

Table I. Characteristics of the cases studied

Case no. Gender Age, y Location-symptoms Treatment Follow-up
1 Male 28 Painless buccal and lingual bone expansion 1st surgical treatment: curettage; 18 y after the radical
of the right mandible, premolar-molar local recurrence (2 y later). surgical treatment,
region 2nd surgical treatment: the patient was
segmental mandibulectomy. free of the
The defect was closed with disease.
iliac bone graft.
2 Female 7 Painless bone swelling of the right maxilla Two conservative operations At 23 y old the
and maxillary sinus (curettage). The histologic patient was free of
examination revealed OF the disease.
associated with ABC. Partial
maxillectomy in Germany
with free vascularized bone
graft that failed 6 mo later.
Finally placed iliac bone graft
and implants (in our clinic).
3 Female 16 Painless buccal bone swelling of the right Curettage 17 y later the patient
mandible, premolar region was free of the
disease.
4 Male 25 Bone swelling of the right mandible, Enucleation (total excision) 13 y later the patient
premolar region was free of the
disease.
5 Female 14 Bone swelling of the left mandible, molar Curettage 12 y later the patient
region was free of the
disease.
6 Male 54 Buccal and lingual bone expansion of the Curettage 11 y later the patient
right mandible, premolar-molar region was free of the
disease.
7 Female 41 Painless buccal expansion of the right Curettage 9 y later the patient
mandible, molar region was free of the
disease.
8 Male 11 Painless bone swelling of the right maxilla, Enucleation (total excision) 8 y later the patient
canine region was free of the
disease.
9 Female 40 Painful buccal and lingual bone expansion Segmental mandibulectomy. The 8 y later the patient
of the right mandible with perforation of defect was closed with a was free of the
lingual cortical plate, premolar-molar reconstruction titanium plate. disease.
region Six mo later the metallic plate
was removed and replaced by
an iliac bone graft.
10 Female 51 Painless bone swelling of the left Enucleation (total excision) 7 y later the patient
mandible, molar region was free of the
disease.
11 Female 43 Buccal bone swelling of the right Curettage 4 y later the patient
mandible, premolar-molar region was free of the
disease.
12 Female 37 Painless bone swelling of the right Curettage 3 y later the patient
mandible, premolar-molar region was free of the
disease.
13 Female 52 Swelling of the left mandible, molar region Enucleation (total excision) 3 y later the patient
was free of the
disease.
14 Male 55 Buccal bone swelling of the right Curettage 2 y later the patient
mandible, premolar-molar region was free of the
disease.
OF, Ossifying fibroma; ABC, aneurysmal bone cyst.

All the patients received surgical treatment. Small used for 3 patients who had large tumors with perfora-
and well demarcated tumors were treated by enucle- tion of cortical plates of the mandible or spread of the
ation or curettage (11 patients). Radical surgery with lesion in the maxillary sinus. The surgical defect in
partial maxillectomy or segmental mandibulectomy these cases with radical resection (nos. 1, 2, and 9) was
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Volume 114, Number 2 Triantafillidou et al. 195

RESULTS
The lesion was painful in only 1 patient (7%). Bone
swelling or expansion was the standard clinical sign,
presented in 100% of the patients, but buccal perfora-
tion was present only in case 9 (7%). Three of the
patients received radical surgery, and the remaining 11
patients underwent to enucleation or curettage of their
lesions. The follow-up was 2-18 years. Recurrences
have been recorded in 2 cases (nos. 1 and 2; ⬃14%). In
one of these cases (no. 2), the lesion that recurred was
initially diagnosed as ABC. The mean time of recur-
rence appeared to be ⬃2.5 years. All the patients,
independent of the surgical modality, were free of the
disease at the time of writing (Table I).

DISCUSSION
Figure 1. Typical appearance of ossifying fibroma in axial Ossifying fibroma of craniofacial bones is a benign
computerized tomographym showing well demarcated mar- neoplasm, mainly composed of 2 components: fibrous
gins, an oval or spherical expansion, and a heterogeneous stroma and bone elements that show various degrees of
density (patient no. 6). maturation. The stroma consists of fibroblasts and co-
lagenous fibers. Bone elements include mineralized
bodies (ossicles), osteoids, fiber bone (woven bone),
restored with an autogenous bone graft fixed with a and mature bone (lamellar bone).
reconstruction plate (Table I). Ossicles connect to form bone trabeculae that usually
For patient no. 2, the initial biopsy revealed a large is surrounded by osteoblasts and occasionally by oste-
cystic cavity of the maxilla with brisk venous bleeding. oclasts. Rounded cementum-like masses may be pres-
The clinical differential diagnosis included central giant ent either alone or together with the trabeculae.1,2,11,14
cell granuloma (CGCG), aneurysmal bone cyst (ABC), Because of the variation in the configuration of these
and vascular malformation. The microscopic findings calcified deposits, such tumors have been referred to as
were consistent with ABC. For this patient, a conser- both ossifying and cementifying fibroma.2-4,9,16
vative initial treatment with curettage was obligatory, Marx and Stern2 have stated that OF occurs fre-
owing to the patient’s age (7 years old). Four years quently in the jaws, probably because these lesions are
later, and 3 years after a recurrence and a new curettage related to an extensive mesenchymal cellular induction
into bone and cementum, required in odontogenesis.
of the recurred ABC, the lesion recurred again and the
Therefore, when there is an error in the tissue induction
patient developed increasing pain. A new biopsy of the
process, an OF may be developed in the jaws. It is
lesion was obtained through a Caldwell-Luc approach.
thought that some fibro-osseous lesions arise from the
The histologic diagnosis was consistent with ABC in
periodontal ligament, which contains pluripotential
association with an OF (Fig. 3). The patient went to
cells capable of forming cementum, bone, and fibrous
Germany for further treatment. The maxillary lesion tissue.1-4 The neoplastic nature of OF is attributed to
was removed by partial maxillectomy. The defect the fact that large lesions exhibit aggressive behavior,
closed with a free vascularized bone graft which failed producing significant osseous destruction. Addition-
6 months later. After 1 year, the patient returned to our ally, recurrences, though rare, have been described in
clinic, the therapeutic treatment was completed with an some cases of OF. Others categorize this lesion as a
iliac bone graft, and 2 dental implants were placed localized dysplastic process in which bone metabolism
(Table I). At the time of writing, the patient is 23 years is altered.2,3,5
old and free of the disease without functional or esthetic “Juvenile” OF is a subtype of OF that arises within
problems. the craniofacial bones of children under the age of 15
Histopathologic diagnosis of the lesions was based years. However, there are reports with patient ages
on an examination of the HandE-stained tissue sections. ranging from 3 months to 72 years.17-19 The histologic
The characteristic microscopic criteria for diagnosis of features of JOFs are distinctive and include a cell-rich
OF included the presence of spicules of woven bone in fibrous stroma containing bands of cellular osteoid
a vascularized fibrous stroma. In addition, osteoblastic without osteoblastic lining, osteoid strands and cement
rimming was usually found (Fig. 4). particles.2,9,20 The JOFs are classified into 2 distinct
ORAL AND MAXILLOFACIAL SURGERY OOOO
196 Triantafillidou et al. August 2012

Figure 2. Appearance of ossifying fibroma in panoramic radiography, showing a mixed radiolucent-radiopaque area and teeth
displacement in the lower jaw (patient no. 1).

local behavior.2,9 The aggressive nature of these lesions

with the high rate of recurrence (30%-58%) suggests
that JOF should be treated as a locally aggressive
neoplasm. Therefore, surgical resection rather than con-
servative curettage would be the preferred method of
treatment.18,19,22 None of the lesions of the present
study had histopathologic features characteristic of
JOF, although 3 of the patients were ⬍20 years in age.
In the literature there are reported cases of fibro-
osseous lesions associated with a range of other giant
cell lesions of the jaws, such as ABC and CGCG.23
This coexistence may be a reaction to a stromal change
within the original lesion.24,25 Kaplan et al.24 reported
3 cases of CGCG associated with OF among 51 in-
traosseous lesions of the jaws. They thought that in
Figure 3. Coexistence of ossifying fibroma (OF; white ar- combined lesions the primary lesion is OF and that
row) and secondary aneurysmal bone cyst with solid config- through some yet unknown trigger, the mesenchymal
uration (black arrowheads; hematoxylin-eosin, ⫻100). The spindle cells of the tumor release cytokines that induce
OF shows typical pattern of anastomosing trabeculae of wo-
differentiation toward osteoclast giant cells. El Deeb et
ven bone lined by active osteoblasts and separated by cellular
fibrovascular stroma. The black arrow indicates the cortical
al.23 found that 21% of jaw ABCs occurred in asso-
bone of origin for both lesions. ciation with other bone lesions. Trent and Byl26
found associated bone lesions in 12% of ABCs in the
jaws, and Wojno and McCarthy27 reported 5 cases of
clinicopathologic entities: the trabecular and the psam- ABC associated with fibro-osseous lesions of the
momatoid types. Trabecular JOF is characterized by the craniofacial bones. Padwa et al.28 found that 22% of
presence of trabeculae and fibrillar osteoid and woven jaw ABCs reported in the literature were associated
bone. Psammomatoid JOF is characterized by the pres- with another bone lesion, as fibrous dysplasia, ossi-
ence of small uniform spherical ossicles that resemble fying fibroma, or giant cell tumor.
psammoma bodies.4,21,22 The clinical management and It is thought that the ABC may develop secondary to
the prognosis of the JOF is uncertain. Some may pres- a hemorrhagic “blow out” of a preexisting bone lesion
ent with minimal symptoms, and others, particularly in (ABC “plus”). The initial lesion may remain in part or
very young children, may present with “aggressive” may be totally destroyed.24,28 Buraczewski and Dab-
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Volume 114, Number 2 Triantafillidou et al. 197

the lesions were large and expanded. The outcome of

the curettage for these 2 patients was recurrence, so the
next surgical resection was radical (maxillectomy or
segmental mandibulectomy) without recurrence for
⬎15 years. In any case, the decision on whether to
enucleate or resect radically, depends on involvement
of the inferior border of the mandible and the spread of
the lesion in the soft tissues or in the maxillary sinus
and nasal cavity.2,7,31,32 Both surgical methods of treat-
ment for OF (conservative or radical) are acceptable by
most authors in the English-language literature during
the past 30 years, as reported in their clinical stud-
ies,1,3,5,31-33 (Table II). Conservative surgery was ad-
dressed for small lesions, whereas larger lesions re-
quired radical surgery. The above surgical protocol was
also applied for the patients of the present study. Three
patients (nos. 1, 2, and 9) were treated with radical
resection (segmental mandibulectomy or partial maxil-
lectomy), 7 with enucleation and curettage, and 4 with
enucleation alone. The CT scan of these 11 cases that
were treated with conservative procedures showed well
delineated margins and an oval or spherical expansion
of the bone. In all cases, the alveolar nerve was re-
served and the outcome of curettage or enucleation was
successful.
By either method of treatment, the recurrence rate for
OF is low.2,9 For aggressive tumors that show rapid
enlargement, the recurrence rate is estimated to be
20%-25%. Such tumors will usually require radical
Figure 4. A, Ossifying fibroma is composed of spicules of resection. It is advisable that the clinician follow these
woven bone of varying shape are set in a vascularized fibrous patients with yearly examination.1,2,4,5 The recurrence
stroma (hematoxylin-eosin [HE], ⫻200). B, The stromal cells rate in the present patients (14%) is in accordance to
in ossifying fibroma evolve into rimming of osteoblasts,
that estimated in literature.
which line the bone spicules. This feature serves to make the
distinction between ossifying fibroma and fibrous dysplasia
Differential diagnosis between OF and other fibro-
(HE, ⫻400). osseous lesions sometimes is difficult, because all of
these lesions may exhibit similar clinical, radiographic,
and histologic features.4,7,9 Distinguishing between fi-
ska29 thought that the “new” lesion almost totally oblit- brous dysplasia (FD) and OF is the primary differential
erates the traces of the “older” lesion. Therefore, the diagnostic challenge. The most helpful feature distin-
distinction between “pure” ABC and ABC “plus” is guishing FD and OF is the well circumscribed appear-
difficult, and thorough histologic examination of the ance of OF in radiography and the ease with which it
entire specimen is essential. Martinez and Sissons30 can be separated from normal bone in surgery. Histo-
found that the combination of ABC and a neoplasm was logically, FD is reported to contain only woven bone,
more common in widely resected specimens than in without evidence of osteoblastic rimming of bone. The
curetted samples. For patient no. 2 of the present study, presence of mature lamellar bone in histology is be-
the second biopsy of the lesion revealed an ABC in lieved to be characteristic of OF.1,2,4,9 If the OF occurs
association with an OF (Table I). around tooth roots, it may also resemble a cemento-
The treatment of choice for OF is surgical excision. blastoma or florid cemento-osseous dysplasia. These
Small and well demarcated lesions can be excised by rare lesions may be distinguished from an OF by their
enucleation and curettage, whereas larger lesions, that radiographic appearance. The cementoblastoma is
show a more aggressive pattern, especially in the max- fused to the root of the involved tooth, and florid
illa, require radical surgery within healthy margins.2,5,8 cemento-osseous dysplasia exhibits not 1 but several
The initial treatment for 2 of our patients (nos. 1 and 2) sclerotic densities in the alveolar bone of one or both
was conservative surgical excision (curettage) although jaws.2,33
ORAL AND MAXILLOFACIAL SURGERY OOOO
198 Triantafillidou et al. August 2012

Table II. Clinical studies of ossifying fibroma (OF) in the past 30 years: a review of the literature
Authors and no.
of cases Location of OF Treatment Follow-up
Zachariades et al.: Mandible (9) Complete removal of the lesion (14 cases). 1 recurrence 1.5 y after the
16 Maxilla (7) Resection of the mandible and iliac bone original operation
graft (2 cases).
Eversole et al.: 64 Mandible was the site of predilection Surgical curettage or enucleation the initial The recurrence rate after
accounting for 89% of the cases. treatment of choice. curettage was found to be
28%.
Sciubba and Mandible (14) Curettage and peripheral osteotomy (2 12 mo to 21 y, 1 recurrence
Younai: 18 Maxilla (4) cases). Subtotal mandibulectomy (1 (a simple bone cyst in
case). Excisions as conservative as association with the
possible (13 cases). 1 case untreated and neoplastic lesion)
1 case resection of the mandible
(corticocancellous graft from iliac crest).
Su et al.: 316 From the 316 cases, 75 characterized More than one-half of lesions treated by No data
as cemento-ossifying fibroma or as enucleation or block resection
OF according of the
histopathologic features
Mintz and Velez: Mandible (18) Curettage or resection when the inferior The recurrence rate was low.
20 Maxilla (2) border of the mandible was involved
Two of the patients presented and the lesion did not appear to be well
bilateral lesions. circumscribed
Chang et al.: 28 Mandible (26) Conservative total excision (17 cases). No lesion recurrence was
Maxilla (2) Enucleation (3 cases). Curettage (4 found.
cases). Segmental mandibulectomy (1
case). Partial maxillectomy (2 cases). 1
case only incisional biopsy.

In conclusion, OF is a benign fibro-osseous tumor, 7. Vegas-Bustamante E, Gargallo-Albiol J, Berini-Aytés L, Gay

most commonly found in the jaws, especially in the Escoda C. Benign fibro-osseous lesions of the maxillas: analysis
of 11 cases. Med Oral Patol Oral Cir Bucal 2008;13:E653-6.
mandible. Buccal or lingual cortical bone swelling or
8. Gurol M, Uckan S, Guler N, Yatmaz PI. Surgical and recon-
expansion is the most common clinical feature. The structive treatment of a large ossifying fibroma of the mandible
radiographic picture of OF is more frequently a well in a retrognathic patient. J Oral Maxillofac Surg 2001;59:
defined mixed lesion (radiolucent/radiopaque). Most 1097-100.
OFs can be treated by conservative surgical excision. 9. Stewart JCB. Benign nonodontogenic tumors. In: Regezi JA,
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extremely low, the patients must be followed carefully, tions. 3rd ed. Philadelphia: Saunders; 1999: p. 357-60.
10. Cheng C, Takahashi H, Yao K, et al. Cemento-ossifying fibroma
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sive, recurring sometimes after conservative surgical review. Acta Otolaryngol Suppl 2002;547:118-22.
procedures. 11. Fu YS, Perzin KH. Nonepithelial tumors of the nasal cavity,
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