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Odontogenic fibroma is a benign ectomesenchymal tumor classified as central and peripheral on the basis of its location and as an epithelium rich or epithelium poor based on its histological features. Radiological findings consist of radiolucent areas with well-defined bony margins. The lesion is detected early because of its location and usually treated with surgical excision and curettage. We present a case of giant odontogenic fibroma of right maxilla presenting as gross facial deformity and posing a dual challenge of excising the tumor mass and reconstructing the ensuing defect.
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Annals of Maxillofacial Surgery | July - December 2014 | Volume 4 | Issue 2 211
Giant odontogenic fibroma of maxilla
Brajendra Baser, Arvind Kinger, Geeti V. Mitra1, Manya Thakur Roy
Departments of ENT and 1Oral and Maxillofacial Surgery, SAMC and PG Institute, Indore, Madhya
Pradesh, India
Address for correspondence:
Dr. Brajendra Baser, Department of ENT, SAMC and PG Institute, Indore,
Madhya Pradesh, India.
E-mail: baserbv@gmail.com
Odontogenic fi broma is a benign ectomesenchymal tumor classifi ed as central and peripheral on the basis of its location and
as an epithelium rich or epithelium poor based on its histological features. Radiological fi ndings consist of radiolucent areas
with well-defi ned bony margins. The lesion is detected early because of its location and usually treated with surgical excision
and curettage. We present a case of giant odontogenic broma of right maxilla presenting as gross facial deformity and posing
a dual challenge of excising the tumor mass and reconstructing the ensuing defect.
Keywords: Giant, maxilla, odontogenic fi broma, reconstruction
INTRODUCTION
Odontogenic broma is a benign mesodermal tumor originating
from the dental follicle, periodontal ligament or dental papilla.[1]
WHO has de ned this lesion as a rare neoplasm consisting of
collagenous brous tissue with varying amount of odontogenic
epithelium.[2] It is classi ed on the basis of its location as central or
intraosseous and peripheral or extraosseous.[3] The lesion occurs
most commonly in mandible although cases have been reported
in maxilla as well. The lesion involves maxilla in its anterior region
and mandible in its posterior region.[4] The age of af icted persons
ranges from 4 to 80 years with a female predilection. The lesion
grows slowly and becomes symptomatic only when it reaches
a considerable size. Initial clinical presentation can be a slight
expansion of the buccal or lingual cortical plate. Very large lesions
are present with external facial deformity.
Radiological features in smaller lesions may show unilocular
radiolucent area with well-defined borders. Larger lesions
may show multiloculation with scalloped margins and may
cause cortical expansion or tooth loosening or displacement.[5]
Depending on the amount of epithelium seen histologically,
The histological classification of odontogenic fibroma as
epithelium poor (simple type) and epithelium rich (complex
or WHO type).[2] The treatment includes surgical excision and
curettage. The recurrence rate is very low as reported in the
literature. We present a case of giant odontogenic broma
presenting as gross facial deformity with upward and outward
displacement of orbit and downward and medial displacement
of palate. Such a large tumor was a dual challenge in excising the
tumor mass and in reconstruction of the surgical defect.
CASE REPORT
A 20-year-old male patient reported to the Department of ENT
at SAIMS Medical College with the chief complaint of painless
swelling in the right maxillary region, which was slowly growing
for the past 3 years causing marked facial dis gurement with
palatal and orbital displacement [Figure 1]. There was gaping
between teeth of right upper alveolus. Externally, the swelling
was approximately 10 cm × 13 cm in size, rm, nontender, and
non uctuant in nature. It was superiorly encroaching on the right
orbit and displacing it in a superolateral direction. Posteroinferiorly,
it was going up to the angle of mandible. Anteroinferiorly, it
displaced the upper alveolus and palate inferiorly. Laterally, it
extended up to preauricular region. Medial extension of the tumor
pushed the lateral wall and septum, blocking the nasal cavities. The
nose was deviated to the opposite side, and the mouth opening
was normal but patient had slight dif culty in mastication and
speech. The patient also had diplopia because of superolateral
deviation of the orbit.
A computed tomography (CT)-scan revealed a well-circumscribed
ABSTRACT
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DOI:
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Baser, et al.: Giant odontogenic fibroma of maxilla
Annals of Maxillofacial Surgery | July - December 2014 | Volume 4 | Issue 2212
solid-cystic mass of size 11.6 T × 11.4 AP × 12.9 CC cm [Figure 2].
There was marked thinning of the sinus wall and superolateral
displacement of right eyeball with no obvious in ltration of
intra-/extra-conal fat. Posteriorly, it occupied infratemporal fossa
with obliteration of pterygomaxillary ssure, pterygopalatine
fossa, vidian canal, and inferior orbital ssure. It was inseparable
from pterygoid plates and reached up to the nasopharynx. It was
just touching the oor of middle cranial fossa with no skull base
erosion. Inferiorly, it involved the alveolar process with gross mal-
alignment and displacement of teeth. Medially, it involved the right
ethmoidal cells, obliterating the frontoethmoidal recess with gross
deviation of lateral nasal wall and septum. The bony and soft tissue
window of CT-scan showed an expansile, well-circumscribed,
multilocular, solid-cystic mass with centric, and eccentric foci of
mineralization with marked bony remodeling causing thinning
and expansion of the bony cortex [Figures 3 and 4]. These ndings
suggested probable diagnosis of either a giant cell tumor or
giant cell reparative granuloma. Fine-needle aspiration cytology
smear showed a cluster of spindle cells in a myxoid background
suggesting bromyxoid tumor.
The large tumor was excised completely using Weber-Fergusson
incision [Figures 5 and 6]. It was a multilocular swelling with
well-de ned bony margins [Figure 7]. The bony wall of orbital
oor, the lower margin of alveolus and oor of maxillary sinus were
intact. The lateral nasal wall defect on the right side was closed using
the available mucosa. Horizontal plate of expanded palate was
osteotomized parasagittally to elevate the palate. More osteotomies
were done all around the alveolus to reposition it vertically forming
part of medial maxilla. The alveolus thus formed was stabilized with
2 mm plates and 11, 12, and 13 teeth were extracted. Titanium mesh
was shaped, prepared and adjusted to such a size that it could be
xed using mini screws. The complete hollowed out defect was
then covered with 3 cm × 7 cm titanium mesh superiorly to the
nasal bone and zygomatic arch and inferiorly to the new formed
lower margin.
The mass was sent for histopathology. Grossly it was a solid
cystic mass measuring 11 cm × 11 cm × 13 cm [Figure 7]. The
cut surface showed multiple cysts of varying sizes, lled with
gelatinous material. The sections showed a tumor composed of
brous tissue with cystic spaces containing dentine like material
with scattered areas of calci cation, suggesting the diagnosis of
odontogenic broma [Figure 8].
Figure 4: Computed tomography-scan soft tissue window picture
Figure 2: Computed tomography-scan picture with three-dimensional
reconstruction
Figure 3: Computed tomography-scan bony window picture
Figure 1: Photograph of patient showing right maxillary swelling with
facial deformity
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Baser, et al.: Giant odontogenic fibroma of maxilla
Annals of Maxillofacial Surgery | July - December 2014 | Volume 4 | Issue 2 213
Once the tension from the removal of the tumor mass was
released, the orbit slowly moved to a medial position. Six months
later when the eye was in a more neutral position, the patient
was taken for another surgery. Titanium mesh was removed,
and zygoma implant (Biopore) was xed. Two years later when
the remodeling of palate took place, the patient was tted with
a partial denture. Three years follow-up of the patient showed
no sign of recurrence of disease. The patient has a cosmetically
acceptable face with normal mastication and binocular
vision [Figure 9].
DISCUSSION
Odontogenic broma of maxilla is a rare tumor. Wesley et al.
described odontogenic broma as a slow progressive, painless
cortical expansion of the jaw, which is seen as multilocular
radiolucent lesion with or without unerupted/displaced teeth in
radiography and dense collagen with broblasts and odontogenic
epithelium histopathologically.[1] Most of the studies in literature
observed that the lesions were smaller with higher prevalence
in females in the second to fourth decade of life and mandible
was involved more than maxilla.[6,7] Our case is a male patient
of 20 years with giant lesion involving right maxilla. Earlier
cases of odontogenic broma in the maxilla, although rare, have
been reported but such giant odontogenic broma has not been
reported as per our search in the available literature.
Radiographic studies demonstrate a soft tissue mass with some
areas of calci cation and do not involve underlying bone.[8] The
CT-scan in our case showed a solid-cystic mass with marked bony
Figure 5: Per-operative picture of the tumor
Figure 7: Excised specimen showing multiple cysts
Figure 8: Section showing tumor composed of fi brous tissue and cystic
spaces containing dentine like material (H and E, ×400)
Figure 6: Defect after excision of tumor showing intact infraorbital and
alveolar margins
Figure 9: Three year postoperative photograph
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Baser, et al.: Giant odontogenic fibroma of maxilla
Annals of Maxillofacial Surgery | July - December 2014 | Volume 4 | Issue 2214
thinning of sinus as well as adjacent bony walls. It was labeled as a
benign swelling with marked displacement of adjoining structures.
The challenge was complete excision as well as repair of the
defect. Weber-Fergusson approach was taken for complete
removal, and immediate reconstruction was done using titanium
mesh. The recurrence of this tumor is uncommon with no
evidence of malignant transformation.
Odontogenic broma is histologically characterized by broblastic
proliferation with scattered amount of calci ed material and a
variable amount of odontogenic epithelium.[9] Similar histological
features have been observed in other lesions like hyperplastic
dental follicles.[10] The histological features in our case were
consistent with ndings of odontogenic broma. The patient is
followed for 3 years and no signs of recurrence were seen.
REFERENCES
1. WesleyRK, WysockiGP, MintzSM.  e central odontogenic  broma.
Clinical and morphologic studies. Oral Surg Oral Med Oral Pathol
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2. BarnesL, EvesonJW, ReichartP, SidranskyD. World Health Organization
Classi cation of Tumors. Pathology and Genetics of Head and Neck
Tumors. Lyon: IARC Press; 2005. p.317.
Cite this article as: Baser B, Kinger A, Mitra GV, Roy MT. Giant odontogenic
broma of maxilla. Ann Maxillofac Surg 2014;4:211-4.
Source of Support: Nil, Con ict of Interest: None declared.
3. RaubenheimerEJ, No eCE. Central odontogenic  broma-like tumors,
hypodontia, and enamel dysplasia: Review of the literature and report of
a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:74-7.
4. AllenCM, HammondHL, StimsonPG. Central odontogenic  broma,
WHO type. Areport of three cases with an unusual associated giant cell
reaction. Oral Surg Oral Med Oral Pathol 1992;73:62-6.
5. DanielsJS. Central odontogenic  broma of mandible: A case report and
review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol
Endod 2004;98:295-300.
6. GarciaBG, JohannAC, da Silveira-Júnior JB, AguiarMC, MesquitaRA.
Retrospective analysis of peripheral odontogenic  broma(WHO-type)
in Brazilians. Minerva Stomatol 2007;56:115-9.
7. D aleyTD, WysockiGP. Peripheral odontogenic  broma. Oral Surg Oral
Med Oral Pathol 1994;78:329-36.
8. NevilleBW, DammDD, AllenCM, BouquotJE. Odontogenic cysts and
tumors. Oral and Maxillofacial Pathology. 3rd ed.. Noida: Elsevier; 2009.
p.678-740.
9. GardnerDG. Central odontogenic fibroma current concepts.
JOral Pathol Med 1996;25:556-61.
10. GardnerDG, RaddenB. Multiple calcifying hyperplastic dental follicles.
Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1995;79:603-6.
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... Odontogenic fibroma is a rare neoplasm of mature fibrous connective tissue, with variable amounts of inactive-looking odontogenic epithelium, with or without evidence of calcification (El-Naggar et al., 2017). The clinical and radiological characteristics of odontogenic fibroma resemble other odontogenic and/or non-odontogenic tumours, and the differential diagnosis is based on a biopsy (de Matos et al., 2011;Wu et al., 2013;Baser et al., 2014;Santoro et al., 2015;El-Naggar et al., 2017). ...
... Concerning treatment of COFs, the best choice involves enucleation, since the lesion is usually easily removed, showing little bone adhesion and no tendency to undergo malignant transformation (Zachariades, 1986;Melo et al., 2011;Takeoka et al., 2012;Baser et al., 2014;Schussel et al., 2014). The follow-up information, after a period of about 3 years, indicates that this type of tumour has a benign behaviour due to its clinical and radiographic aspects. ...
... The follow-up information, after a period of about 3 years, indicates that this type of tumour has a benign behaviour due to its clinical and radiographic aspects. In addition, it has been shown that WHO-type COF has no tendency for infiltrative growth or relapse (Doyle et al., 1985;Handlers et al., 1991;Calvo et al., 2002;Baser et al., 2014). ...
Article
Purpose: To integrate the available data published on central odontogenic fibroma (COF) into a comprehensive analysis of its clinical/radiologic/histological features. Methods: An electronic search was undertaken in September 2017. Eligibility criteria included publications reporting cases of COF having enough clinical, radiological and histological information to confirm the diagnosis. Demographic data, lesion site and size, treatment approach, and recurrence were analysed. The cases included in the analysis presented follow-up time. Concerning recurrence analysis, tumour location, cortical bone perforation, lesion locularity, radiopacities, lesions associated with a tooth, tooth displacement, histological type and treatment used were evaluated. Results: Eighty-three publications reporting 173 COFs were included. Lesions were slightly more prevalent in men than women (M: F - 1.13: 1); mean age was 31.6 years, with the highest prevalence in the second decade of life. Lesions were more prevalent in the posterior mandible. The difference in recurrence rate (when information about recurrence was provided) presented a statistically significant result for COF for location, cortical bone perforation and locularity of the lesion. Conclusions: Our paper highlights that patients with COF who present a lesion located in the maxilla with multilocular aspects and cortical bone perforation tend to show a higher recurrence rate.
... B, Panoramic radiograph acquired over 9¼ years before surgery. Note the emerging radiolucency in the left lower jaw (regions [35][36][37][38]. C, Panoramic radiograph acquired approximately 3¾ years before surgery. ...
... Diagnostic delay allowed the tumor to expand massively from the left mandibular body well into the ramus and to even affect the coronoid process. The occurrence of a COF of such great size is extremely rare and even more so in the literature [34,35]. The present follow-up radiography findings underscore the slow-growing, expanding, and non-infiltrative nature of COF. ...
Full-text available
Article
Introduction Central odontogenic fibroma (COF) is a rare, benign, slow-growing intraosseous odontogenic tumor, and accounts for 0.1% of all odontogenic tumors. It is often confused with other entities, such as keratocysts, ameloblastomas, and odontogenic myxomas. Complete enucleation followed by curettage is the treatment of choice for COF to ensure the lowest possible chance of recurrence. Case presentation We report the case of a young Caucasian woman with COF that went undiagnosed for several years despite repeated radiologic examinations. Finally, a massive tumor was surgically removed and the wound was curetted. The specimen was histologically confirmed to be a COF. The patient remains under regular follow-up, and thus far there have been no clinical or radiologic signs of recurrence. Discussion This rare case of COF, which was documented over a period of 20 years, has helped us to describe the features of this tumor. It also confirms that adequate surgical treatment can lead to impressive bone regeneration in healthy individuals, as evident from the radiologic findings acquired before, during, and after enucleation of the COF in our patient. Our findings also confirm the view that COF has a favorable prognosis regardless of its final size. Conclusion Early diagnosis is key to successful treatment of COF. The slow but steady increase in the size of a COF with no accompanying symptoms has not been reported previously. To our knowledge, this is the only documented case of a COF that has been under continuous radiologic observation for over 20 years.
... 2,3 However, there are scant reports of other benign odontogenic tumors reaching very large sizes. [4][5][6][7][8] Dentinogenic ghost cell tumor (DGCT), a rare, benign OT, with aggressive behavior, causes local destruction and occurs more commonly in the posterior regions of the maxilla and the mandible. 9,10 Microscopically, DGCT is characterized by deposition of variable amounts of dentinoid, associated with odontogenic epithelial cells organized in solid ameloblastomatous proliferations and presence of clusters of ghost cells. ...
Article
Dentinogenic ghost cell tumor (DGCT) is a rare, aggressive and benign odontogenic tumor that causes bone destruction and cortical expansion. We report a case of DGCT in a 38-years-old male, presenting an enormous extra-oral protrusion, that radiographically was predominantly radiolucent with radiopaque areas. Microscopically, it was observed a solid ameloblastomatous proliferation with pseudoglandular structures associated with clusters of ghost cells. Abundant dentinoid material adjacent to the epithelial sheets containing entrapped epithelial tumor cells were also evident. Immunohistochemistry revealed positivity for pan-Cytokeratin (CK), CK-14, CK-7 and CK-19, CD138, and β-catenin. Ki-67 proliferative index was very low, less than 1%. The clinical, histopathological and immunohistochemical features led to the diagnosis of DGCT. The patient was submitted to partial mandibulectomy and follows without recurrences. To the best of our knowledge, this is the largest DGCT described in the English literature.
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Central odontogenic fibroma is an extremely rare benign tumor, accounting for less than 0.1% of all odontogenic tumors. The most recent literature review revealed that only 69 cases have so far been reported in the English literature, and only 1 of these cases showed radiologic appearance of a dentigerous cyst and this involved impacted maxillary incisors. A case of central odontogenic fibroma occurring in the mandible of a 30-year old female with radiological appearance of a dentigerous cyst involving an impacted lower first molar, presented here, appears to be the first reported case of central odontogenic fibroma of mandible with radiological appearance of dentigerous cyst in the English-language literature.
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This article describes two additional examples of this unusual condition; two others were reported previously. The microscopic features are those of the hyperplastic dental follicles that occur in regional odontodysplasia. However, the teeth are not defective, as in regional odontodysplasia. This rare condition is sufficiently distinctive to be considered a pathologic entity.
The clinical, pathologic, and ultrastructural features and pertinent case history of an 11-year-old boy with a rare benign odontogenic neoplasm, the central odontogenic fibroma, are presented. A review of the literature reveals seven other lesions with clinical and pathologic findings similar to those of this case. This study supports the concept that the central odontogenic fibroma is a distinct odontogenic neoplasm of bone which occurs most commonly in the mandible as a multilocular, radiolucent, and slowly growing expansile lesion with no tendency to recur after surgical enucleation. The ultrastructural findings indicate that the central odontogenic fibroma and the odontogenic myxoma share many common morphologic features and have an apparently similar histogenesis.
Three cases of central odontogenic fibroma, WHO type, that exhibited a prominent giant cell granuloma-like histopathologic component are described in this report. All three lesions occurred in women, and all were located in the mandibular premolar-molar region. One lesion recurred after conservative excision, and the recurrence displayed the two histologically distinct tissues of the original lesion. The authors think that this pathologic process does not represent a "collision lesion" but, instead, is a unique presentation of a central odontogenic fibroma, WHO type.
The peripheral odontogenic fibroma is characterized by a fibrous or fibromyxomatous proliferation that contains varying amounts of odontogenic or presumed odontogenic epithelium. It has been considered a rare gingival neoplasm that, because of a lack of follow-up information in most reported cases, has had an unknown biologic behavior. Clinical data from this study indicate that the lesion is more common than previously reported and that it has a significant recurrence rate. Light and electron microscopic data reveal a relatively broad spectrum of epithelial and mesenchymal components, including a rare granular cell type. Because the presence of both epithelial and mesenchymal elements are required for its diagnosis, the lesion is logically classified as a mixed epithelial/mesenchymal odontogenic tumor, rather than a purely mesenchymal tumor.
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The author reviews current knowledge concerning the central odontogenic fibroma, which at present in incompletely understood, and reaches the following conclusions. 1) The separation of this lesion into simple and WHO types remains valid because they exhibit different histologic features. However, more care should be taken in rendering the diagnosis of the WHO type than in the past; unlike the simple type, it is a fibroblastic lesion. 2) Complex central odontogenic fibroma is a more appropriate term than the WHO type because the WHO does not use the latter term in its 1992 manual. 3) The microscopic distinction of simple odontogenic fibroma from desmoplastic fibroma remains difficult in some cases. 4) The granular cell odontogenic tumor, which has sometimes been referred to as a type of odontogenic fibroma, is a separate entity, although some simple odontogenic fibromas exhibit scattered granular cells. 5) The separation of lesions that have been reported recently as odontogenic fibromas with giant cell reactions from central giant cell granulomas that exhibit foci of odontogenic epithelium requires further study.
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Multiple calcifying hyperplastic dental follicles is a rare condition characterized by multiple unerupted teeth with abundant calcifications and rests of odontogenic epithelium in their enlarged dental follicles. This article describes an additional example of this condition.
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A patient with multiple odontogenic fibroma-like tumors in the mandible and enamel dysplasia is presented, bringing the total number of cases reported in the literature to 3. In addition to these manifestations, this case had hypodontia. The absence of associated teeth, the size of the lesions, the lingual expansion, and the green-yellow polarization of collagen with Picrosirius stains supported the neoplastic nature of the central odontogenic fibroma-like tumors in the case presented. Laminated psammomatous deposits distinguished the tumors from the World Health Organization-type central odontogenic fibroma.
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Central odontogenic fibroma is an extremely rare benign tumor, accounting for less than 0.1% of all odontogenic tumors. The most recent literature review revealed that only 69 cases have so far been reported in the English literature, and only 1 of these cases showed radiologic appearance of a dentigerous cyst and this involved impacted maxillary incisors. A case of central odontogenic fibroma occurring in the mandible of a 30-year old female with radiological appearance of a dentigerous cyst involving an impacted lower first molar, presented here, appears to be the first reported case of central odontogenic fibroma of mandible with radiological appearance of dentigerous cyst in the English-language literature.