EN BLOC RESECTION OF OSSIFYING FIBROMA OF THE JAW

Ganendra Anugraha*, Okky Prasetio**

Resident of Oral and Maxillofacial Surgery Faculty of Dentistry, Airlangga University Surabaya
Indonesia.
Staff of Oral and Maxillofacial Surgery, Dr Soewandhie Hospital, Surabaya - Indonesia

Abstract

Background:

Ossifying fibroma is a benign neoplasm usually presented as a painless, slow-growing,

expansile lesion which is believed to be confined to the jaws and characterized by the

replacement of normal bone by fibrous tissue and varying amounts of newly formed bone or

cementum-like material, or both.

Aim:

This paper discussed the treatment of ossifying fibroma and the feasibility of immediate

reconstruction to restore aesthetics and function.

Case:

A 31 years old woman with hard, painless swelling of the right body of the mandible. The

patient had been aware of the swelling for approximately one year but had not sought

treatment.

Methods:

The lesion was exised by en bloc resection of the mandible that include buccal plate from 43

region until right angulus mandible for the complete resection with free surgical margins and

plating.

Conclusion:

The diagnosis of ossifying fibroma of the jaw can be established quite consistently based on

clinical, radiographic, and microscopic features. Elective surgery was planned under general

anaesthesia for en bloc tumour resection and reconstruction due to its size, cosmetic

deformity and clinical presentation.

Key words: Ossifying fibroma, en bloc resection, mandible reconstruction

Introduction

Ossifying fibroma (OF) is classified as, and behaves like, a benign bone neoplasm. It

is often considered to be a type of fibro-osseous lesion. It can affect both the mandible and

the maxilla, particularly the mandible. This bone tumour consists of highly cellular, fibrous

tissue that contains varying amounts of calcified tissue resembling bone, cementum or both.1

The origin of Ossifying Fibroma is thought to be the periodontal membrane. 7 Some

Ossifying fibromas do, in fact, contain prevalent cementum-like calcifications and others show

only bony material, but a mixture of the two types of calcification is commonly seen in a single

lesion.4,5 It can occur at any age, however, many authors confirmed that Ossifying fibroma of

the jaw tended to occur middle-aged patients. 5,7 Ossifying fibroma of the jaw bone shows a

predilection for females.2,8,9 Ossifying fibroma predominantly affects the craniofacial bone and

rarely involves the long bones.

Ossifying fibroma develops from the multipotential mesenchymal cells of periodontal

origin which are able to form both bone and cementum. 10,11 Although the precise pathogenesis

is still unknown, Wenig et al 12 has suggested that trauma induced stimulation may play a role.

In general, ossifying fibroma is an asymptomatic lesion until growth causes swelling and

moderate deformation.3,4 Displacement of the teeth6,15 can be an early clinical manifestation.14

The teeth associated with the lesion preserve their vitality and may present root resorption. 12,15
14-17
The lesion is relatively slowgrowing, as a result of which the overlying cortical bone layer

and mucosa remain intact,13,15 and thus the tumor may be present for a number of years

before a diagnosis is made.15

It is described in the World Health Oraganisation histological typing of odontogenic

tumours as an actively growing lesion consisting of a cell rich fibrous stroma, containing

bands of cellular osteoid without osteoblastic lining, together with trabeculae of more typical

woven bone. Small foci of giant cells may also be present. The lesion is non encapsulated

but well demarcated from surrounding bone.18

Case report

A 31 years old female was referred to our institute with a chief complaint of swelling

since 2 years ago. This swelling evolving growth slowly over the past 4 months until now.
There was no significant medical and dental history. There is no mobility teeth in the region of

the mass. On clinical examination it was 5 x 4 x 2 cm in size approximately extending in

relation to 45, 46, 47 with expansion more on the buccal aspect with bulging of the region in

distobuccal 46 of the mandible. The swelling was non tender, bony hard in consistency with

intact overlying mucosa. There was no mobility teeth in 45, 46, 47. There was no paresthesia

or hypoesthesia. The associated teeth were vital. Laboratory findings were wit in the normal

limits.

Figure 1. A large swelling over the patients right cheek involving the lower border of

the mandible.
 Figure 2. Intra orally, the tumor noted on the buccal side of the mandible obliterating the

mucobuccal fold;

Figure 3. On panoramic a round shape radiolucency noted in the right mandible showing

sharp, sclerotic margin, multiloculation and spots of radiopacity in the centre of the lesion

Panoramic revealed mixed image with well defined borders extending from the right

lower first molar to right retromolar pad with intact lower border of the mandible. Teeth

associated with lesion were displaced. There was an impacted teeth of 48 involve in corpus

region of right mandible.

The lesion was well defined, non homogenous with radiopaque foci. Histopathology revealed

cellular connective tissue stroma with numerous bony trabeculae mature. Stroma comprised

of numerous spindle shaped cells with bony trabeculae showing osteoblastic rimming among

spindle fibroblast proliferation with smooth cromatin The result of the examination suggested

a diagnosis of ossifying fibroma.

The tumor was removed together with the adjacent bone with segmental resection of

the left mandible. Upon removal of the bone segment it was found that the tumor was a

round, encapsulated mass with multiple foci of whitish component in the centre of the lesion

reflecting the tissue with calcified material.

 Figure 4. Intra operative view

Enbloc resection was performed under general anesthesia in July 2016 through an

extra oral and intraoral approach. The enbloc resection was performed in buccal segment of

right mandible. The teeth 43, 45, 46 needed to be removed. Enbloc resection and Curettage

of the tumorous base was performed. It also odontectomy of 48.

Figure 5. After enbloc resection and curettage of the tumour

 The cavity resulting from the tumor removal was cleaned followed by the closing of

the vestibular incision. The defect in the mandible was reconstructed by placement of bridging

plate using reconstruction plate. The histopathologic result shows that the tumor consists of

fibroblast proliferation with radiopaque foci which confirm the diagnosis of ossifying fibroma.

Figure 6. placement a reconstruction plate in the defect of the mandible

 Figure 7. The resected mandible showing a well encapsulated mass involving the buccal aspect of the

body mandible and the involving teeth

Postoperative care was incidentfree and the patient was able to leave the hospital the

day following the operation. He was checked 8 days later and showed good postoperative

healing. Another check-up 30 days later showing satisfactory progression and complete

recovery. She was given a schedule of regular check-ups every 3 months during the first year,

then twice a year from July 2016 onward. An anatomopathological examination of the excised

tissue revealed numerous islets of varying sizes, converging toward each other, even in

texture within the bony tissue, demonstrating their identification as lesions of ossifying fibroma

with no indication of malignancy, thus confirming the diagnosis.

Discussion

Ossifying fibroma is a benign, uncommon, monostotic well defined unilocular or

multilocular fibrous-osseous tumor, arising from the periodontal ligament composed of fibrous

connective tissue with variable amounts of metaplastic bone and mineralization. The lesion is

generally encapsulated a fact that serves to distinguish it from fibrous dysplasia, which may

exhibit similar clinicopathological features.14-16 Although the lesion is preferentially located in

the jaws, it can also be found elsewhere including the frontal, ethmoid, sphenoid and temporal

bones or orbit, as well as in the anterior cranial fossa. 17,18 Some authors have pointed to
antecedents of trauma in the area of the lesion, the performance of tooth extractions, and the

prior existence of periodontitis, as possible triggering factors. 19

A neoplastic etiology of ossifying fibroma is supported by examples of lesions that

achieve a large size, exhibit aggressive behavior, and produce significant osseous

destruction.2 Additionally, recurrences, though rare, have been described in some studies of

ossifying fibroma. Chromosomal translocations have been identified in a few cases of

ossifying fibroma, however, the molecular mechanisms that underlie the development of this

tumor remain unknown.2

Ossifying fibroma most frequently occurs in female patients (age range 1059 years,

mean 32 years) with an incidence peak in the third and fourth decades. 16 Similarly two of our

cases were in third and fourth decade and were female. The mandible, including the ramus,

more commonly in the molar and premolar zone, is the region most commonly affected as

seen in our cases.17,18 It appears as a hard, localized and slow-growing mass that displaces

the teeth, though the latter remain vital and the overlying mucosa is characteristically intact. 19

The size of the lesion can range from 0.2- 15 cm; in our case the first two cases measured 4 -

5 cm in diameter. Radiologically, the lesion appears well circumscribed, and is initially seen as

an osteolytic image followed by gradual trans formation into a mixed lesion in exceptional

cases becoming radiopaque.15,19,22 Some authors have described two basic radiological

patterns: a unilocular radiotransparency with or without radiopaque foci, and a multilocular

radiotransparency.12,14

With a condition evolving slowly, painlessly, and with no related neurological

symptoms, the diagnosis should look toward a benign neoplastic process. Ossifying fibroma,

by its aggressive nature and extragnathic development, progresses by targeting the maxillary

areas and ends up invading the paranasal sinus, the orbital cavities, the frontal bones, and

the structures of the base of the skull 6.

The characteristic macroscopic features of this tumor is replacement of normal bone

by a benign connective-tissue matrix with varying amounts of mineralized substances,

however, there are some variations in microscopic features of this tumor. The microscopic

findings mirror the radiographic findings. The more radiolucent lesions are composed of

cellular fibrous connective tissue, frequently in a whorled pattern. 22 Collagen fibers are often
arranged haphazardly, although a whorled, uniform pattern may be evident. Calcified deposits

are noted throughout the fibrous stroma. The nature of the hard tissue is generally quite

variable within a given tumor as well as between lesions. Irregular trabeculae of woven bone

or lamellar bone are most consistently noted in these tumors. Additional patterns of calcified

material include small, ovoid to globular, basophilic depostis and anastomosing trabeculae of

cementum-like material.2 These variations in hard tissue configuration make no difference to

the clinical behaviour of the tumour. However, recognition of these structures is important in

establishing its diagnosis.20 Osteoblast may or may not be evident at the periphery of the

bone deposits. A thin outer zone of fibrous connective tissue is usually present, separating the

fibro-osseous tissue from the surrounding normal bone. 22 The foci of calcified material appear

as bony trabeculae with evidence of osteoblastic rimming at the periphery of the trabeculae

therefore the term ossifying fibroma is applied as the histopathologic diagnosis.

Treatment of ossifying fibroma generally has been by conservative enucleation or

curettage or radical surgery15,16,21 depending on the size and location of the individual lesion. 21

Treatment is mostly surgical and consists of enucleoresection of the smaller ossifying fibroma

and the complete removal of the growth combined with bone reconstruction in cases of larger

ossifying fibroma. They are characterized by easy shell out from the surrounding bone. 16

Conservative surgery is therefore recommended even if the tumour is large with bowing and

erosion of the inferior border of the mandible and radical treatment of the tumour such as an

en bloc resection should only be considered if there are recurrences due to its aggressive

nature. En bloc resection followed by surgical ostectomy performed in this patient was the

least aggressive surgical treatment in this case series since there was no history of rapid

tumor growth, clinically and radiographically it was relatively not aggressive, and there still

remained sufficient amount of bone in the inferior border of the mandible after excision of the

tumor.

Conclusion

As the conclusion of this paper, although it is relatively not difficult to establish the

diagnosis of ossifying fibromas from clinical, radiographic, and microscopic features, these

tumors may exhibit variations in their neoplastic behaviors. It affects primarily membranous
bones such as those of the maxillofacial skeleton. The fibroma can be found in children or

adults 2030 years of age. It occurs mostly on the mandible. The difficulty of identifying this

type of tumor resides in the fact that it requires a precise diagnosis not readily provided by

clinical or radiographic examinations.

It is, therefore, important to take into account the individual tumor behavior when one is

planning a proper surgical treatment in order to eliminate the tumor completely and avoid

tumor recurrence and at the same time improve the patients cosmetic and functional

problems.

Reference
1. Ayub T, et al. En bloc resection of huge cemento-ossifying fibroma of mandible : Avoiding

Lower Lip Split Incision. Journal of the College of Physicians and Surgeons Pakistan 2011, Vol.

21 (5): 306-308
2. Waldron CA, Giansanti JS. Benign fibro-osseous lesions of the jaw: a clinical-radiologic review

of sixty-five cases. Oral Surg Oral Med Oral Pathol 1973; 35: 340350.
3. Eversole LR, Merrell PW, Strub D. Radiographic characteristics of central ossifying fibroma.

Oral Surg Oral Med Oral Pathol 1985; 59: 522527.

4. Pindborg JJ, Kramer IRH. Histological typing of odontogenic tumors, jaw cysts and allied

lesions. In: International histological classification of tumors. Geneva: WHO, 1971, pp 31 34.
5. Kramer IRH, Pindborg JJ, Shear M. Neoplasm and other lesions related to bone. Histologic

typing of odontogenic tumors, World Health Organization. Berlin, Springer-Verlag 1992: 2831.
6. Reichart PA, Philipsen HP, Sciubba JJ. The new classification of Head and Neck Tumours (WHO)

any changes? Oral Oncol 2006; 42: 757758.

7. Krausen AS, Pullon PA, Gulmen S, Schenk NL, Ogura JH. Cementomasaggressive or innocuous

neoplasm? Arch Otolaryngol 1977; 103: 349354.

8. Hammer JE, Linghtbody PM, Ketcham AS, Swertlow H. Cemento-ossifying fibroma of the

maxilla. Oral Surg Oral Med Oral Pathol 1968; 26: 576587.
9. Longdon JD, Rapidis AD, Patel MF. Ossifying fibro-osseous lesions of the jaw. Br J Oral Surg

1976; 14: 111.

10. Bartolini F, Caradonna L, Bianchi B, Sesenna E, Multiple ossifying fibroma of the jaws: a case report. J

Oral Maxifac Surg 2001; 60: 225-29. doi:10.1053/joms.2002.29832 PMid:11815929

11. Wenig B L, Sciubba JJ, Goldstein MN, Cohen A, Abramson AL. A destructive maxillary cemento ossifying

fibroma following maxillofacial trauma. Laryngoscope 1994, 810-15.

12. Zachariades N, Vairaktaris E, Papanicolau S, Triantafyllou D, Papavassiliou D, Mezitis M. Ossifying fibroma

of the jaws. Review of the literature and report of 16 cases. Int J Oral Surg 1984;13:1-6.

doi:10.1016/S0300-9785(84)80049-6
13. Jayachandran S., Sachdeva S. Cemento-ossifying fibroma of mandible:Report of two cases. Journal of

Indian academy of Oral Medicine & radiology, 2010;22(1):53-56

14. Martn-Granizo R, Snchez-Cuellar A, Falahat F. Cemento ossifying fibroma of the upper gingivae.

Otolaryngol Head Neck Surg 2000;122:775. doi:10.1016/S0194-5998(00)70216-6

15. Kramer IRH, Pindborg JJ, Shear M. World Health Organization histological classification of tumours.

Histological typing of odontogenic tumours, 2 nd edn. Berlin:Springer-Verlag, 1992.

16. Booth PW, Schendel SA, Hausamen JE: Maxillofacial surgery. 2nd ed. St. Louis, Missouri: Churchill

Livingstone; 2007. p. 5069.

17. Regezi JA, Sciubba JJ. Oral pathology-clinical pathologic correlations. 3rd ed. Philadelphia: WB Saunders

Co; 1999. p. 35760.

18. Brannon RB, Fowler CB. Benign fibro-osseous lesions: a review of current concepts. Adv Anat Pathol

2001; 8: 12643.
19. Kos M, Luczak K, Godzinski J, et al. Treatment of monostotic fibrous dysplasia with pamidronate. J

Craniomaxillofac Surg 2004; 32: 1015.

20. Neville BW, Damm DD, Allen CM, Bouquot JE. Oral & maxillofacial pathology. Philadelphia: WB Saunders

Co; 1995. p. 46970.

21. Ong AHM, Siar CH, Cemento-ossifying fibroma with mandibular fracture. Case report in a young patient.

Austrl Dent J 1998; 43(4): 22933.

22. Sapp JP, Eversole LR, Wysocki GP: Contemporary oral and maxillofacial pathology. 2nd ed. St Louis,
Missouri: Mosby; 2004. p. 1167.