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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 3  |  Issue : 1  |  Page : 53-57

Ossifying fibroma of the maxilla: An uncommon tumor presenting diagnostic and management dilemma for the clinician: A rare case report


Department of Oral and Maxillofacial Surgery, Tatyasaheb Kore Dental College and Research Centre, Mahatma Gandhi Hospital Campus, New Pargao, Kolhapur, Maharastra, India

Date of Web Publication6-May-2015

Correspondence Address:
Dr. Shahanavaj I Khaji
Department of Oral and Maxillofacial Surgery, Tatyasaheb Kore Dental College and Research Centre, Mahatma Gandhi Hospital Campus, New Pargaon - 416 137, Kolhapur, Maharastra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2277-4696.156534

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  Abstract 

Ossifying fibromas form a part of the spectrum of fibro-osseous lesions of the jaws. They are rare, benign, nonodontogenic tumors that are commonly seen in the head, and neck region. Ossifying fibroma of the jaw is a benign fibro-osseous lesion that is a part of larger family of fibro-osseous lesions that includes - juvenile aggressive ossifying fibroma, psammomatous ossifying fibroma, extragnathic ossifying fibroma of the skull.
Patients generally present with a history of painless expansion of a tooth bearing portion of the mandible, whereas the lesions of the maxilla are less common. Benign fibro-osseous of the maxilla constitutes a varied group of lesions with a common histologic characteristic: The substitution of normal bone by tissue composed of collagen and fibroblasts with variable amounts of mineralized substance that may be bone, cementum or both.
In the present case, we hereby report a 35-year-old male patient who presented with painless swelling over middle one third of face - left side since 8 months. Patient had no significant medical history in the previous past (history of trauma 6 years earlier). Panoramic radiography revealed rounded mixed type of image over left side of posterior maxilla in relation to 23, 24, 25, 26 region, respectively. An incisional biopsy was carried out which yielded a definitive diagnosis of ossifying fibroma of the maxilla. Management of the lesion was carried out by taking into account the benign nature, growth pattern, and behavior of the lesion clinically and radio graphically with regular and periodic follow-up postoperatively.

Keywords: Benign fibro-osseous lesion, maxilla, ossifying fibroma


How to cite this article:
Khaji SI, Shah S, Baheti MR. Ossifying fibroma of the maxilla: An uncommon tumor presenting diagnostic and management dilemma for the clinician: A rare case report. J Dent Allied Sci 2014;3:53-7

How to cite this URL:
Khaji SI, Shah S, Baheti MR. Ossifying fibroma of the maxilla: An uncommon tumor presenting diagnostic and management dilemma for the clinician: A rare case report. J Dent Allied Sci [serial online] 2014 [cited 2019 Aug 19];3:53-7. Available from: http://www.jdas.in/text.asp?2014/3/1/53/156534


  Introduction Top


Historically, ossifying fibroma has been referred to as fibro osteomas, osteofibroma benign fibro-osseous lesion. In 1872, Menzel [1] first described the known as ossifying fibroma, but it was Montgomery [2] whom in 1927 coined the term ossifying fibroma.

It is a rare, benign primary bone tumor that occurs most commonly in the lower jaw. Fibro-osseous lesions constitute a rare type of pathology classified as benign tumors of nonodontogenic lineage that affect the craniofacial area. [3] Ossifying fibroma is characterized by the replacement of normal bone by fibrous tissue and varying amount of newly formed bone or cementum like material or both. [4]

It is usually presented as a painless, slow growing, expansile intra bony lesion, which is believed to be confined to the jaws and craniofacial complex. Lesions with fibrous and osseous components include:

  • Fibrous dysplasia.
  • Ossifying fibroma.
  • Cemento-ossifying fibroma. [5]


Fibro-osseous lesion other than fibrous dysplasia seems to arise from the periodontal membrane. [6] Ossifying fibroma is commonly seen between third and fourth decade of life. More frequently in women than in men (4:1). [7] Clinically these tumors manifest as slow growing intrabony mass that is normally well delimited and asymptomatic though over time the lesion may become large enough to cause facial deformation. [8]

According to the classification proposed by Waldrom in 1993 fibro-osseous lesions are divided into - fibrous dysplasia, ossifying fibroma, Desmoplastic fibroma. [9]

One remarkable finding is the large size of the maxillary tumors at the time of diagnosis, probably attributable to the large amount of available space in the maxillary sinus into which they could expand similar to our case study. Though the growth was sufficiently large, patient did not present with significant clinical symptoms as it was extending over the sinus region. [10]


  Case Report Top


A 35-year-old male patient reported to the author's clinic with the chief complaint of swelling over the left side of cheek region since 8 months. To begin with it was nonprogressive and asymptomatic. General physical examination did not reveal any abnormalities. Patient reported having suffered trauma in that same area years ago.

Extraorally lesion presented as fullness with ill-defined borders over middle 1/3 rd of face on the left side. Overlying skin was normal in appearance [Figure 1].
Figure 1: Patient on initial presentation showing mild facial swelling caused by lesion progression over left side

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Intra oral examination revealed diffuse palatal expansion of the jaw over the maxillary posterior region on the left side in relation to 23, 24, 25, 26 region, respectively, extending anteroposteriorly from distal aspect of upper left canine till the tuberosity region; measuring 3.5 cm × 2.5 cm in size approximately [Figure 2].
Figure 2: Preoperative intra oral view showing significant expansion of the palatal cortex in relation to 24, 25, 26 region

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Overlying swelling was nontender; hard in consistency associated with grade I mobility in relation to 24, 25, and 26. He had good oral hygiene status with no clinically detectable dental caries and periodontal problems. Vitality of 24, 25, and 26 was unresponsive and fine needle aspiration cytology was negative. Based on clinical presentation a provisional impression of a benign neoplasm was considered.

Panoramic view revealed well circumscribed, mixed radio opaque lesion obliterating the entire maxillary sinus associated with 23, 24, 25, 26 region on the left side. Similar features evident on true maxillary occlusal radiograph [Figure 3] and [Figure 4].
Figures 3: Preoperative OPG showing well circumscribed radio opaque lesion over left side of posterior maxillary region

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Figures 4: Preoperative true maxillary occlusal radiograph showing well circumscribed mixed radio opaque lesion (osteolytic lesion) relation to 23, 24, 25, 26 regions

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For careful evaluation of neighboring structures axial and coronal sections of computerized tomographic scans were obtained. The borders were ill defined, with rounded and elongated osteolytic lesion showing thinning and bulging of vestibular areas. Core region showed rounded radio opaque images in between [Figure 5]a and b.
Figure 5: (a) Axial section of computerized tomographic scan showing an osteolytic zone containing central radio opaque images (b) Coronal section of computerized tomographic scan showing an osteolytic zone on left side

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Incisional biopsy was carried out and definitive diagnosis of ossifying fibroma was reached. Complete surgical removal of the lesion (enucleation) was carried out under local anesthesia with conscious sedation. After raising a mucoperiosteal flap in toto removal of the lesion, extraction of 24, 25, 26, gross debridement was done and the bone bed was subjected to curettage followed by wound closure [Figure 6].
Figure 6: Intraoperative view of the lesion

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The excised specimen was stored in 10% formalin and subjected to histopathological evaluation. The postoperative course was favorable and 2 years later the patient reported no discomfort in that zone with satisfactory wound healing [Figure 7].
Figure 7: Two years postoperative clinical view showing satisfactory wound healing devoid of recurrence

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Postoperative panoramic radiograph confirmed good bone regeneration [Figure 8], which discarded possible relapses. Patient was periodically followed-up thereafter on regular visits.
Figure 8: Postoperative panoramic radiograph (2 years after surgery)

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  Discussion Top


Although there are persistent differences in terms of classification and diagnosis of fibro-osseous lesions in craniofacial area, there is consensus on the common characteristics of these lesions. All of them show the replacement of normal bone tissue with fibroblasts and collagen fiber tissue, with varying quantities of mineralized substances. [9]

Today there is a general agreement that ossifying fibroma and cemento-ossifying fibroma are a separate entity from fibrous dysplasia and an effort to simplify the classification, reflecting the view of Waldron that both of these lesions fall within the spectrum of same disease entity (this is supported by the fact that the periodontal ligament is able to elaborate both bone and cementum, acting on the alveolar periosteum as well as attachment of the tooth. [11]

In 1971, the WHO classified four types of cementum containing lesions:

  • Fibrous dysplasia.
  • Ossifying fibroma.
  • Cemento-ossifying fibroma.
  • Cementifying fibroma. [12]


According to the second WHO classification, benign fibro-osseous lesions in the oral and maxillofacial region were divided into two categories:

  • Osteogenic neoplasm's.
  • Nonneoplastic bone lesions. [13]


Various other classifications were also described for better understanding of various fibro-osseous lesions occurring in the head and neck region [Table 1] and [Table 2].
Table 1: Classification of fibro osseous lesions of the maxillofacial region

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Table 2: A working classification of the three clinicopathologic variants of ossifying fibroma

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The WHO classifies ossifying fibroma as a fibro-osseous neoplasm included among the nonodontogenic tumors derived from the mesenchymal blast cells of periodontal ligament, with a potential to form fibrous tissue, cement and bone, or a combination of such elements. [14],[15]

Pathogenesis

Although the underlying cause is not known, there have been reports of past trauma in the areas of the lesion. The same was evidenced in our case report. This point to trauma as a possible triggering factor in some presentations of the lesion, postulating the latter as representing a connective tissue reaction rather than a genuine neoplasm. [16],[17]

Suggested hypothesis include:

  • According to KEMPSOH [18] - bone deposition and resorption occurring on the same spicules of the bone. It was postulated that ossifying fibroma resulted from excessive resorption of bone with fibrous repair of the defect.
  • Trauma induced stimulation of progenitor cells has been suggested by WEING.
  • HAMMER suggested ossifying fibroma - considered as tumor of periodontal ligament origin (mesenchymal blast cells). [19]


A comprehensive review of the lesion was published by EVERSOLE in their report of 64 cases. Ossifying fibromas occur over a wide age range. The majority of cases are encountered in the third and fourth decades of life (same evidenced in the present case report, as the age was 35 years). Clinically, the lesion presents as painless, slow growing intrabony mass of the jaw where displacement of teeth may be the only early clinical feature. [7]

Differential diagnosis

The set of entities in differential diagnosis appearing as benign neoplasm include:

  • Fibrous dysplasia.
  • Gorlin cyst (calcifying epithelial odontogenic cyst).
  • Odontogenic cysts.
  • Adenomatoid odontogenic tumor.
  • Calcifying epithelial odontogenic tumor.
  • Myxoma.
  • Osteoblastoma.
  • Osteosarcoma.
  • Cementoblastoma.
  • Chondrosarcoma.
  • Cemento osseous dysplasia.
  • Paget's disease.
  • Central giant cell granuloma. [20]


Due to good delimitation of the tumor, surgical removal and curettage is the treatment of choice. In case of very large lesions with important tissue ablation, the challenge is to replace the affected tissue. [21] The circumscribed nature of the lesion permits the local enucleation or curettage of smaller lesions. [7]

The treatment of ossifying fibroma generally has been grouped into:

  1. Conservative enucleation.
  2. Curettage.
  3. Radical surgery.


The recommended treatment of choice of the ossifying fibroma is excision. The entire tumor should be removed including a rim of normal tissues. Management should be individualized and case specific, depending on the size, location, benign nature and growth behavior of the lesion accordingly. [22]

In the present case, we selected conservative treatment that is, enucleation of the lesion with thorough debridement, curettage of the bone bed was done to promote good healing of the defect. Patient was followed-up regularly and after 2 years postoperatively patient had sound bone regeneration devoid of recurrence at the operated site.

According to MacDonald-Jankowski [23] has indicated enucleation or curettage as the first treatment option affords a recurrence rate of 0-28%; if relapse is identified in the course of follow-up, conservative resection is obligate. Recurrence rates of aggressive forms of ossifying fibromas are about 30-38%.


  Conclusion Top


Despite the many years of dedicated study by numerous investigators, the concepts and parameters of fibro-osseous diseases are still in flux. Ossifying fibromas are comparatively rare benign tumors of maxillofacial region. Being capsulated it can be easily differentiated from other dysplastic conditions of the bone.

Treatment of ossifying fibroma should always be conservative excision of the well circumscribed without resection of the adjacent structures unless involved by the lesion; this should be accompanied with extraction of involved teeth and primary closure of wound respectively with timely follow-up.

From a clinical stand point, the fibro-osseous lesions may vary from the extensive and cosmetically or functionally disturbing lesions detected only during a routine radiographic examination. Any discussion of such lesions must begin with the observation that the term fibro-osseous is largely descriptive, nosologically limited, and diagnostically nonspecific.

 
  References Top

1.
Menzel A. A failed case of osteofibroma of the corpus mandible. Arch Klin Chir 1872;13:212.  Back to cited text no. 1
    
2.
Montgomery AH. Ossifying fibromas of the jaws. Arch Surg 1972;15:30.  Back to cited text no. 2
    
3.
Suarez-S A, Baquero-Ruiz de la Hermosa MC, Minguez-M I, Floría-García LM, Barea-Gámiz J, Delhom-Valero J, Risueño-Mata P. Management of fibro-osseous lesions of the craniofacial area. Presentation of 19 cases and review of the literature. Med Oral Patol Oral Cir Bucal 2013;18:e479-85.  Back to cited text no. 3
    
4.
Booth PW, Schendel SA, Hausamen JE. Maxillofacial Surgery. 2 nd ed. St. Louis, Missouri: Churchill Livingstone; 2007. p. 506-9.  Back to cited text no. 4
    
5.
Regeizzi JA, Scuibba JJ. Oral Pathology - Clinical Pathologic Correlations. 3 rd ed. Philadelphia, PA, Saunders; 1999. p. 57-360.  Back to cited text no. 5
    
6.
Demetrio T, Vasilios T, Ionasis T. Cemento ossifying fibroma of maxilla: A case report. Acta Stomatol Croat 2005;39:319-21.  Back to cited text no. 6
    
7.
Eversole LR, Leider AS, Nelson K. Ossifying fibroma: A clinicopathologic study of sixty-four cases. Oral Surg Oral Med Oral Pathol 1985;60:505-11.  Back to cited text no. 7
[PUBMED]    
8.
Perez GS, Berini AL, Gay EC. Ossifying fibroma of the maxilla - presentation of a case and review of literature. Med Oral 2004;9:333-9.  Back to cited text no. 8
    
9.
Waldron CA. Fibro-osseous lesions of the jaws. J Oral Maxillofac Surg 1993;51:828-35.  Back to cited text no. 9
    
10.
Kuta AJ, Worley CM, Kaugars GE. Central cementoossifying fibroma of the maxillary sinus: A review of six cases. AJNR Am J Neuroradiol 1995;16:1282-6.  Back to cited text no. 10
    
11.
Waldron CA, Giansanti JS. Benign fibro-osseous lesions of the jaws: A clinical-radiologic-histologic review of sixty-five cases. Oral Surg Oral Med Oral Pathol 1973;35:190-201.  Back to cited text no. 11
[PUBMED]    
12.
Pindborg JJ, Kramer IR. Histologic Typing of Odontogenic Tumors, Jaw Cysts and Allied Lesions. International Histological Classification of Tumors. Geneva: WHO; 1971. p. 31-4.  Back to cited text no. 12
    
13.
Kramer IR, Pindborg JJ, Shear M. Neoplasm and other lesions related to bone. In: WHO. Histologic typing of odontogenic tumors. Berlin. Springer-Verlag; 1992. p. 28-31.  Back to cited text no. 13
    
14.
Liu Y, Wang H, You M, Yang Z, Miao J, Shimizutani K, et al. Ossifying fibromas of the jaw bone: 20 cases. Dentomaxillofac Radiol 2010;39:57-63.  Back to cited text no. 14
    
15.
Ong AH, Siar CH. Cemento-ossifying fibroma with mandibular fracture. Case report in a young patient. Aust Dent J 1998;43:229-33.  Back to cited text no. 15
    
16.
Brademann G, Werner JA, Jänig U, Mehdorn HM, Rudert H. Cemento-ossifying fibroma of the petromastoid region: Case report and review of the literature. J Laryngol Otol 1997;111:152-5.  Back to cited text no. 16
    
17.
Feller L, Buskin A, Raubenheimer EJ. Cemento-ossifying fibroma: Case report and review of the literature. J Int Acad Periodontol 2004;6:131-5.  Back to cited text no. 17
    
18.
Hahn SB, Kang ES, Jahng JS, Park BM, Choi JC. Ossifying fibroma. Yonsei Med J 1991;32:347-55.  Back to cited text no. 18
    
19.
Hamner JE 3 rd , Scofield HH, Cornyn J. Benign fibro-osseous jaw lesions of periodontal membrane origin. An analysis of 249 cases. Cancer 1968;22:861-78.  Back to cited text no. 19
    
20.
Delilbasi C, Sencimen M, Okcu KM. A large mass in the maxilla: Clinical features and differential diagnosis. J Can Dent Assoc 2009;75:269, 272-3.  Back to cited text no. 20
    
21.
Sanchis JM, Peñarrocha M, Balaguer JM, Camacho F. Cemento-ossifying mandibular fibroma: A presentation of two cases and review of the literature. Med Oral 2004;9:69-73.  Back to cited text no. 21
    
22.
Brannon RB, Fowler CB. Benign fibro-osseous lesions: A review of current concepts. Adv Anat Pathol 2001;8:126-43.  Back to cited text no. 22
    
23.
MacDonald-Jankowski DS. Fibro-osseous lesions of the face and jaws. Clin Radiol 2004;59:11-25.  Back to cited text no. 23
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]
 
 
    Tables

  [Table 1], [Table 2]



 

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