|Year : 2014 | Volume
| Issue : 1 | Page : 22-25
Recurrent central odontogenic fibroma: An uncommon clinical presentation
Suresh K. Sachdeva1, Pradhuman Verma1, Dinesh Verma2, Kanika Gupta Verma3
1 Department of Oral Medicine and Radiology, Surendera Dental College and Research Institute, Sri Ganganagar, Rajasthan, India
2 Department of Oral and Maxillofacial Surgery, Surendera Dental College and Research Institute, Sri Ganganagar, Rajasthan, India
3 Department of Pedodontics and Preventive Dentistry, Surendera Dental College and Research Institute, Sri Ganganagar, Rajasthan, India
|Date of Web Publication||21-May-2014|
Suresh K. Sachdeva
Departments of Oral Medicine and Radiology, Surendera Dental College and Research Institute, H.H Gardens, Power House Road, Sri Ganganagar 335 001, Rajasthan
Source of Support: None, Conflict of Interest: None
Central odontogenic fibroma (COF) is an uncommon benign neoplasm composed by varying amounts of inactive looking odontogenic epithelium embedded in a neoplastic mature and fibrous stroma. It originates from odontogenic ectomesenchyme and accounts for 0.1% of all odontogenic tumours. It appears as an asymptomatic expansion of the cortical plates of jaws. Radiographically, it appears as unilocular or multilocular radiolucency. It responds well to surgical enucleation. Recurrence is rare. Here is a rare case of recurrence of COF reported in 18-year-old female in the left mandibular posterior region after 1 year.
Keywords: Central, mandible, odontogenic fibroma, recurrent
|How to cite this article:|
Sachdeva SK, Verma P, Verma D, Verma KG. Recurrent central odontogenic fibroma: An uncommon clinical presentation. Saudi Surg J 2014;2:22-5
|How to cite this URL:|
Sachdeva SK, Verma P, Verma D, Verma KG. Recurrent central odontogenic fibroma: An uncommon clinical presentation. Saudi Surg J [serial online] 2014 [cited 2020 Jul 15];2:22-5. Available from: http://www.saudisurgj.org/text.asp?2014/2/1/22/132900
| Introduction|| |
The World Health Organization (WHO) has defined the odontogenic fibroma as "a benign odontogenic neoplasm of fibroblastic origin characterized by relatively mature collagenous fibrous tissue and varying amounts of odontogenic epithelium with potential to occur in either a central or an extra osseous location." The osseous part is designated as central odontogenic fibroma (COF).  The peak incidence is in the second decade of life with predominance in females in mandibular posterior region.  In general, the lesion is asymptomatic except the swelling of the jaw and less frequently pain and paraesthesia are observed. Radiographically, all COFs appears as well delineated, non-corticated radiolucency with or without expansion of cortical plates.  The mode of treatment for COF is enucleation and curettage. Recurrence is very rare. 
Here is a rare case of recurrence of COF reported in 18 years female in the left mandibular posterior region after 1 year.
| Case Report|| |
The present case report is about a 18-year-old female patient who reported to the Department of Oral medicine and Radiology with the chief complaint of painless swelling over the left side of the face since 1 month. History of presenting illness revealed similar type of extra-oral swelling with intraoral pedunculated soft-tissue mass at the same site 1 year back [Figure 1]. There was a history of surgical extraction of 38 along with excision of soft-tissue mass 1 year back [Figure 2]. The histopathological report of the excised specimen was suggestive of COF. Patient was put on follow-up, but she didn't report back. Now after 1 year she reported to the department with large painless swelling in the lower 1/3 rd of the left side of face, which gradually increased to attain the present in size. There was no history of paraesthesia and pus discharge. Medical and family history was non-contributory.
General physical examination revealed gross facial asymmetry with solitary diffuse swelling over the left side of lower jaw, extending from (the) zygomatic arch superiorly up to 2.4 cm below the lower border of body of mandible inferiorly. Anteriorly, it extends from left oral commissure to the angle of mandible posteriorly. The overlying skin was of normal color without any discharge [Figure 3]. On palpation, overlying skin was pinchable with no localized increase in temperature. The swelling was non-tender, firm in consistency and non-fluctuant. Two left sub mandibular lymph nodes which were tender, mobile, firm in consistency and measuring 1.0 and 1.5 cm in maximum dimensions respectively were noted on palpation. All the vitals were within the normal limits.
Intraoral examination showed solitary, exophytic soft-tissue growth with diffuse reddish blue discoloration of the overlying mucosa, measuring 9 × 4 × 3.5 cm extending from mesial surface of 36 to pterygo-mandibular region posteriorly. Medially it extends 1.5 cm into the floor of mouth and laterally causing obliteration of buccal vestibule. There were cuspal indentations of upper molars over the superior surface of the lesion [Figure 4]. On palpation, the mass was mobile, firm in consistency, non-tender and did not cause any bleeding on provocation. The expansion of buccal and lingual cortical plates was noted. The electrical pulp test of 36 and 37 showed normal response.
|Figure 2: Previous Orthopantomogram showing mesio-angular 38 with pericoronal radiolucency|
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|Figure 4: Intra-oral photograph showing the pedunculated mass in left retro molar region|
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Based upon the history and clinical examination provisional diagnosis of benign bony lesion involving left mandibular angle was made and differential diagnosis of recurrent COF, central giant cell granuloma, Ameloblastic Fibroma, Pindborg's tumor and Odontogenic Myxoma were considered.
Hematological investigations revealed raised erythrocyte sedimentation rate. All the other values were within the normal limits. Orthopantomogram showed well-defined oval shaped homogenous radiolucency with corticated margins measuring 1.5 cm in maximum diameter extending from the anterior border of ramus of mandibular up to 1.5 cm in front of posterior border of ramus of mandible. There was erosion of bone at the left angle of mandible causing discontinuity of lower border of body of mandible. There was loss of corticated margins of inferior dental canal [Figure 5].
|Figure 5: Orthopantomogram showing extensive bony destruction on left side of jaw|
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Contrast computed tomography scan (axial and coronal) showed large heterogeneous moderately enhancing expansile, lytic soft-tissue mass (9 × 6 × 5 cm) involving left angle of mandible and extending along the condyle and body of the mandible. The mass showed central non enhancing areas. Significant bone erosion was noted at the left angle of mandible [Figure 6].
|Figure 6: Axial computed tomography scan showed large heterogeneous moderately enhancing expansile lytic soft tissue mass (9 × 6 × 5 cm) involving left angle of mandible|
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The incisional biopsy was taken and sent for histo-pathological examination which showed intermingling bundles of loose collagen fibers situated in a myxoid stroma along with few odontogenic epithelial nests suggestive of COF (WHO type) [Figure 7]. The patient was referred to oral and maxillofacial surgery department where the resection of the lesion was done along with hemi-mandibulectomy followed by reconstruction [Figure 8] . The resected specimen was again sent for histo-pathological examination which confirmed the previous diagnosis.
|Figure 7: Photomicrograph of histopathology slide of specimen (H and E, ×40)|
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|Figure 8: Intra-operative photograph showing resection of the lesion with hemi-mandibulectomy followed by reconstruction|
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| Discussion|| |
In a study by Wesley et al. suggested a set of diagnostic criteria for COF as follows. (a) Clinically, the lesion is central in the jaws and has a slow persistent growth that results in painless cortical expansion. (b) Radiographically, its appearance varies, but such as ameloblastoma and odontogenic myxoma, most examples are multilocular radiolucent lesions that involve relatively large portions of the jaws in the later stages. (c) Histopathologically, the most consistent feature is a tumor composed predominantly of mature collagen fibers with numerous inter-spreaded fibroblasts. The presence of small rest of inactive odontogenic epithelium is a variable feature. (d) The lesion is benign and responds well to surgical enucleation with no tendency to undergo malignant transformation.  Using these criteria, the present case is tenth such case diagnosed in mandible. This confirms rarity of the tumor. Gardner in 1980 classified lesions as COF into three categories: (a) The hyperplastic dental follicle. (b) A fibrous neoplasm with collagenous fibrous connective tissue containing odontogenic epithelium-(simple type). (c) A lesion with fibrous tissue, odontogenic epithelium and myxoid areas-(WHO type). The lesion may or may not contain dysplastic dentine or tissue like cementum.  In our case the histopathology proved it to be COF (WHO type). The varying clinical behavior and radiographic appearance leads to odontogenic myoxoma, ameloblastic fibroma in differential diagnosis. The clinical behavior of odontogenic myxoma may be similar to COF, but soap-bubble appearance radiographically is feature of myxoma.  Ameloblastic fibroma are distinguished from COF by the fact that both epithelial and mesenchymal components are neoplastic, while in COF, is only the mesenchymal.  Management of COF includes enucleation and curettage as it is easily removed and the lesion is slow-growing. Recurrence has been reported, but is uncommon. The different authors have reported different recurrence rates ranging 7.4-13%.  The reason for recurrence is due to an incomplete removal of the initial tumor as it is not encapsulated and in some cases COF has involved the inferior alveolar nerve which makes its precise excision more difficult.  The present case was also a case of recurrence of COF as patient was diagnosed with similar lesion in the same site 1 year back. The reason for recurrence in our case may be that the lesion was not completely removed at time of first surgery. According to Marx and Diane, if reoccurrence is observed in COF, incisional biopsy must be done because their presentation may be suggestive of aggressive disease, which was done in present case.  According to the literature available, the cases in which the recurrence has been reported should be treated with more extensive surgical excision.  The similar treatment planning was chosen in this case as there was recurrence with more extensive lesion and aggressive features in the young female patient.
| Conclusion|| |
Clinically and radiographically, COF may mimic to other similar lesion. The general dental practitioner must be aware of clinical features, imaging findings and histopathological features of COF. This case report focus on the importance of patient's periodic clinical and radiographic examinations and histopathological examination in case of COF. At the initial presentation of COF, it should be surgically excised completely to avoid recurrence at later stage. Even though, the recurrence rate of COF is low, a post-operative follow-up is a must after surgery.
| Acknowledgement|| |
Proper institutional guidelines were followed and informed consent of the patient was obtained.
| References|| |
|1.||Weber A, van Heerden WF, Ligthelm AJ, Raubenheimer EJ. Diffuse peripheral odontogenic fibroma: Report of 3 cases. J Oral Pathol Med 1992;21:82-4. |
|2.||Gardner DG. Central odontogenic fibroma current concepts. J Oral Pathol Med 1996;25:556-61. |
|3.||Eversole LR. Odontogenic fibroma, including amyloid and ossifying variants. Head Neck Pathol 2011;5:335-43. |
|4.||Wesley RK, Wysocki GP, Mintz SM. The central odontogenic fibroma. Clinical and morphologic studies. Oral Surg Oral Med Oral Pathol 1975;40:235-45. |
|5.||Gardner DG. The central odontogenic fibroma: An attempt at clarification. Oral Surg Oral Med Oral Pathol 1980;50:425-32. |
|6.||Rocha AC, Gaujac C, Ceccheti MM, Amato-Filho G, Machado GG. Treatment of recurrent mandibular myxoma by curettage and cryotherapy after thirty years. Clinics (Sao Paulo) 2009;64:149-52. |
|7.||Chen Y, Li TJ, Gao Y, Yu SF. Ameloblastic fibroma and related lesions: A clinicopathologic study with reference to their nature and interrelationship. J Oral Pathol Med 2005;34:588-95. |
|8.||Svirsky JA, Abbey LM, Kaugars GE. A clinical review of central odontogenic fibroma: With the addition of three new cases. J Oral Med 1986;41:51-4. |
|9.||Hrichi R, Gargallo-Albiol J, Berini-Aytés L, Gay-Escoda C. Central odontogenic fibroma: Retrospective study of 8 clinical cases. Med Oral Patol Oral Cir Bucal 2012;17:e50-5. |
|10.||Marx RE, Diane S. Oral and Maxillofacial Pathology: A Rational for Diagnosis and Treatment. 2 nd edition: Quintessence Publishing Co. Inc.; 2007. p. 672-4. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]