CASE REPORT

https://doi.org/10.5005/jp-journals-10063-0146
CODS Journal of Dentistry
Volume 15 | Issue 2 | Year 2023

Central Odontogenic Fibroma: A Rare Case Report and Review of Literature

Sandeep Garg1, Navjot Kaur2, Supreet Kaur3, Harvinder Singh4

1,2Department of Oral and Maxillofacial Surgery, Rayat Bahra Dental College and Hospital, Rayat Bahra University (RBU), Sahibzada Ajit Singh Nagar, Punjab, India

3Department of Oral and Maxillofacial Surgery, DJ College of Dental Sciences and Research, Ghaziabad, Uttar Pradesh, India

4Department of Orthodontics & Dentofacial Orthopaedics, National Dental College and Hospital, Sahibzada Ajit Singh Nagar, Punjab, India

Corresponding Author: Navjot Kaur, Department of Oral and Maxillofacial Surgery, Rayat Bahra Dental College and Hospital, Rayat Bahra University (RBU), Sahibzada Ajit Singh Nagar, Punjab, India, Phone: +91 9915873990, e-mail: dr.navjot88@gmail.com

Received: 12 February 2024; Accepted: 10 March 2024; Published on: 17 May 2024

ABSTRACT

Aim: The aim of this report is to present an additional case of mandibular central odontogenic fibroma (COF) of complex type in a pediatric patient as well as to compare its clinical, radiological, and histological features with the cases reported to date in the review and to understand the need of long-time postoperative follow-up and to develop a better understanding of COF.

Background: Odontogenic fibromas can be divided into central (intraosseous) odontogenic fibroma and peripheral (extraosseous) odontogenic fibroma according to the anatomical sites involved. COF is a rare, benign, slow-growing intraosseous mesenchymal odontogenic tumor of the jaws. It presents a diagnostic dilemma to the clinician and the pathologist because its clinical and radiological features resemble other odontogenic and/or nonodontogenic tumors, and the differential diagnosis is based on histological examination.

Case description: Here, a case of COF of the posterior mandible in a 13-year-old boy is described. The lesion was an asymptomatic swelling with well-defined borders covered by normal mucosa. It presented as a well-defined unilocular radiolucency along with thinning of buccal and lingual cortices. The lesion was surgically enucleated and analyzed histopathologically. There were no postoperative complications. The aim of this case report is to present a case of COF in the posterior mandible and to compare its clinical, radiographic, and histopathological features with the present literature.

Conclusion: Highlighting a subtype that was dropped from the last World Health Organization Classification of Head and Neck Tumors is important to accumulate more information about this lesion and to show its different features. Despite its rarity, COF should be included in the differential diagnosis of intrabony tumors of the jaws.

Clinical significance: The findings of this case report can better educate oral and maxillofacial surgeons about the unusual nature of this lesion, help establish a correct diagnosis and give the appropriate therapeutic management.

How to cite this article: Garg S, Kaur N, Kaur S, et al. Central Odontogenic Fibroma: A Rare Case Report and Review of Literature. CODS J Dent 2023;15(2):55–58.

Source of support: Nil

Conflict of interest: None

Patient consent statement: The author(s) have obtained written informed consent from the patient’s parents/legal guardians for publication of the case report details and related images.

Keywords: Case report, Central odontogenic fibroma, Differential diagnosis, Tumor enucleation

INTRODUCTION

Central odontogenic fibroma (COF) is a rare, benign mesenchymal odontogenic tumor occurring within the jaw.1 The World Health Organization (WHO) has defined it as a tumor consisting of mature fibrous connective tissue, with variable amounts of inactive-looking odontogenic epithelium, with or without the presence of calcification.2 Histologically, there are two main subtypes of COF—the simple COF (epithelium-poor) type and the complex (epithelium-rich) type, also called the WHO type.3-5 However, this classiႡcation was discontinued in the year 2017 because of its poor definition and documentation. The new classification of the lesion is now based on its location (central or peripheral) rather than on the basis of histological features.6

In this case report, we describe a case of COF in a 13-year-old male, occurring in the left posterior mandibular region.

CASE DESCRIPTION

A 13-year-old male child was referred to the oral surgery clinic with the chief complaint of swelling in the lower left posterior region of the jaw. The patient noticed the swelling 2 months ago when he experienced pain while chewing. On intraoral examination, a firm swelling measuring approximately 2 × 2.5 cm over the left mandibular retromolar region abutting the tonsillar area was noticed. The posterior buccal sulcus was found to be obliterated. The overlying mucosa was inflamed, but no scar, sinus or fistula was observed. Palpation revealed thinning of both the buccal and lingual cortices. No ulceration, regional paresthesia or lymphadenopathy was noticed. The computed tomography (CT) scan sections revealed a unilocular radiolucent lesion with well-defined margins in the left mandibular angle region associated with an unerupted permanent second molar (Figs 1 and 2). Complete enucleation of the lesion and the removal of the associated tooth and its follicular lining were done under general anesthesia (Fig. 3). Adjuvant therapy in the form of peripheral osteotomy and chemical cauterization was also done to prevent a recurrence. The histopathological analysis of the biopsied tissue showed stratified squamous epithelium and highly cellular connective tissue with plump fibroblasts and numerous scattered inactive appearing islands of the odontogenic epithelium (Fig. 4). Calcified cementum-like material was also noticed in isolated areas of connective tissue stroma (Fig. 5). These histopathological features were conclusive to establish a diagnosis of COF. Regular follow-up was carried out to rule out any recurrence. Satisfactory healing with no recurrence noted till 1 year of postoperative follow-up.

Fig. 1: Preoperative CT scan (coronal view)

Fig. 2: Preoperative CT scan (sagittal view)

Fig. 3: Intraoperative photograph

Fig. 4: Hematoxylin and eosin-stained section showing highly cellular fibrous connective tissue stroma with plump fibroblasts (red arrow) and numerous scattered inactive appearing odontogenic epithelium islands (yellow arrow) (40×)

Fig. 5: Calcified cementum-like material seen in the connective tissue stroma (hematoxylin and eosin, 40×)

DISCUSSION

According to Wesley et al.,7 odontogenic fibroma is diagnosed by the following criteria:

Gardner, in 1980,8 classified COF into three types:

The most recent literature review was by Kim et al.,9 who presented a report of 29 cases of COF in the Korean population along with tooth management, with an average age of 33.1 years and a male-to-female ratio of 0.8. The authors found the lesions to be more frequent in the maxilla (65.5%) than in the mandible (34.5%). Bennabi et al.,10 in their review, identified 135 cases of COF with a mean age of 30 years and a male-to-female ratio of 0.8. Out of these, 53.3% were present in the mandible and 46.7% in the maxilla. Out of the lesions located in the mandible, 58.3% were present in the molar, while 38.9% involved the premolar region, and 26.4% of the lesions involved the ramus. In the maxilla, 63.4% of the lesions were present in the premolar region, followed by the anterior region (49.2%) and finally the molar region (19%). In the review by Correa Pontes et al.,11 52.6% were found in the mandible and 45.1% in the maxilla, with a male-to-female ratio of 1.13:1 and an age range of 3–77 years. The patient’s age in this case report (13 years old) was much lower than the reported mean age of COFs (40 years old). Also, the patient’s gender (male) did not correspond to the overall gender predilection (strong female predilection). However, the site of the tumor in our patient was in accordance with the predominant site (posterior mandible).

The most common clinical signs are swelling and facial asymmetry. Also, COF shows a slow growth rate that results in painless expansion of the cortices. Clinical symptoms such as pain and paresthesia are rare. Dental signs, such as delayed tooth eruption mobility, are also very uncommon.12 Our case was without any symptoms but with intraoral swelling and expansion of the cortices.

Radiologically, COF presents as a well-defined unilocular or multilocular radiolucent lesion. Complications such as severe resorption and/or displacement of the roots of adjacent teeth are mostly associated with the multilocular radiolucent type.13 Bennabi et al.10 found that the majority of the lesions were unilocular (54.1%). However, Correa Pontes et al.11 found a prevalence of multilocular radiolucencies with cortical thinning and perforation, tooth displacement and bone expansion. The radiological appearance of our case was unilocular expansile radiolucency in the left mandibular angle region with well-defined margins and an unerupted tooth bud of the permanent second molar. However, no evidence of adjacent tooth displacement or resorption of roots was seen.

Histologically, the simple odontogenic fibroma has stellate fibroblasts arranged in a whorled pattern. Sometimes, odontogenic islands may be present with foci of dystrophic calcifications.5 The WHO type is complex with odontogenic epithelium seen in long strands or nests in cellular fibrous connective tissue matrix. The fibrous component may vary from myxoid to densely hyalinized. Calcifications made of cementum-like material or dentinoid are also seen.5 Bennabi et al.10 found almost equal distribution between complex and simple types (34.1 and 28.9%, respectively). However, Correa Pontes et al.,11 in their study observed WHO type to be most common (68.78%) followed by simple type (31.22%). According to this histologic pattern, the case reported in this study case is WHO type with the presence of stratified squamous epithelium and fibrocollagenous connective tissue stroma with nests of odontogenic epithelium and few scattered areas of calcified cementum-like material. However, since both the simple and complex subtypes show nearly the same clinical signs and symptoms, this classification is not in further use now.

The COF shows a good prognosis with a very low recurrence rate reported in the literature. Correa Pontes et al.,11 in their systematic review of the literature, reported a recurrence rate of 10%. They also found that the maxilla presents a greater rate of recurrence than the mandible. Larger, multilocular and aggressive lesions were also associated with a higher rate of recurrence in their study. However, Bennabi et al.10 found a lower rate of recurrence (6%) in their literature review.

Histopathological differential diagnoses of COF include desmoplastic fibroma, ameloblastic fibroma, desmoplastic ameloblastoma, and odontogenic myxoma. An absolute distinction is mandatory between desmoplastic fibroma and myxoma because they have a higher recurrence rate as compared to COF.13

The treatment of choice of COF is surgical enucleation. The lesion is easily enucleated because it shows very little adhesion to bone and no tendency to convert into a malignant lesion.14 The lesion has a good prognosis. However, careful follow-up for a few years is necessary.15

CONCLUSION

Apart from the clinical and radiographic findings, histopathological examination is also very necessary for every specimen so as to make a correct final diagnosis. Since COFs are reported to have low recurrence rates, an incisional biopsy followed by enucleation and curettage is considered the preferred choice of management.16

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