Cemento-ossifying mandibular fibroma: A presentation of two cases and rewiev of the literature

SANCHIS JM, PEÑARROCHA M, BALAGUER JM, CAMACHO F. CEMENTO-OSSIFYING MANDIBULAR FIBROMA: A PRESENTATION OF TWO CASES AND REVIEW OF THE LITERATURE. MED ORAL 2003;9:69-73.

SUMMARY

We present two clinical cases of radioluscent mandibular lesions in young women that simulated chronic periapical infectious pathology. The detection of both cases was fortuitous since they were totally assymptomatic. Diagnosis was reached in one case (upon periapical surgery and anatomo-pathologic study) after endodontic treatment and after verifying non-resolution of affected periapical area. The other case was an extensive lesion, which involved the periapices of the four inferior incisors in which surgery was directly performed upon verifying pulp vitality of these teeth. After surgery endodontic treatment was performed on the teeth that had lost their vitality.
In both cases the histopathologic tests revealed the presence of a cemento-ossifying fibroma, the initial clinical and radiographic diagnosis of which could easily be overlooked.

Key words: Periapical lesion, fibroma, cementum

INTRODUCTION

Cemento-ossifying fibroma (COF) is considered a benign osseous tumor, very closely related to other lesions such as fibrous dysplasia, cementifying periapical dysplasia or cemento-osseous florid dysplasia, however, forming its own entity according to the 1992 classification of the WHO (1). It is a bony tumor of the maxillas of possible odontogenic origin. It is believed to derive from the cells of the periodontal ligament (2-4). Consequently, one of its principal characteristics is the massive formation of cementum, cementoid substance or calcified material in the interior of a predominantly fibrous tissue (5-7).

They manifest themselves as slow-growing, assymptomatic, intraosseous masses, most frequent in women between 35 and 40 (7,8). Differential diagnosis should be performed, preferably with other fibro-osseous lesions of the maxilla such as fibrous dysplasia or osseo-cementifying dysplasia (5,6).

In this article we present two cases of cemento-ossifying fibroma manifested as radioluscent and assymptomatic mandibular lesions, the differential diagnosis of which, based on clinical manifestations and conventional radiographic studies, could be controversial. There are some stand-out publications that discuss the possibility of benign (9-11) or malignant (12-14) lesions to manifest in some way as periapical lesions of which diagnostic error could lead to incorrect treatment.

CLINICAL CASES

CASE 1
A 32 year old woman in which a radioluscent image of 0.8 x 1cm in the periapical level of the left canine was observed upon making a panoramic radiograph for conventional dental treatment. The patient reported traumatism to the area some years earlier. Her dentist treated the tooth endodontically and observed that the pulp was vital. After a two month follow-up changes on the apical radioluscency was observed (Figure 1A) and periapical surgery of the lesion was performed (Figure 1B). 

At the same time a tissue sample was obtained for a histopathological study that showed a fibrous connective tissue with bone trabeculae and small, rounded, calcified foci that group themselves into lobulated masses (Figure 1C). The anatomopathologic diagnosis was cemento-ossifying fibroma. After one year of evolution, the patient did not complain of any type of symptomatology whatsoever, and upon making a new panoramic radiograph, we were able to appreciate good bony regeneration on the affected area.

CASE 2
A 35-year-old woman, of uninteresting medical history, sought to her dentist for conventional treatment. The panoramic radiograph revealed a 4x2-cm radioluscent image, located around the 4 apices of the inferior incisors, which however, was clinically assymptomatic. The pulp vitality of the affected teeth was positive and the blood test did not show altered values. The computerized axial tomography (CAT scan) of the mandible revealed a round osteolytic lesion located between the mandibular canines, with well defined borders, which thinned and slightly made the lingual and vestibular corticals convex (Figure 2A). Excisional biopsy of the lesion was performed. Histopathologic analysis revealed a fibrous stroma with abundant collagen fibers, in the sinus of which was found basophil masses of diverse morphology with areas of dystrophic calcification (Figure 2B). The histopathologic diagnosis established was cemento-ossifying fibroma. On the posterior follow-up appointments, pulp vitality tests of the 4 inferior incisors were negative hence, endodontic treatment was done on them. After one year evolution the patient was assymptomatic and without radiographic alterations.

DISCUSSION

The cemento-ossifying fibroma commonly affects adults between the third and fourth decade of life, although it occurs in a wide interval of ages. Predilection is on the mandible and on the feminine sex (5,6,8,15). Su et al. (6) expressed that in 50% of the cases of COF some initial symptom is manifested. 76% of the cases were women whose mean age was 32 years. In some cases the existence of a previous trauma on the area has been established as a possible etiological agent (4), as the case provided by Wening et al. (16) of a 26 year old patient with facial bone traumatism and cemento-ossifying fibroma on the superior maxilla. In one of our cases the patient also mentioned a history of previous trauma to the mentonian zone. Comparable to those cases garnered in the literature, those that we presented affected women between 30 and 40 years old, with slow-growing and assymptomatic lesions.

These tumors usually appear as slow growing, assymptomatic masses, the diagnosis of which could be confused with idiopathic Stafne's bone cavity (17). They could occasionally grow more rapidly and extensive (18) and could even provoke a mandibular fracture (19). An abundant reference exists in the literature regarding the aggressive behavior of these lesions (20-22), especially when manifested in the premaxillary locations as the mastoid region (4), ethmoidal sinuses (2,3), parapharyngeal spaces (23) or ear auricle (24). Moreover, a clinical form exists which is basically more aggressive, and is more common in those less than 15 years of age which receives the name juvenile cemento-ossifying fibroma (25). Some multiple forms of COF are also described in the literature (28,29).

Both cases were manifested as well defined, radioluscent, intraosseous lesions, without affecting nor displacing affected teeth and without the area of peripherical osteocondensation of which according to Su et al. (6) could appear in up to 45% of the cases. In the second case, CAT scan revealed a considerable thinning and expansion of the lingual and vestibular corticals, which should be considered to highlight other types of lesions. At times and due to a diverse grade of tumor calcification, radiographic diagnosis could display a partial radioluscency or opacity, and not to mention a likely controversial CAT scan (30).

A differential diagnosis is brought up with other fibro-osseous entities such as the osseo-cementifying dysplasia (florid and periapical and focal dysplasia) (5,6), fibrous dysplasia (1) and other such as osteoid osteoma, osteoblastoma or chronic sclerosing osteomyelitis (31). When there is close proximity to the apices of the teeth, as the presented cases, differential diagnosis could be done with chronic periapical periodontitis (32-35), although other radioluscent mandibular lesions should never be pointed out as a solitary osseous cyst, keratocyst, ameloblastoma, giant cell granuloma, myxoma, etc (36). In this perspective other keratocysts are described (10,11) which are manifested as periapical lesions, adenomatoid odontogenic tumor (12) and diverse malignant lesions as multiple myeloma (13), lung carcinoma metastases (14) or renal carcinoma (15). The two presented cases were also manifested as periapical lesion, only one was treated as is by her dentist while the other was only correctly diagnosed upon surgery. What is common in both cases is the lack of symptomatology, non-specific radiology (the absence of intralesional calcifications) and the facility to come up with an erroneous diagnosis since it is closely related to the dental apices.

In its histopathology it is typical to encounter a benign fibroblastic stroma with varying cellularity, although mitosis is rare. Within the fibrous stroma are mineralized tissue masses of basophil aspect that correspond to osteoid or cementoid material. This is distributed throughout the tumor in a greater or lesser quantity and is accompanied by dystrophic calcifications that adopt a basophillic or darker color (5,7). According to Voytec et al. (32) the differences between fibrous dysplasia and COF is not always exhibited in each and every section of the tumor, the reason for which some cases a poorly diagnosed. The histologic pattern of the two lesions coincide in what the authors expressed (5,7,31) with respect to a predominantly fibrous cellularity and the presence of masses of cementifying tissues, with greater or lesser grade of calcification.

Surgical management consists of conservative extirpation of the lesion, facilitated by the existence of a zone of surgical cleavage and the presence of a well-defined tumor mass. The two cases were treated surgically, performing a simple excision and curettage of the cavity, which is actually the proposed treatment for these types of tumors (16,34,35), and later observing a good evolution of the patients in both cases. In the more aggressive cases surgical difficulty is due more to restitution of the affected bone (floor of the maxillary sinus, wall of the nasal cavity, etc.) than the tumor extirpation itself, which should always be completed with curettage of the surgical mass (16). Tumor recurrences are described (33) but are very uncommon (34,35). After 1-year follow-up of the patients no recurrent lesions were observed.

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