Successful Implant Placement in a Case of Florid Cemento-Osseous Dysplasia: A Case Report and Literature Review

Osseous-dysplasia (OD) refers to a group of fibro-osseous conditions of bone. It arises due to the superseding of bone to fibrous connective tissue.¹  Generally, 3 main classifications have been described for OD: periapical, florid, and focal cemental dysplasia.²  These lesions are reminiscent of histologic spectrum, albeit differ in their extent of jaw involvement.³  The florid cemento-osseous dysplasia (FCOD) is a disease of a rare distribution, the precise etiology of which has been remained occult.⁴  Basic histopathology studies unraveled the biological plausibility of reactive or dysplastic changes of the periodontal ligament.⁵  The epidemiologic studies point to the middle-aged black female as a predominant affected population; moreover, it also appears in Caucasians and Asians.^1,2,6  The results of a systematic review by MacDonald-Jankowski in 2003 illustrated the prevalence of FCOD as following: within 158 identified cases, 97% of which were females, 59% were black; 37%, Asian; and 3% were Caucasian.¹ 

Dental panoramic radiography provides a key diagnostic tool for FCOD. Radiographically, a variety of lesions exist, the size of which can vary from 1 cm to 11 cm.^5,7  Usually FCOD is located in the periapical alveolar area, albeit there is a strong possibility for aggressive lesions to extend more inferiorly or superiorly.^5,7  Likewise it is a poorly circumscribed lesion, which is deemed to be radiolucent initially, while maturation results in opacification. Evidences from number of cases introduce the classic appearance of FCOD as diffuse, lobular, irregular-shaped radiopacities throughout the alveolar process of the maxilla and mandible.^5,7,8  Given the significant role of X-ray images in diagnosis of FCOD, there is no need for biopsy and histopathological examination. It should be taken into consideration that biopsy may foster the risk of infection or fracture of the jaw and the resultant detrimental effects of the patient's health.⁹  We searched the PubMed database to reveal clinical and radiological findings on FCOD along with demographic data of cases from January 2012 to January 2017. The following searches were performed:

• cemento-osseous dysplasia

• florid cemento-osseous dysplasia

• cementoma

• osseous dysplasia

• #1 OR #2

• #3 OR #4

• #5 AND #6

The management of FCOD presents challenges, since it could persist asymptomatically for a long period of time, while the management of symptomatic patients is more challenging.¹⁰  It has been highly recommended that extraction and even any elective surgical procedures should be avoided n patients with FCOD. The surgical procedure in this group of patients may lead to infection, sequestrum formation, or fracture of the jaw.¹⁰  Therefore, treating the edentulous area near FCOD lesions is a challenge. Determining what the best treatment plan may be in these cases is a critical question for clinicians. Would implantation near FCOD lesions be a successful treatment plan?

Herein we describe evidence of a patient who was diagnosed with FCOD on the basis of clinical and radiographic aspects, who is successfully being treated by implantation. To the best of our knowledge, a case of FCOD with implant success has not been reported previously.

A 62-year-old Caucasian woman presented to the private office with a chief complaint of mandibular partial edentulous (bilateral posterior teeth), meeting the demand through implantation. Her familial and medical histories were unremarkable. The results of laboratory tests, including alkaline phosphatase, calcium, and phosphor, were within the normal limits.

Dental panoramic radiograph (OPG) showed the bilateral radiopaque lesions in edentulous regions of mandible and mandibular anterior teeth alike. Cone beam computerized tomography (CBCT) took aim at dissecting the lesions. CBCT showed several radiopaque lesions in the periapical region of mandibular incisors (Figure 1), surrounded with a radiolucent border. Likewise, 2 identical lesions were found bilaterally in the region of mandibular molars. All mandibular teeth were vital, and no root resorption was detected. The findings of X-ray images were attributable to FCOD.

After clarifying the possible adverse effects of the implant surgery, written informed consent was obtained from the patient. Conservative step-by-step 2-stage implant surgery was planned. Two dental implants were placed in the mandibular left edentulous region. Considering the underlying assumption of a devastating effect of any surgical cut regarding FCOD lesions, a highly conservative surgery procedure was performed to address this critical trait of the lesion. In keeping with the conservative theme of the implantation, OPG images were taken immediately after the surgery, as well as 2, 4, and 6 months after the surgery, and there was no change regarding the size and characteristics of the lesion, which reinforces that implantation was well adopted. After 6 months, the implant was loaded with fixed prosthesis. To iron out any possible devastating event, the OPG images were taken 12 and 18 months after the initial implant surgery. An optimal functional rehabilitation and complete integration of implants were shown in X-ray images 18 months after initial implantation. Additionally, there was no change in the size of FCOD lesion (Figure 2).

FCOD is a rare fibro-osseous lesion, with multifocal involvement in mandibular tooth-bearing areas.^4,11–13  Clinically this non-neoplastic, reactive fibro-osseous lesion is usually asymptomatic, albeit the possibility of dull pain, alveolar sinus tract, and overt exposure of bone into the oral cavity resulting from secondary infection has been left open.¹⁴  Although there is evidence of genetic and familiar histories for some oral lesions,^15,16  there is a poor familial and genetic background for FCOD.¹  A variety of terms have been used to report FCOD, such as multiple cemento-ossifying fibromas, multiple enostosis, sclerosing osteomyelitis, multiple osteomas, multiple enostosis, periapical cementoblastoma, gigantiform cementoma.¹⁷  In the second edition of the World Health Organization's classification of odontogenic tumors, the term gigantiform cementoma was replaced by florid cemento-osseus dysplasia.¹⁷ 

Evidence from a systematic review has shown that symmetry of the affected sextants is a salient trait of FCOD.¹  Several lines of evidence illustrated how best to combine clinical and radiographic features to become capable of reaching an expedient diagnosis.¹⁸  The radiologic findings of FCOD could vary from radiolucent lesions to areas of mixed radiopaque and radiolucent lesions and to mainly opaque masses, which is related to the degree of maturation of the lesion. During the maturation, lesions tend to become radiopaque.¹⁰  Paving the clinical investigations, there is now a growing body of evidence indicating that for all lesions visible in radiographs, histologic examination should not always be done. Therefore, only clinical and radiologic features are deemed to be critical to diagnosis of FCOD.^7,10,19  It has been reported that the disease is more common among Africans and Asians, and bilateral and symmetric lesions in the molars of black females.²⁰  The clinical and panoramic images findings of the FCOD cases reported during the last 5 years have been summarized in Table 1. Among the 27 identified cases, 83% were females; 52% were Caucasian; 32%, Asians; and 16% were black. 68% were asymptomatic and the most common radiographic finding was radiopaque masses (72%). The cumulative data of the past 5 years identified FCOD cases points to a shift from the black female to the Caucasian female as a predominant affected population. Our case was a 62-year-old Caucasian woman with bilateral symmetric lesions in edentulous posterior area, thus she was included in the high-risk group. She came to the office with no specific symptoms and requested for implantation in the mandibular partial edentulous area, while the radiographic images were illustrating symmetric radiopaque lesions bilaterally in mandibular posterior region, in agreement with data from the literature (Table 1). Based on both clinical and radiographic findings, the lesion was diagnosed as FCOD.

It has been elucidated that extraction and any even elective surgical procedures should be avoided in patients with FCOD. The core problems have been reported as poor healing, sequestrum formation, and risk of infection and fracture of the jaw.^9,10,21  It has been illustrated that the quality and quantity of jaw bones are 2 notable factors in making a decision for implant placement, and for the success rate of implants.²²  Holding the lessons from these findings, in our case, biopsy was not performed. Gerlach et al¹⁷  reported a case of implant failure in a patient with both FCOD and cemento-ossifying fibroma. They reported that the patient returned with swelling, pain, and the sensation of a “loose” implant. Cutting the lesion during implantation could be the culprit in such an implant failure. Since the FCOD lesion of the right side of our patient was located in the center of the edentulous area, we did not plan implantation for the right edentulous site of the mandible. Surrogate treatment plan was a 3-unit dental bridge to obviate the need for function in this area. As the chief complaint of our patient was mandibular partial edentulous (bilateral posterior teeth) for the left edentulous site of the mandible a highly conservative step-by-step implant surgery performed, which successfully led to optimal functional rehabilitation.

There is nothing like a clinical success to open up a new opportunity for therapeutics. This report confirms that treating the edentulous area near the FCOD lesions can be planned, if conservative step-by-step implant placement has been considered. More studies in more patients are needed to confirm results of such a therapeutic modality. It is also notable that dissecting radiographic images of the patients is of high importance, and it is necessary for the successful implant placement in all patients, with a relatively high emphasis on patients with suspected lesions. Considering the therapeutic challenges in FCOD cases, we recommend recall examinations, prophylaxis, and regular follow-up with more emphasis on improvement of oral hygiene to prevent tooth loss.

Abbreviations

CBCT

cone beam computerized tomography

FCOD

florid cemento-osseus dysplasia

OD

osseous-dysplasia

OPG

panoramic radiograph

The authors declare that they have no conflict of interest. The authors declare that there is no financial support for this work.