Florid cemento-osseous dysplasia is a controversial fibro-osseous lesion of the jaws. Literature regarding the subject has been briefly reviewed illustrating the wide range of opinions as to the tissue of origin, biological nature and nomenclature. This is the first reported case in a Hispanic individual. The report is presented to review the histopathologic similarity of this lesion to other fibro-osseous lesions, the importance of radiological interpretation in terms of differential diag­nosis and its significance in avoiding overtreatment and the complication of osteomyelitis.
 

Controversy concerning the tissue of origin and the biological nature of florid cemento-osseous dysplasia still exists. Opinions range from it being a dysplastic osseous lesion, illustrated by the names florid osseous dysplasia¹, multiple osteomas², mul- tiple enostosis³, Paget's disease of the mandible⁴, and multiple periapical osteofibromatosis⁵ to a dysplastic cemental lesion, illustrated by the names sclerotic cemental masses⁶, multiple cemen- tomas⁷, gigantiform cementoma³'⁹, monstrous cementoma⁸, familial multiple cementomas⁹, periapical cementoblastoma¹⁰, periapical cemen- tal dysplasia with multiple lesions¹¹, and sclerosing cementomas¹²; through a combined dysplastic cemento-osseous lesion, illustrated by the name multiple cemento-ossifying fibroma¹³; to it being an inflammatory lesion, as seen in the names sclerosing osteitis¹⁴, sclerosing osteomyelitis¹⁵, chronic sclerosing osteomyelitis, and chronic dif- fuse sclerosing osteomyelitis¹⁶. However, it is believed that these latter names have been erroneously used to describe florid cemento-osseous dysplasia, or florid cemento-osseous dysplasia with superimposed inflammation.¹⁷¹⁸

The entity designated as florid cemento-osseous dysplasia refers to changes in the jaws characterized radiographically by dense radiopaque foci, mixed with radiolucencies¹'⁶¹⁸"²⁰ which, depending on the clinical condition, may or may not be symptomatic. There may be displacement of the cortical bony plates as well as inflammation due to infection.¹ Familial forms have been described.²¹²⁴ Although the vast majority of cases are stated to occur in middle-aged black females, several cases have been reported in other ethnic groups,¹'¹⁸¹⁹²¹"²⁶ and a few in males²³²⁴. A case in a Hispanic female is being reported. 

A 33-year-old Mexican female was referred to the fourth author for extraction of multiple post- erior teeth with severe periodontitis. Her medical history was not significant except for intake of birth control "pills". Clinical examination revealed severe periodontitis with mobility of the maxillary right first molar, right lateral incisor, and left first molar, and mandibular left second and first molars, left second premolar, right second premolar and right second molar.

Radiographically, radiolucent-radiopaque areas were noted in the maxillary right canine-lateral incisor region [Fig. 1], and in the mandibular anterior region from the left canine to the lateral incisor [Fig. 2]. A radiopaque mass was noted at the apices of the maxillary central incisors. The pulps of all involved teeth tested were vital. During subsequent appointments, extraction of the mobile teeth mentioned, except the maxillary right lateral incisor, and incisional biopsy of the mandibular lesion were carried out. Extraction of mandibular incisors, maxillary right lateral incisor, and left sec- ond molar, with removal of the mandibular and maxillary lesions, and endodontic treatment of the maxillary right canine and mandibular left canine were carried out later.

The first biopsy specimen of the mandibular lesion [Fig. 3] was diagnosed in the Department of Oral Pathology, Radiology & Medicine, The Uni- versity of Iowa, as central ossifying fibroma. The rebiopsy and the maxillary biopsy [Fig. 4], done more than a month later, were diagnosed as central ossifying fibroma versus periapical cemental dysplasia. Further interpretation of the submitted radiographs referred additionally to another simi- lar, but smaller, mandibular mixed radiolucent- radiopaque mass distal to the mandibular right sec- ond molar, and to multiple, poorly to moderately well-defined, radiolucent areas superimposed over the roots of the mandibular left first molar, and maxillary left second molar. The appearance was interpreted as suggestive of periapical cemental dysplasia versus florid cemento-osseous dysplasia. The radiolucent areas associated with the man- dibular second molars were interpreted to repre- sent either manifestations of the same disease pro- cess, or rarefying osteitis due to periodontal dis- ease. At appointments three and five months post- operatively, the patient was asymptomatic and recovering well.

Integrating the histopathology and radiology reports, we came to the conclusion that this lesion represents a case of florid cemento-osseous dysplasia.
 

In the 1971 World Health Organization publica- tion "Histological typing of odontogenic tumors, jaw cysts and allied lesions", Pindborg ef al used the term gigantiform cementoma to describe a lesion which predominantly affected middle-aged black females, in which multiple sclerotic masses occupied the jaws. The term familial multiple cementomas was used as a synonym.⁹ The litera- ture before and after that publication contains a large number of case reports of similarity described disease processes under several names mentioned in the introduction.

In 1975, Waldron ef al described the condition as sclerotic cemental masses of the jaws.⁶ They considered these lesions as part of the spectrum of benign fibro-osseous lesions of periodontal liga- ment origin, representing an exuberant prolifera- tion of cementum. They maintained that the dense mineralized tissue is cementum, and elaborated on its differentiating characteristics under polarized light microscopy.

In 1976, Melrose ef al introduced the term florid osseous dysplasia, describing the process as an exuberant, multiquadrant involvement that repre- sents the end of the spectrum of osseous dysplasia which includes, according to their classification, cementoma, periapical cemental dysplasia, periapical fibrous dysplasia and periapical osteofibrosis.¹ The subjects in their study group were 34 females, out of whom 32 were black, with a mean age of 42.

Although in almost all references the lesion is thought to affect mainly middle-aged black females, several cases have been reported in other groups including Caucasoid²¹²⁵, Oriental¹'¹⁹²⁶ Indian¹⁹, and West Indian²⁶. Here, we report a case in a Hispanic (Mexican) female. Likewise, a few cases have been reported in males and in a child.²³²⁴ Cases with a familial pattern were notably reported in Caucasoids.²¹²⁴ Melrose ef al found that 14 of their 34 cases were associated with simple bone cysts.¹ There is also one report in a white male with hereditary hemorrhagic telangiec- tasia²⁵ but this was probably coincidental.

Clinically, the condition may be asymptomatic, and discovered as a coincidental radiographic finding, or may cause facial deformity resulting from displacement of cortical bony plates.¹ When symptomatic, there may be pain alone or in association with sinus tracks with minimal purulent drain- age.⁶ Interestingly, the majority of symptomatic cases occur in edentulous patients, either after extractions or wearing of dentures.¹ ,M 7,19,20,27 |_t j_s reasoned that the symptoms are probably related to secondary infection following tooth extraction or denture trauma, where the poorly vascularized hard tissue, with relatively little or no capacity for physiologic resorption, is incapable of combat- ing,¹¹⁹ thus, resulting in chronic osteomyelitis. This is by far the most common complication,¹'⁶¹⁹ and probably the source of the confusion of this lesion with primary osteomyelitis and the errone- ous names which describe an inflammatory process¹⁷'¹⁸.

Radiographically, there is often multiquadrant involvement²⁰ by mixed radiolucent-radiopaque masses, referred to by some as a "pagetoid appearance"¹. These masses are sometimes surrounded by cystic radiolucencies,¹'⁶'¹⁸¹⁹ which may also be seen as isolated entities, forming the previously mentioned simple bone cysts seen in some cases. If the lesional area communicates with the oral cav- ity, the margins at the exposure site(s) may appear irregular.⁶ Sequestra may form as well.²⁰

Histopathologically, the lesions are usually found to consist of a bland fibrous matrix with globules, trabeculae or large masses of mineralized tissue which are argued to be bone, cementum or both [Figs. 3 and 4].¹'⁶

The importance of radiological interpretation as an adjunct to diagnosis cannot be over emphasized.²¹²⁶ We are of the opinion that his- topathological distinction between this condition and other fibro-osseous lesions is not always reli- able, nor is the distinction between cementum and bone.²¹ Waldron et al are of the opinion that bone and cementum could be differentiated on the basis of their polarized light microscopy features.⁶ We agree to considering the sclerotic masses as repre- sentative of bone, cementum, or a combination of both, which depends on the way the responsible mesenchymal progenitor cells differentiate.²⁰ Hence, we prefer the term florid cemento-osseous dysplasia, which was used by Waldron in 1985, although he insisted that the lesional tissue was more compatible with cementum than bone.²⁷ Even if it were cementum only, radiographs would still be necessary to differentiate this florid condition from localized ones, such as periapical cemen- tal dysplasia. Furthermore, identical mineralizations may be found in fibro-osseous lesions affect- ing bones other than the jaws.¹

It is important that the correct diagnosis of florid cemento-osseous dysplasia be established in order that conservative treatment be initiated, thereby avoiding the cumbersome complication of chronic osteomyelitis. The condition can be monitored through routine patient recall visits, with surgical intervention only to avoid secondary infection through apical lesions or decubitus ulcers, and there is no need for overtreatment of the condition, such as by attempting to excise such diffusely distri- buted lesions.¹'⁶'¹⁸²⁰²⁷ The process is benign, and there is only one report in the literature of osteosarcoma of the mandible, which developed in a patient after irradiation of the area.²³

In our case, the patient did not develop complications after surgical intervention. Had her periodontitis been controlled at an earlier stage and the nature of the lesions recognized earlier, surgery would not have been the treatment of choice. In our experience with some of the cases referred to our surgical oral pathology service, this condition is unfortunately only recognized after surgical intervention and after osteomyelitis has developed.