Cemento-ossifying fibroma-a rare case

[Downloaded free from http://www.jisppd.com on Friday, March 06, 2015, IP: 117.199.204.69] || Click here to download free Android application for this journal ISSN 0970 - 4388 Cemento-ossifying fibroma—a rare case SARWAR H. G.1, JINDAL M. K.2, AHMAD S. S.3 Abstract The concept of ‘fibro-osseous lesions’ of bone has evolved over the last several decades and now includes two major entities: fibrous dysplasia and ossifying fibroma, as well as the other less common lesions such as florid osseous dysplasia, periapical dysplasia, focal sclerosing osteomyelitis, proliferative periostitis of garrie, and ostitis deformans. The cemento-ossifying fibroma is a central neoplasm of bone as well as periodontium which has caused considerable controversy because of confusion regarding terminology and the criteria for its diagnosis. The cemento-ossifying fibroma is odontogenic in origin, whereas ossifying fibroma is of bony origin. This article reports a rare case of an 11-year-old male who came to us with the history of swelling in the maxillary anterior region causing difficulty in closing of mouth as well as in mastication. Keywords: Cemento-ossifying fibroma, fibro-osseous lesions, maxilla, odontogenic tumor, ossifying fibroma Roentgenographic features In the early stages, the cemento-ossifying fibroma appears as a radiolucent lesion with no evidence of internal radiopacities. As the tumor matures, there is increasing calcification so that the radiolucent area becomes flecked with opacities until ultimately the lesion appears as an extremely radiopaque mass. Displacement of adjacent teeth is common. One additional important diagnostic feature is that there is a centrifugal growth pattern rather than a linear one and therefore the lesions grow by expansion equally in all directions and present as a round tumor mass. Cemento-ossifying fibroma is a fibro-osseous lesion that arises from the periodontal membrane.[1] The periodontal membrane is a layer of fibrous connective tissue surrounding the roots of teeth. It contains multipotential cells that are capable of forming cementum, lamellar bone, and fibrous tissue.[2,3] Although central cemento-ossifying fibromas of the mandible are common, they are unusual in the maxilla.[4] In 1872, Menzel gave the first description of a variant of ossifying fibroma, calling it a cemento-ossifying fibroma, in a 35-year-old woman with a long-standing large tumor of the mandible.[5] Histologic features Histology reveals many delicate interlacing collagen fibers, seldom arranged in discrete bundles, interspersed with large numbers of active, proliferating fibroblasts and cementoblasts. As the lesion matures, the islands of cementum increase in number, enlarge, and ultimately coalesce. This, along with the increase in degree of calcification, accounts for the increase in the radioopaqueness of the lesions on the roentgenograms. This lesion should be distinguished from fibrous dysplasia of bone and certain other fibro-osseous lesions that do not represent true neoplasia.[6] A close histogenetic relationship exists between the central cemento-ossifying fibroma and the central ossifying fibroma. It is based on the marked similarity between the two regarding predilection of age of occurrence, sex, race, location, roentgenographic appearance, and clinical behavior, these two lesions represent the same basic neoplastic process. The only difference between the two being in the type of cell involved and its end product—cementum in one case and bone in the other. Treatment and prognosis The lesion, which is sharply circumscribed and demarcated from bone, should be excised conservatively. Recurrence is rare. Clinical features This tumor generally occurs in young and middle-aged adults. There is a marked predilection for the female sex, the female: male ratio being 2:1. The mandible is more commonly involved than the maxilla. The lesion is generally asymptomatic until the growth produces a noticeable swelling and mild deformity; displacement of teeth may be an early clinical feature. Case Report An 11-year-old male child reported to the outpatient department of Pedodontics and Preventive Dentistry, Faculty of Medicine, complaining of inability to close the mouth and inability to chew food since 6–8 months [Figure 1]. The patient was apparently asymptomatic 18 months back, when he developed a small swelling in the maxillary anterior labial region, which gradually increased in size. He had initially approached a physician who had suggested a dental consultation; it was following this that the patient came to us with the same complaint. 1 Pediatric Maxillofacial Consultant, Department of Oral & Maxillofacial Surgery, 2Reader & Head, Department of Pedodontics & Preventive Dentistry, 3Prof. & Consultant Histopathologist, Department of Pathology, Faculty of Medicine, Z. A. Dental College, A.M.U. Aligarh-202 002, India. J Indian Soc Pedod Prevent Dent - September 2008 128 [Downloaded free from http://www.jisppd.com on Friday, March 06, 2015, IP: 117.199.204.69] || Click here to download free Android application for this journal Cemento-ossifying fibroma On examination, a uniform rounded swelling was present in the maxillary anterior region [Figure 1] due to which patient could not close his mouth. The size of the lesion was 3 × 3 cm and the shape was ovoid. The overlying mucosa was pinkish in color and firm in consistency. The texture was smooth; there was no compressibility or depressibility. Routine blood examination was done and showed Hb: 11 gm%; TLC: 12900/mm3; DLC: neutrophils 65%, lymphocytes 27%, eosinophils 8%, and basophils and monocytes 0%. The erythrocyte sedimentation rate was 24 mm at 1 h, bleeding time was 2.55 min, and clotting time 6.35 min. fibroma, and cementifying fibroma.[1] In the maxilla the clinical and radiological differential diagnoses include fibrous dysplasia, giant cell lesions, calcifying epithelial odontogenic tumor (Pindborg tumor),[7] and peripheral giant cell granuloma. In fibrous dysplasia, an intimate continuity between the lesion and normal bone is generally found. The tumor expands throughout its length and the margins are diffuse and radiographically poorly defined. Its radiological structure is more homogenous than that of ossifying fibroma or cementoossifying fibroma, both of which are filled with radiopaque foci. Cemento-ossifying fibroma is a well-circumscribed tumor that grows expansively and has clearly defined margins. Lesions are oval, spherical, or multilocular and are clearly separated from the surrounding bone by osteolytic borders.[2] Radiographs An occlusal view of the maxilla [Figure 2] was advised, and the radiograph revealed a well-defined radiolucent area present between the permanent maxillary central incisors, with displacement of incisors posteriorly and to either side towards the canines, with the result that the permanent maxillary central incisors were located at the usual position of the permanent canines. The left anterior region was flecked with multiple small radiopacities. Central cemento-ossifying fibromas are typically well-defined, solitary radiolucencies with scattered radiopaque foci. They maintain a spherical shape, expand cortical bone without causing perforation, and may cause tooth divergence.[7] Management Albendazole, 400 mg, was advised 1 week before surgery to settle down the count of eosinophils. Excisional biopsy was planned and a preoperative antibiotic (amoxicillin, 250 mg TDS) was advised for 3 days. Surgical excision [Figures 3 and 4] under local anesthesia was done, followed by primary closure of the wound with 3-0 silk sutures. The excised mass was sent to the pathology department for this to pathological examination. Antibiotics and anti-inflammatory agents were continued for the next 1 week. Alternate sutures were removed on the fifth postoperative day and the remaining sutures on the seventh postoperative day. At this time the lesion showed signs of regression and the patient was quite satisfied because he could eat food without any difficulty. Re-examination of the patient was done after 15 days and showed that the lesion had regressed [Figure 5] and there was a normal edentulous ridge. The patient was referred to the department of orthodontics for expert management of the flaring of the permanent maxillary anterior teeth that had been caused by the lesion. The neoplasm presents an extremely variable roentgenographic appearance, depending upon its stage of development. However, despite the stage of development, the lesion is always well circumscribed and well demarcated from surrounding bone, in contrast to true fibrous dysplasia. One additional important diagnostic feature of the lesion is its effect upon the inferior border of the mandible when the lesion reaches such a size as to encroach upon it. The central cementifying fibroma and its related lesions, the central ossifying fibroma and central cemento-ossifying fibroma, have a centrifugal growth pattern rather than a linear one. Therefore these lesions grow by expansion equally in all directions and present as round tumor masses. Other lesions (fibrous dysplasia) expand the cortex linearly and the outline of the expanded mandible is not in continuity with the remainder of the outline of the lesion. Peripheral giant cell granuloma may also resemble the cemento-ossifying fibroma clinically, with both types of lesions commonly occurring anterior to the molars and presenting as pedunculated or sessile masses. However, peripheral giant cell granuloma can be ruled out on the basis of the size of the lesion (0.5–1.5 cm), its pedunculated nature, and the presence of peripheral cuffing and giant cells on histopathological examination. Peripheral cuffing, due to superficial erosion of the bone, is the pathognomic radiographic feature. Another lesion which also clinically resembles the cemento-ossifying fibroma is the calcifying epithelial odontogenic tumor. The age of occurrence of this tumor is 8–92 years with a mean age of 40 years, whereas cemento-ossifying fibroma is generally not seen in middle age. In calcifying epithelial odontogenic tumors, the common site of occurrence is the molar area, whereas the cemento- Histopathological examination Histopathological examination showed [Figure 6] pseudoepitheliomatous hyperplasia of stratified squamous epithelium. At one edge there was an ulcer with acute inflammation. The deeper areas showed intense fibroblastic proliferation with mononuclear cell infiltrate; cementum and bony trabeculae were rimmed by fibroblasts. Discussion Cemento-ossifying fibroma of the maxilla is an uncommon tumor. Lesions with fibrous and osseous components include fibrous dysplasia, ossifying fibroma, cemento-ossifying 129 J Indian Soc Pedod Prevent Dent - September 2008 [Downloaded free from http://www.jisppd.com on Friday, March 06, 2015, IP: 117.199.204.69] || Click here to download free Android application for this journal Cemento-ossifying fibroma Figure 1: Frontal view of the patient Figure 4: Tumor mass immediately after excision Figure 2: Occlusal view of maxilla shows flecked radiopacities and flaring of the anterior teeth Figure 5: Regressed lesion with normal edentulous ridge 2 weeks after surgery Figure 3: Surgical area after excision Figure 6: Histopathological examination of the lesion ossifying fibroma is generally seen anterior to the molars. Calcifying epithelial odontogenic tumor is also generally associated with an unerupted or impacted tooth. Initial radiographic features of both the lesions are common (both being radiolucent) but as the lesion progresses it takes on a ‘honeycomb’ or ‘driven snow’ appearance. Histologically it can be differentiated by the presence of polyhedral epithelial cells as well as multinucleated giant cells. J Indian Soc Pedod Prevent Dent - September 2008 130 [Downloaded free from http://www.jisppd.com on Friday, March 06, 2015, IP: 117.199.204.69] || Click here to download free Android application for this journal Cemento-ossifying fibroma Cemento-ossif ying fibroma is a slow-growing lesion composed of cellular fibroblastic tissue containing basophilic masses of cementum-like tissue. In addition, varying amounts of bony trabeculae are interspersed within the lesion, giving it its characteristic features.[8] Reed[9] used the presence or absence of woven and lamellar bone in histopathological section to differentiate the cemento-ossifying fibroma from the other fibro-osseous lesions. In uncomplicated cases, fibrous dysplasia contains no lamellar bone but, rather, has arrested woven bone. On the other hand cemento-ossifying fibroma and ossifying fibroma contain woven bone and are often rimmed by osteoblasts that have laid down layers of lamellar bone. Additionally cemento-ossifying fibroma may have areas of cementum, appearing as psammoma bodies embedded in a benign fibrous stroma. Spiet[10] et al. supported Reed’s classification and stated that the predominant pattern of bone architecture is usually apparent and allows one to differentiate between the two types of lesions. C. Juvenile aggressive cement-ossifying fibroma: Case report and review of the literature. Laryngoscope 1993;103:1385-90. 3. Hamner JE 3 rd, Lightbody PM, Ketcham AS, Swerdlow H. Cemento-ossifying fibroma of the maxilla. Oral Surg Oral Med Oral Pathol 1968;26:579-87. 4. Sarita M, Raj KA, Daya SM, Rohtas KY. Cemento-ossifying Fibroma of the Maxilla. Indian J Radiol Image 2000;10:103-4. 5. Hamner JE 3rd, Scofield HH, Cornyn J. Benign fibro-osseous jaw lesions of periodontal membrane origin: An analysis of 249 cases. Cancer 1968;22:861-78. 6. Waldron CA, Giansanti JS. Benign fibro-osseous lesions of the jaw: A clinical-radiologic-histologic review of 65 cases. Oral Surg Oral Med Oral Pathol 1973;35:190-201. 7. Montgomery AH. Ossifying fibroma of the jaw. Arch Surg 1938;36:874-98. 8. Kuta AJ, Worley CM, Kaugars GE. Central Cemento-ossifying fibroma of the Maxillary sinus: A review of six cases. AJNR Am J Neuroradiol 1995;16:1282-6. 9. Reed RJ. Fibrous dysplasia of bone: A review of 25 cases. Arch Pathol 1963;75:480-95. 10. Spiet HJ, Dorfman HD, Fetchner RE, Ackerman LV. Tumors of bone and cartilage in atlas of tumar pathology. Armed Forces Institute of Pathology; section II; face V, 1971. References 1. 2. Reprint request to: Dr. Ghulam Hashmi Sarwar, C-32 Medical Colony, Aligarh Muslim University, Aligarh-202 002, India. E-mail: [email protected] Huebner GR, Brenneise CV, Ballenger J. Central ossifying fibroma of the anterior Maxilla: Report of case. J Am Dent Assoc 1988;116:507-10. Bertrand B, Eloy P, Cornelis JP, Gosseye S, Clotuche J, Gilliard 131 View publication stats J Indian Soc Pedod Prevent Dent - September 2008