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J Oral Maxillofac Surg 60:216-218, 2002

Ameloblastic Fibroma: Report of a Case


Su-Gwan Kim, DDS, PhD,* and Hyun-Seon Jang, DDS, PhD
Ameloblastic broma (AF) is a relatively rare, slowgrowing, benign odontogenic tumor. It is usually asymptomatic except for the eventual expansion of the jaw.1-3 AF is most common in adolescents and young adults, and is generally found in the mandible. This report describes a 3-year-old girl with AF in the mandible. The differential diagnosis, histology, and treatment are discussed.
vimentin staining was observed in some areas of the immature, dental papilla-like cells and in the basement membrane of the odontogenic epithelium (Figs 3, 4). No tumor was found in the tissue curetted from the surrounding bone. The nal diagnosis was ameloblastic broma. Different areas of the specimen showed different histologic ndings (Figs 5, 6). No calcied dental structures were apparent in the specimen. Areas of mesenchymal tissue without epithelial strands were present, and large areas devoid of ameloblastic strands and dental papilla were also seen (Fig 5). Mesenchymal growth appeared to be more active in areas devoid of epithelium (Fig 6). The postoperative course was uneventful and the patient was discharged for further follow-up. Healing was uneventful and there were no signs of recurrence 22 months after surgery (Fig 7).

Report of a Case
A 3-year-old girl was referred to our hospital on January 22, 1999 after a swelling developed in the right cheek a few days after trauma to that area. Her medical, surgical, family, and social histories were unremarkable. A systemic review was within normal limits, and no medication had been administered. Intraoral examination showed a hard swelling in the right mandibular body. A panoramic radiograph showed a large, multilocular, radiolucent lesion with scalloped margins. It extended from the right rst deciduous molar area to the ascending ramus and coronoid process (Fig 1). The radiograph also revealed expansion of the cortex and resorption of the roots of the lower right second deciduous molar. A displaced, developing permanent rst molar was associated with the lesion. The clinical diagnosis was dentigerous cyst. Enucleation of the lesion, extraction of the lower right permanent rst molar crown, and open packing were performed under general anesthesia. During the surgery, the lesion was found to be encased in bone. It measured 4 3 16 cm, was dark brown, and was soft in consistency. It was removed in pieces and the surrounding bone was curetted. The inferior alveolar nerve was preserved during the procedure and the patient retained normal sensation. Furacin gauze was used as the packing material. Packing was continued for 30 days and the gauze was changed daily. Microscopic examination of the tissue showed the presence of dental lamina-like strands, cords, and nests of odontogenic epithelium within a dental papilla-like myxomatous mesenchymal matrix (Fig 2). Positive cytokeratin staining was seen in the odontogenic epithelium, while positive

Discussion
AF is a relatively rare, benign neoplasm of the group of mixed odontogenic tumors. It represents only 2% of odontogenic tumors.4 It is characterized by the simultaneous proliferation of epithelial and mesenchymal tissue, without the formation of dentin or enamel.5 AF is a true mixed tumor in which both the epithelial and ectomesenchymal elements are neoplastic. There are 2 rare variants: granular cell AF and peripheral AF.6 The precise etiology of AF is not known. However, it is believed to arise de novo during a particular stage of odontogenesis, possibly as a result of overzealous elaboration of the basal lamina without further odontogenic differentiation.7 Trodahl8 reported that in 58% of cases the presenting symptom is swelling. Moreover, there is no significant gender difference in the frequency of AF or race predilection.8 It has been reported as occurring at ages ranging from 6 months to 42 years, with an average from 14.6 to 15.5 years. The youngest patient was a 7-week-old infant reported recently.9,10 Over 80% of these tumors occur in the mandible, usually in the canine-molar region.11,12 Only 4 tumors have been reported in the anterior maxillary region.13 AF exhibits somewhat slower growth than ameloblastoma, and does not tend to inltrate. Instead, it enlarges by gradual expansion so that the periphery of the lesion often remains smooth. The tumors frequently go unnoticed by patients, and are discovered accidentally during radiographic examination. Pain, tenderness, or mild swelling of the jaw may prompt the patient to seek relief from a dentist. In the case presented, the chief complaint was swelling of the cheek. 216

Received from the College of Dentistry, Chosun University, KwangJu, Korea. *Assistant Professor, Department of Oral and Maxillofacial Surgery, Oral Biology Research Institute. Lecturer, Department of Oral Pathology. Address correspondence and reprint requests to Dr Kim: Department of Oral and Maxillofacial Surgery, College of Dentistry, Chosun University, 421, SeoSeogDong, DongKu, KwangJu City, Korea; e-mail: SGCKIM@mail.chosun.ac.kr
2002 American Association of Oral and Maxillofacial Surgeons

0278-2391/02/6002-0017$35.00/0 doi:10.1053/joms.2002.29829

KIM AND JANG

217

FIGURE 1. A panoramic radiograph showing a multilocular osteolytic lesion in the mandibular body and ramus.

FIGURE 2. Dental lamina-like strands, cords, and nests of odontogenic epithelium are seen within a dental papilla-like myxomatous mesenchymal matrix (hematoxylin-eosin stain, original magnication 200).

FIGURE 4. Positive vimentin staining is observed in some immature dental papilla-like cells as well as in the basement membrane of the odontogenic epithelium (original magnication 200).

FIGURE 3. Positive cytokeratin staining is observed in the odontogenic epithelium (original magnication 200).

FIGURE 5. Large areas devoid of ameloblastic strands and dental papilla are seen (hematoxylin-eosin stain, original magnication 40).

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AMELOBLASTIC FIBROMA: REPORT OF A CASE

FIGURE 6. Active mesenchymal growth is present (hematoxylin-eosin stain, original magnication 100).

Radiographically, AF usually appears as a multilocular radiolucency with sclerotic margins. It typically ranges from 1 to 8 cm in diameter.1 Smaller tumors may appear unilocular, whereas larger ones may extend through the cortices of the bone.1 In the case presented, a multilocular osteolytic image was observed. Histologically, both the epithelial and connective tissue components of AF are neoplastic. The epithelial component is usually in the form of strands and islands, and often has a peripheral layer of cuboidal or columnar cells. The central area resembles the stellate reticulum of the embryonic enamel organ. Mitotic activity is uncommon. In fact, if mitotic activity is readily apparent, particularly in the mesenchymal element, the pathologist should consider the possibility of malignant transformation. The connective tissue component is much more cellular than in ameloblastoma. The cells mimicking the dental papilla or primitive pulp tissue are round or angular, and there is little surrounding collagen. The degree of cellularity

varies within the same tumor and between tumors. A denite capsule is considered an unusual feature of AF. Various immunohistochemical markers of intra and extracellular proteins have been studied in AF and other benign odontogenic mixed tumors. Proliferating cell nuclear antigen and MIB-1 are proteins recently well-recognized in relation to cellular proliferating activity.14 Evaluation of the growth potential in AF and related lesions could be of help in understanding tumor aggressiveness and in selecting appropriate surgical procedures. In the present case, immunohistochemical staining for cytokeratin and vimentin was performed and the tumor cells stained positively. The differential diagnosis includes ameloblastic brosarcoma.15 AF is treated by enucleation and curettage of the surrounding bone and removal of the affected teeth.16 Although recurrence of AF is rare, long-term follow-up is recommended.4 Recurrence is a result of conservative treatment with regrowth of residual tumor, often aided by an attempt to retain involved teeth.

References
1. Cawson RA, Binnie WH, Speight PM, et al: Lucass Pathology of Tumors of the Oral Tissues (ed 5). Hong Kong, Churchill Livingstone, 1998, pp 65-69 2. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pathology (ed 4). Philadelphia, PA, Saunders, 1983, pp 304-306 3. Regezi JA, Sciubba JJ: Oral Pathology: Clinical-Pathologic Correlations. Philadelphia, PA, Saunders, 1989, pp 393-394 4. Blankestijn J, Panders AK, Wymenga JP: Ameloblastic broma of the mandible. Br J Oral Maxillofac Surg 24:417, 1986 5. Regezi JA, Kerr DA, Courtney RM: Odontogenic tumours:Analysis of 706 cases. J Oral Surg 36:771, 1978 6. Takeda Y: Ameloblastic broma and related lesions: Current pathologic concept. Oral Oncol 35:535, 1999 7. Eversole LE, Tomich CE, Cherrick HM: Histogenesis of odontogenic tumors. Oral Surg 32:569, 1971 8. Trodahl JN: Ameloblastic broma: A survey of cases from the Armed Forces Institute of Pathology. Oral Surg 33:547, 1972 9. Slootweg PJ: An analysis of the interrelationships of the mixed odontogenic tumors: Ameloblastic broma, ameloblastic broodontoma and the odontomas. Oral Surg 51:266, 1981 10. Mosby EL, Russel D, Noren S, et al: Ameloblastic broma in a 7-week-old infant: A case report and review of the literature. J Oral Maxillofac Surg 56:368, 1998 11. Zallen RD, Preskar MH, McClary SA: Ameloblastic broma. J Oral Maxillofac Surg 40:513, 1982 12. Young AH: Ameloblastic broma in an infant. J Oral Maxillofac Surg 43:289, 1985 13. Heringer WW: Ameloblastic broma in an anterior maxilla: Report of a case. J Dent Child 45:408, 1978 14. Yamamoto K, Yoneda K, Yamamoto T, et al: An immunohistochemical study of odontogenic mixed tumors. Oral Oncol 31:122, 1995 15. Leider AS, Nelson JF, Trodahl JN: Ameloblastic brosarcoma of the jaws. Oral Surg Oral Med Oral Pathol 33:559, 1972 16. Dallera P, Bertoni F, Marchetti C, et al: Ameloblastic broma: A follow-up of six cases. Int J Oral Maxillofac Surg 25:199, 1996

FIGURE 7. A postoperative panoramic radiograph showing no evidence of recurrence 22 months after surgery.