Ameloblastic Fibro-Odontoma in Maxilla and Its Literature Review

Size: px

Start display at page:

Download "Ameloblastic Fibro-Odontoma in Maxilla and Its Literature Review"

Todd Edwards
5 years ago
Views:

1 Kor J Oral Maxillofac Pathol 2012;36(6): 상악에서발생한법랑모세포섬유치아종 박원홍 1), 임원봉 1), 김지선 1), 김상우 1), Zheng Hui 1), Kou Ni 1), 전상미 1), 박지일 2), 최홍란 1), 김옥준 1) * <Abstract> 전남대학교치의학전문대학원구강병리학교실 1), 광주보건대학교치위생과 2) Ameloblastic Fibro-Odontoma in Maxilla and Its Literature Review Won Hong Park 1), Won Bong Lim 1), Ji Sun Kim 1), Sang Woo Kim 1), Hui Zheng 1), Ni Kou 1), Sang Mi Jeon 1), Ji Il Park 2), Hong Ran Choi 1), Ok Joon Kim 1) * These first two authors, WonHong Park and WonBong Lim contributed equally to this work Dental Science Research Institute and Medical Research Center for Biomineralization Disorders, School of Dentistry, Chonnam National University, 1) Department of Dental Hygiene, Gwangju Health University 2) Ameloblastic fibro-odontoma has been defined as a lesion similar to ameloblastic fibroma by WHO, as it shows inductive changes which forms enamel and dentin. Ameloblastic fibro-odontoma is a very rare mixed dentition tumor in children, and the symptom shows indolent edema in maxillary and mandibular molar area. The prevalence is two times higher in male than in female, and two times higher in maxilla than in. Radiologically, it shows clear border and characteristics of both fibroma and odontoma histologically. This review reports a case that a 4-year old female visited in dental clinic of this school for edema as chief complaint in Feb, Emergency surgical curettage was performed right after initial diagnosis as odontoma, then confirmed diagnosis as Ameloblastic fibro-odontoma after biopsy. Currently, after 6 month, no sign of can be seen. Ameloblastic fibro-odontoma is very rare mixed dentition tumor. Moreover, as it is the case of female maxilla, this case is worth of publishing. Furthermore, accurate diagnosis of Ameloblastic fibro-odontoma is difficult. This review is published for accurate diagnosis through differential diagnosis of several important mixed dentition tumors. Key words:ameloblastic Fibro-Odontoma, Maxilla, Oral Pathology Ⅰ. Introduction Ameloblastic fibro-odontoma (AFO) has recently been described by the World Health Organization (WHO) as a lesion similar to ameloblastic fibroma but also showing inductive changes that lead to the formation of both dentin * Correspondence : Ok Joon Kim, Department of Oral Pathology, College of Dentistry, Chonnam National University, Bug-Gu, Gwangju, , Korea. Tel: , Fax: , js3894@chonnam.ac.kr * Acknowledgements: This work was supported by the Korea Research Foundation Grant funded by the Korean Government (MOEHRD) (KRF ). Received: Nov 7, 2012; Revised: Nov 9, 2012; Accepted: Nov 16, 2012 and enamel 10). It is a rare mixed odontogenic tumor. It was first described by Hooker in 1967 and it is usually diagnosed in the first two decades, 98.8% of the cases occurring before the age of 20 years 11). The average age was 9-years, and the male:female ratio was 2:1. Clinically, the tumor is painless and slow growth, with tumor expansion being observed. Most tumors are associated with the crown of an unerupted tooth, a fact that guides the diagnosis of the lesion. The size of the tumor shows marked variation, ranging from lesions detectable only microscopically to giant tumors consisting of extensive calcified masses 6). Radiographs show a well-defined radiolucent area containing various

Hard structures are composed of dentine-like material or osteodentine in addition to enamel matrix 1). The treatment of this lesion is conservative enucleation and curettage.

2 amounts of radiopaque material of irregular sizes and forms with density similar to dental hard tissues. Histopathologically, AFO is composed of strands, cords and islands of odontogenic epithelium in a primitive ectomesenchyme. Hard structures are composed of dentine-like material or osteodentine in addition to enamel matrix 1). The treatment of this lesion is conservative enucleation and curettage. The prognosis for this lesion is excellent and is rare. The aim of this paper is to report a AFO in 4-year old girl. Radiographic and histological features, in addtion, are discussed with english written review. Ⅱ. Case report A 4-year-old girl was referred to department of Oral & maxilofacial surgery (OMS) at Chonnam national university dental hospital for evaluation of mass located in the right posterior maxilla. Her first chief complain was for my gingiva is swelling. The patient initially was seen by her pedodontist for mass between #53 and #54 at palatal posterior area. The patient s medical history was unremarkable. She reported no pain, but swelling. Intraoral examination was significant for cortical expansion on palatal area and no pain on palpation. Extraoral exam had no significant states. It was measured by cm in the anteroposterior, transverse, and cephalocaudal dimensions, respectively. A panoramic radiography revealed an extensive, circumscribed, large mixed radiolucent lesion in the right posterior maxilla(fig. 1. A). Computerized tomography (CT) was performed to define the mass(fig. 1. B, C). The scan demonstrated a large hypodense expansile lesion with obliteration of maxillary Fig. 1. A. Initial panoramic radiograph shows a large mixed radiolucent lesion in the right posterior maxilla with near obliteration of the maxillary sinus. B, C. preoperative CT demonstrates proximity obliterarion of maxillary sinus. 348

sinus. The margins otherwise appeared sclerotic. The initial clinical diagnosis was complex odontoma. An excisional biopsy was performed under general anesthesia.

palisaded arrangement at the periphery and centrally a loose arrangement resembling the stellate reticulum of the enamel organ.

Amorphous eosinophilic material compatible with dentinoid material (Figs. 3-4), as well as basophilic material compatible with rudimentary enamel, was observed on various planes of the specimen (Fig.

Discussion and Literature review AFO is usually diagnosed in the first two decades, often found in the molar area.

3 sinus. The margins otherwise appeared sclerotic. The initial clinical diagnosis was complex odontoma. An excisional biopsy was performed under general anesthesia. Histopathologic examination showed a benign ectomesenchymal neoplasia of odontogenic origin characterized by the proliferation of islands, nests and cords of epithelial cells that exhibited a palisaded arrangement at the periphery and centrally a loose arrangement resembling the stellate reticulum of the enamel organ. Squamous metaplasia was observed at the periphery in some of these islands. The mesenchyme of the lesion was richly cellularized, containing spherical, stellate or spindle-shaped cells (Figs. 2-4). Amorphous eosinophilic material compatible with dentinoid material (Figs. 3-4), as well as basophilic material compatible with rudimentary enamel, was observed on various planes of the specimen (Fig. 5) Based on these characteristics, the histopathologic diagnosis was ameloblastic fibro-odontoma and the patient is currently under observation. Ⅲ. Discussion and Literature review AFO is usually diagnosed in the first two decades, often found in the molar area. The two most common clinical manifestations are failure of tooth eruption and swelling. It has recently been described in the WHO classification as Fig. 2. Proliferation of islands, cords, and strands of epithelial cells in the mesenchyme richly cellularized (H and E 200 ) Fig. 3. Amorphous eosinophilic material compatible with dentinoid mass (H and E 100 ) Fig. 4. Islands of epithelial cells that exhibit a palisaded arrangement at the periphery with reverse polarization (H and E 200 ) Fig. 5. Basophilic material compatible with rudimentary enamel (H and E 200 ) 349

4 a lesion similar to ameloblastic fibroma but also showing inductive changes that lead to the formation of both dentin and enamel. Some investigators believe that this entity represents an immature form of odontoma and therefore should be regarded as a rather than a true neoplastic process. In some cases, however, the tumor can undergo progressive growth, causing bone destruction and significant deformity. Such destructive lesions appear to be true neoplasms. Other lesions may simply represent a stage in the development of an odontoma. The tables below are results of cases published. One notable thing is that prevalence is high on young females Table 1. Literature review of Ameloblastic firo-odontoma author sex age location symptom radiographic histologic treatment follow-up ref Favia G.F. Friedrich R.E. Reichart P.A M 5 M 2 M 8 M 11 M 9 Hu Y. F 17 Silva G.C.C. Soares R.C. Pontes F.S.C. M 14 M 4 F 8 Mainenti P. F 12 Zouhary K.J. F 7 RIU G. M 27 Basili A. Pontes H.A.R. maxilla mandibullar angle mandibullar angle maxilla Rt. Rt. Ant. Maxilla anterior maxilla Rt. Maxilla maxilla painless painless 2.5*4cm wide well-defined radiolucency at ascending ramus wide radiolucency area to alveola crest well defined radiolucent radiolucenct dentine and enamel matrix enucleation no 5 dentine and enamel matrix enucleation no 5 excessive formation of extracellular matrix cementoid globules in fibrillar connective tissue storma enucleation/ 1st, 2nd surgery enucleation 1.3*1.5cm cyst-like radiolucenct enucleation 9*7*6cm/ pain painless unerupted anterior tooth unerupted tooth 2.7*3.2*3.6cm, delayed eruption of 1 st molar, painless asymptomatic CT: large soft tissue mass spindle-shaped fibroblastic matrix enucleation small well-defined unilocu;ar radiolucency extensive circumscribed unilocular radiolucent lesion enamel and dentine matrix formation enucleation 15 mo/ rapidossifying recurrent 3yr/no 3yr/no 18mo/no 2yrs/no islands of epithelial cell enucleation no 6 well circumscribed radiolucency enamel and dentine formation enucleation radiolucent CT: multiple dense calcification rounded smooth radiolucent lesion mixed tummor disclosing benign epithelial tissue irregular masses of dentine, osteodentine, and enamel matrix odontogenic epithlium in a loose primitive-appearing connentive tissue and a mineralized component enucleation 2yrs/no + anterior tooth eruption 23mo/no 12 enucleation no 10 enucleation 1yr/no F 6 Mandible multiple radioluceny lesion enucleation no 8 M 11 Mandible multiple radioluceny lesion enucleation no 8 M 9 M 6 F 13 M 11 Rt. Rt. firm painless swelling firm painless swelling firm painless swelling 5*3*2cm osseous cortical expasion enucleation no 11 enucleation no 11 irregullary radiolucency border enucleation no 11 cellular ectomesenchymal component with dental hard tissue enucleation no

5 and average age was 9.8±5.7. This patient was also 4 year old, which was in general range and occurred on posterior maxilla. However, the ratio of men and women was 2:1, and the ratio of and maxilla was 2:1, which means that the majority was male. The most frequently reported symptom was painless followed by unerupted tooth (teeth). Very rarely, facial asymmetry was reported due to enlarged AFO. The average size of mass was cm. All showed well-defined wall, and radiopacity and radiolucency were mixed. It histologically showed the feature of both fibroma (richly fibrous connective tissue, cord, and strands) and odontoma (dentinoid material, and rudimentary enamel). Recurrence rate was very low and an article reports that conservative curettage is needed but insufficient removal can lead to, which means that it is affected by skill of operator but accurate diagnosis is essential. Differential diagnosis of AFO should include lesions with mixed radiographic patterns, such as calcifying epithelial odontogenic tumour, calcifying odontogenic cyst (COC) and adenomatoid odontogenic tumour (AOT). AFO is defined as a tumor with the general histologic features of an ameloblastic fibroma and odontoma. The differential diagnosis of a mixed radiolucent radiopaque lesion in the jaws of a child or adolescent includes compound odontoma, COC, AOT, CEOT and AFO 13,14). Odontomas are hamartomas of odontogenic origin that present as 2 clinical variants: compound and complex. The complex odontoma is an amorphous calcified mass most often presenting in the posterior or maxilla in association with an unerupted third molar. The compound odontoma is composed of many small tooth-like structures and is surrounded by a dental follicle, and it is found more often in the anterior portions of the jaws. Odontomas comprise 65%-75% of odontogenic tumors. Histologically, odontomas display mature dental tissues in either a structured manner (compound odontoma) or in an unorganized mass (complex odontoma). Both variants are treated by enucleation and curettage, and is not reported 15). COC is a rare unilocular odontogenic cyst which displays features of both a cyst and solid neoplasm. It usually presents as an intraosseous lesion, although an extraosseous variant is also well documented. The central variant is observed from the first to the seventh decade but most commonly presents in the second decade. The COC has no gender predilection and is often encountered in the anterior maxilla and. Radiographically, it shows a mixed radiolucent/radiopaque lesion with variable amount of calcification which may cross the midline and infiltrate adjacent structures such as the maxillary sinus. Histologically, it resembles ameloblastoma with a basal epithelial lining and an overlying layer of stellate reticulum with the addition of pale staining ghost cells and intraepithelial calcifications. With complete enucleation, the COC has no reported 16). Some consider AOT to be a benign odontogenic tumor, and others consider to be a harmatomatous lesion. AOT is slow but progressive growth. This lesion is most often found in the second decade with a female predilection. The three variants, follicular, extrafollicular, and peripheral, are histologically identical. The follicular variant of AOT commonly presents as a unilocular radiolucency in the anterior maxilla and is often associated with the crown of an unerupted tooth. Radiopaque flecks are often present within the cystic space of this lesion 17). Microscopically, AOT contains nodules of columnar or cuboidal epithelial cells forming nests or rosette-like structures with eosinophilic material present. Cuboidal or columnar cells are arranged around empty spaces resembling ducts. Islands of polyhedral eosinophilic epithelial cells may contain masses of amorphous amyloid-like material as well as calcified material. Enucleation is curative, and there is no with this lesion 18). CEOT is a locally invasive benign tumor that has a wide 351

6 age distribution, from 8 to 92 years old, with the average age of detection in the fourth decade of life. It most commonly presents in the posterior as a slow growing painless mass. The lesion usually consists of a unilocular or multilocular radiolucency with radiopaque flecks of varying size and opacity but has poor-defined margin. Histologically, sheets of polyhedral epithelial cells with cytoplasmic eosinophilia and intracellular bridges which may display a degree of pleomorphism are seen. Liesgang ring like calcifications within the tumor and amyloidlike material within the sheets of tumor cells are typical of this tumor. This tumor has a significant rate with enucleation alone, and therefore resection with 1 cm margins is recommended 19). This lesion is generally treated by conservative enucleation and curettage. The prognosis for this lesion is excellent and is rare. The few reported s may be attributed to inadequate removal of the entire lesion. Therefore, it is essential to provide adequate access to ensure complete removal of the lesion, especially around the apices of erupted dentition. At the time of writing, the patient was 6 months past surgery and showed no clinical or radiographic signs of. This case demonstrates the necessity of having a well formed and comprehensive differential diagnosis to guide a successful and conservative surgical approach, even in a locally aggressive tumor. Ⅳ. References 1. Reichart PA, Philipsen HP, Gelderblom HR, Stratmann U: Ameloblastic fibro-odontoma report of two cases with ultrastructural study of tumour dental hard structures. Oral Oncology 2004;40: Hu Y, Liu B, Su T, Zhang W, Zhao Y: A huge ameloblastic fibro-odontoma of the maxilla. Oral Oncology 2005;42: Silva GCC, Jham BC, Silva EC, Horta MCR, Godinho SHP, Gomez RS: Ameloblastic fibro-odontoma. Oral Oncology 2006;42: Pontes FSC, Pontes HAR, Nogueira JES, et al: DS. Ameloblastic fibro-odontoma: Case report with maintenance of the involved teeth. Int J Pediatric Otorhinolaryngology 2008;3: Favia GF, di Alberti L, Scarano A, and Piattelli A: Ameloblastic Fibro-Odontoma: Report of two case. Oral Oncology 1997;33: Soares RC, Godoy GP, Neto JC, de Souza LB, Pinto LP: Ameloblastic fibro-odontoma: Report of a case presenting an unusual clinical course. Int J Pediatric Otorhinolaryngology 2006;1: de Riu G, Meloni SM, Contini M, Tullio A: Ameloblastic fibro-odontoma. Case report and review of the literature. J Cranio Maxillofac Surg 2010;38: Basili A, Almeida A, Cortes S, Alister JP, Martinovic G: Ameloblastic fibro-odontoma, report of two cases. Int J Oral Maxillofac Surg 2011;40: Mainenti P, Oliveira GS, Valério JB, et al: Ameloblastic fibroodontosarcoma: a case report. Int J Oral Maxillofac Surg. 2009;38: Zouhary KJ, Naief NS, Waite PD: Ameloblastic fibro-odontoma: expansile mixed radiolucent lesion in the posterior maxilla: a case report. 2008;106: Pontes HAR, Correa FS, Lameira AG, et al: Report of four cases of Ameloblastic fibro-odontoma in and discussion of the literature about the treatment. J Cranio Maxillofac Surg 2012;40: Friedrich RE, Siegert J, Donath K: Recurrent Ameloblastic Fibro-Odontoma in a 10-Year-Old Boy. J Oral Maxillofac Surg 2001;59: Silva GC, Jham BC, Silva EC, Horta MC, Gomez RS: Ameloblastic fibro-odontoma. Oral Oncology 2006;42: Louis PJ, Fugler RC, August M: Mixed radiolucent/radio- 352

7 paque lesion of the maxilla. J Oral Maxillofac Surg 2000;58: Yeung KH, Cheung RC, Tsang MM: Compound odontoma associated with an unerupted and dilacerated maxillary primary central incisor in a young patient. Int J Ped Dent 2003;13: Souza LN, Souza AC, Gomes CC, Loyola AM: Conservative treatment of calcifying odontogenic cyst : report of 3 cases. J Oral Maxillofac Surg 2007;65: Braunstein S: Adenomatoid odontogenic tumor of the : review of the literature and report of a rare case. Head Face Med 2005;24: Batra P, Prasad S, Parkash H: Adenomatoid odontogenic tumor: review and case report. J Can Dent Assoc 2005;71: Deboni M, Homem M, Junior D, Traina A: Clinical, radiological and histological features of calcifying epithelial odontogenic tumor: case report. Braz Dent J 2006;17: Handschel JG, Depprich RA, Zimmermann AC, 353

A Radiographic technique for differentiating enamel and dentin in odontogenic tumors

ISPUB.COM The Internet Journal of Radiology Volume 12 Number 1 A Radiographic technique for differentiating enamel and dentin in odontogenic tumors D Shetty, A Urs, R Kaur Citation D Shetty, A Urs, R Kaur.