Indian Journal of Dental ResearchIndian Journal of Dental ResearchIndian Journal of Dental Research
HOME | ABOUT US | EDITORIAL BOARD | AHEAD OF PRINT | CURRENT ISSUE | ARCHIVES | INSTRUCTIONS | SUBSCRIBE | ADVERTISE | CONTACT
Indian Journal of Dental Research   Login   |  Users online: 1822

Home Bookmark this page Print this page Email this page Small font sizeDefault font size Increase font size         

 


 
CASE REPORT Table of Contents   
Year : 2008  |  Volume : 19  |  Issue : 2  |  Page : 162-164
A massive ameloblastic fibro-odontoma of the maxilla


1 Department of Oral and Maxillofacial Pathology, SDM College of Dental Sciences, Dharwad, India
2 Department of Oral Pathology and Microbiology, Government Dental College, Bangalore, Karnataka, India

Click here for correspondence address and email

Date of Submission10-May-2007
Date of Decision20-Sep-2007
Date of Acceptance23-Sep-2007
 

   Abstract 

Ameloblastic fibro-odontoma is a rare, benign mixed odontogenic tumor. It occurs exclusively as an intraosseous lesion. It usually has a slow growth and is seen in children and young adults. A painless swelling is the most common clinical sign. Radiographically, ameloblastic fibro-odontoma appears as a circumscribed radiolucency which may contain radiopaque foci. Most cases of ameloblastic fibro-odontoma exhibit benign behavior, but cases of malignant transformation have been reported. The treatment modality in most cases involves conservative surgery, but cases with malignant transformation will require more radical treatment. A massive ameloblastic fibro-odontoma involving the maxilla has been described here with its clinical, radiological, and histopathological features.

Keywords: Ameloblastic fibro-odontoma, mixed odontogenic tumors

How to cite this article:
Hegde V, Hemavathy S. A massive ameloblastic fibro-odontoma of the maxilla. Indian J Dent Res 2008;19:162-4

How to cite this URL:
Hegde V, Hemavathy S. A massive ameloblastic fibro-odontoma of the maxilla. Indian J Dent Res [serial online] 2008 [cited 2019 Mar 21];19:162-4. Available from: http://www.ijdr.in/text.asp?2008/19/2/162/40474
Ameloblastic fibro-odontoma is a rare benign mixed odontogenic tumor. The term ameloblastic fibro-odontoma was first used by Hooker in 1967. [1] It is defined by the WHO as a neoplasm composed of proliferating odontogenic epithelium embedded in a cellular ectomesenchymal tissue that resembles dental papilla; there are varying degrees of inductive changes, resulting in dental hard tissue formation. [2]

It is necessary to differentiate this lesion from odontoameloblastoma (OA). This distinction is important because of the occasional reports of the aggressive and recurrent nature of OA. [3] Howell et al . have shown the potential for malignant transformation of ameloblastic fibro-odontoma to ameloblastic fibrosarcoma. [4] In this paper, we describe a very large ameloblastic fibro-odontoma affecting the maxilla in a 12-year-old girl.


   Case Report Top


A 12-year-old girl presented with 5 months history of a slowly enlarging, painless swelling in the maxilla [Figure - 1]. Her medical, family, and social history were unremarkable and so were the results of the physical examination. There was no history of trauma. Oral examination revealed a large lobulated lesion extending from the upper lateral incisor to the first molar region [Figure - 2]. Teeth from the upper lateral incisor to the molar were clinically missing. The swelling measured about 14 7 cm; there was obliteration of the labial and buccal vestibule, inferior displacement of the upper left central incisor, and incompetence of the lips. The lesion was reddish, nontender, and soft to firm in consistency. A panoramic radiograph [Figure - 3] revealed a large expansile radiolucent lesion, with the teeth pushed towards the periphery of the lesion. The patient was referred to our department for histopathological evaluation.

Histopathology [Figure - 4]: An incisional biopsy was done. The histopathological examination with hematoxylin and eosin staining showed islands of odontogenic epithelium, with peripheral columnar cells resembling ameloblasts and central stellate reticulum-like cells. The mesenchymal component was an embryonic fibrous connective tissue with delicate fibrils resembling dental papilla. Calcified structures resembling dysplastic dentin with enamel space were seen in close proximity to the epithelium. No findings suggestive of a malignant growth were observed. Based on the above findings, a histopathological diagnosis of ameloblastic fibro-odontoma was made. An immunohistochemical study was not possible in our setup.

The lesion was excised intraorally under general anaesthesia [Figure - 5] and partial dentures were given to the patient. Follow-up could not be done as the patient did not turn up later.


   Discussion Top


Odontogenic tumors are lesions that are derived from epithelium or mesenchymal elements or both. Ameloblastic fibro-odontoma is one such example, involving both the epithelial and mesenchymal elements, with inductive changes. [5] Ameloblastic fibro-odontoma is a rare tumor which represents only 1-3% of all odontogenic tumors. [6],[7],[8] This tumor has been confused earlier with other odontogenic mixed tumors. [9] It was identified by a variety of terms until Hooker distinguished it from ameloblastic odontoma and described it as a separate entity. [10] It is a slow growing lesion. [11] Most cases occur before 20 years of age, with the average age of diagnosis being 9 years. It occurs exclusively as an intraosseous tumor. The tumor is found more often in males, and the posterior mandible is affected 2.4 times more often than the entire maxilla. [12]

The two most common presenting complaints are swelling and failure of tooth eruption. [1],[11] The lesion may displace erupted teeth although the teeth remain vital. [3],[11] Radiographically, it usually appears as a radiolucent area that may contain variable amounts of calcified material. Microscopically, the lesion is composed of odontogenic epithelium embedded in a cell-rich primitive ectomesenchyme resembling dental papilla. In addition, there is dentin and enamel matrix formation. [1],[6],[11],[13]

In contrast, mild pain and delayed eruption of teeth are the common presenting features in OA, which is one of the differential diagnoses. OA is extremely rare. Histopathologically, OA is unusual in that a relatively undifferentiated neoplastic tissue is associated with a highly differentiated tissue, both of which may show recurrence after adequate removal. Many structures resembling normal or atypical tooth germs may be found, with or without the presence of calcified dental tissues. The presence of a typical ameloblastoma of one or the other recognized types is a characteristic feature. [14] Additionally, dendritic cells containing melanin pigment and fine granules and coarse aggregates of melanin distributed in the epithelial cells of OA have been occasionally described. A squamous differentiation, similar to that seen in the keratocyst and the calcifying odontogenic cyst, has also been observed. [15]

Ameloblastic fibro-odontoma is generally believed to be benign and its detailed histogenesis is uncertain. [16] Herzog et al . reported a case of a 14-year-old girl with an ameloblastic fibro-odontoma that evolved into an odontogenic sarcoma. Similarly, Howell et al . described two cases of malignant transformation of ameloblastic fibro-odontoma. [4] According to Howell et al ., the major criterion for determining whether malignant transformation is likely to occur is the recurrence of the tumor accompanied by a histologic change in the connective tissue toward a more cellular and unorganized pattern. [4]

The results of the cell kinetics of ameloblastic fibro-odontoma by bromhexidine and PCNA by Sekine et al . suggested that the ectomesenchymal component was more proliferative than the epithelial component. [16] Immunohistochemical (IHC) studies are very informative [17] and useful in the diagnosis of the lesion as this lesion occasionally transforms into an aggressive type. However, we could not do an IHC study as it was not possible in our setup and the patient could not afford it in other laboratories.

The recommended treatment for ameloblastic fibro-odontoma is conservative surgery with enucleation. The potential for malignant transformation does not justify radical treatment. [11] If there is a recurrence accompanied by change of the histologic pattern toward a more unorganized fibrous stroma, with displacement of the epithelial component, then more extensive treatment procedures appear to be indicated. [13] Though rare, malignant transformation can occur [4] and long-term follow-up is recommended. [18] A follow-up of only few months or years is generally inadequate to state that a definite cure has been achieved, as malignant changes can occur after 10-12 years also. [19] In our case, however, the follow-up could not be made as the patient did not turn up afterward.

In the present case, the tumor was massive, measuring 14 7 cm. The location was the anterior maxilla, in contrast to its usual site: the posterior mandible. It was intraosseous in origin, having perforated the buccal cortical plate to present as a soft tissue lesion, an indication of its aggressive nature. Histopathologically, our case showed only minimal amounts of calcified structure in spite of its large size and aggressive behavior.

 
   References Top

1.Sivapathasundharam B, Manikandan R, Sivakumar G, Goerge T. Ameloblastic fibro-odontoma. Indian J Dent Res 2005;16:19-21.  Back to cited text no. 1    
2.Barnes L, Eveson JW, Reihcart P, Sidransky D, editors. WHO international histological classification of tumors, Volume 9. IARC Press; 2005. p. 284.  Back to cited text no. 2    
3.Hutt PH. Ameloblastic fibro-odontoma: Report of a case. J Oral Maxillofac Surg 1982;40:45-8.  Back to cited text no. 3  [PUBMED]  
4.Howell RM, Burkes EJ. Malignant transformation of ameloblastic fibro-odontoma to ameloblastic fibrosarcoma. Oral Surg 1977;43:391-401.  Back to cited text no. 4    
5.Regezi JA, Sciubba JJ, Jordan RC. Oral pathology, 3 rd ed, Saunders Company Ltd: Philadelphia, USA; 2003.  Back to cited text no. 5    
6.Furst I, Pharoah M, Phillips J. Recurrence of a Ameloblastic fibro-odontoma in a nine year old boy. J Oral Maxillofac Surg 1999;57:620-3.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]
7.Philipsen HP, Thosaporn W, Reichart P, Grundt G. Odontogenic lesions in opercula of permanent molars delayed in eruption. J Oral Pathol Med 1992;21:38-41.  Back to cited text no. 7  [PUBMED]  
8.Wu PC, Chang K. A survey of tumors of the jaw bones in Hong Kong Chinese 1963-1982. Br J Oral Maxillofac Surg 1985;23:92-102.  Back to cited text no. 8    
9.Takeda Y. Ameloblastic fibroma and related lesions. Oral Oncol 1999;35:535-40.  Back to cited text no. 9  [PUBMED]  [FULLTEXT]
10.Piette EM, Tideman H, Wu PC. Massive maxillary ameloblastic fibro-odontoma: Case report with surgical management. J Oral Maxillofac Surg 1990;48:526-30.  Back to cited text no. 10  [PUBMED]  
11.Chang H, Shimizu M, Precious DS. Ameloblastic fibro-odontoma: A case report. J Can Dent Assoc 2002;68:243-6.  Back to cited text no. 11    
12.Reichart PA, Philipsen OH. Odontogenic tumours and allied lesions, 1 st ed. Quintessence Publication: USA; 2004.  Back to cited text no. 12    
13.Oghli AA, Scuto I, Ziegler C, Flechtenmacher C, Hofele C. A large ameloblastic fibro-odontoma of the right mandible. Med Oral Patol Cir Bucal 2007;12:E34-7.  Back to cited text no. 13    
14.Shafer, Hine, Levi. Cysts and tumours of odontogenic origin. In : Textbook of oral pathology, 4 th ed. WB Saunders Company: USA; 1993. p. 258-317.  Back to cited text no. 14    
15.Martin-Granizo-Lopez R, Lopez-Garcia-Asenjo J, de Pedro-Marina M, Domνnguez-Cuadrado L. Odontoameloblastoma: A case report and a review of the literature. Med Oral 2004;9:340-4.  Back to cited text no. 15    
16.Sekine J, Kitamura K, Ueno K, Sano K, Inokuchi T, Takahashi H, et al . Cell kinetics in mandibular ameloblastic fibroodontoma evaluated by bromodexyuridine and proliferating cell nuclear antigen immunohistochemistry: Case report. Br J Oral Maxillofac Surg 1996;:450-3.  Back to cited text no. 16    
17.Yamamoto K, Yoneda K, Yamamoto E, Osaki T. An immunohistochemical study of odontogenic mixed tumours. Oral Oncol Eur J Cancer Br 1995;31:122-8.  Back to cited text no. 17    
18.Silva GC, Jham BC, Silva EC, Horta MC, Godinho SH, Gomez RS. Ameloblastic fibro-odontoma. Oral Oncol Extra 2006;42:217-20.  Back to cited text no. 18    
19.Friedrich RE, Siegert J, Donath K, Jakel KT. Recurrent ameloblastic fibro-odontoma in a 10-year-old boy. J Oral Maxillofac Surg 2001;59:1362-6.  Back to cited text no. 19    

Top
Correspondence Address:
Veda Hegde
Department of Oral and Maxillofacial Pathology, SDM College of Dental Sciences, Dharwad
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.40474

Rights and Permissions


    Figures

  [Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4], [Figure - 5]

This article has been cited by
1 Adolescent Mandibular Central Odontoameloblastoma: A Rare Case Report
K. V. Arun Kumar,Apoorva Mowar,Rajat Gupta,D. Deepa
Journal of Maxillofacial and Oral Surgery. 2014;
[Pubmed] | [DOI]
2 Clinical and Radiological Profile of Ameloblastic Fibro-Odontoma: an Update on an Uncommon Odontogenic Tumor Based on a Critical Analysis of 114 Cases
Amos Buchner,Israel Kaffe,Marilena Vered
Head and Neck Pathology. 2013; 7(1): 54
[Pubmed] | [DOI]
3 Clinical and Radiological Profile of Ameloblastic Fibro-Odontoma: An Update on an Uncommon Odontogenic Tumor Based on a Critical Analysis of 114 Cases
Buchner, A. and Kaffe, I. and Vered, M.
Head and Neck Pathology. 2013; 7(1): 54-63
[Pubmed]
4 Odontoameloblastoma
Dive, A. and Khandekar, S. and Bodhade, A. and Dhobley, A.
Journal of Oral and Maxillofacial Pathology. 2011; 15(1): 60-64
[Pubmed]
5 Ameloblastic fibro-odontoma: A clinicopathologic study of 12 cases
Boxberger, N.R., Brannon, R.B., Fowler, C.B.
Journal of Clinical Pediatric Dentistry. 2011; 35(4): 397-404
[Pubmed]



 

Top
 
 
  Search
 
 
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Email Alert *
    Add to My List *
* Registration required (free)  
 


    Abstract
    Case Report
    Discussion
    References
    Article Figures

 Article Access Statistics
    Viewed4118    
    Printed128    
    Emailed5    
    PDF Downloaded595    
    Comments [Add]    
    Cited by others 5    

Recommend this journal