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Table of Contents   
SHORT COMMUNICATION  
Year : 2010  |  Volume : 21  |  Issue : 4  |  Page : 618-620
Ameloblastic fibro-odontoma of the anterior mandible in a 22-month-old boy


1 Department of Oral Surgery, UNMC, College of Medicine, Omaha, USA
2 Department of Oral Biology, UNMC, College of Dentistry, Lincoln, Nebraska, USA

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Date of Submission03-Feb-2009
Date of Decision08-Dec-2009
Date of Acceptance16-Jun-2010
Date of Web Publication24-Dec-2010
 

   Abstract 

We report an ameloblastic fibro-odontoma (AFO) presenting in the anterior mandible as a "bump on his gums" in a 22-month-old boy. An occlusal radiograph revealed a well-circumscribed radiolucency with scattered radiopaque foci. The tumor was enucleated under general anesthesia. The histologic findings were characteristic of an AFO, a mixed odontogenic tumor most common in the posterior jaws, primarily affecting individuals with an average age of 10 years. The clinical presentation, microscopic findings, differential diagnoses, and treatment are discussed.

Keywords: Ameloblastic fibro-odontoma, infant male, mixed odontogenic tumor

How to cite this article:
Wewel J, Narayana N. Ameloblastic fibro-odontoma of the anterior mandible in a 22-month-old boy. Indian J Dent Res 2010;21:618-20

How to cite this URL:
Wewel J, Narayana N. Ameloblastic fibro-odontoma of the anterior mandible in a 22-month-old boy. Indian J Dent Res [serial online] 2010 [cited 2014 Jul 28];21:618-20. Available from: http://www.ijdr.in/text.asp?2010/21/4/618/74237
Ameloblastic fibro-odontoma (AFO) is a mixed odontogenic tumor composed of both ectodermal and mesenchymal elements. The lesion presents as an asymptomatic swelling that usually delays the eruption of either primary or permanent teeth. The radiographic presentation is that of a well-circumscribed, mixed radiolucent-radiopaque lesion superior to the developing dentition. The lesion occurs in both males and females at an average age of 10 years in the posterior mandible, in most instances. [1],[2] Although most odontogenic neoplasms are benign with limited malignant potential, [1] they can be expansive and locally aggressive [3] requiring prompt attention. An ameloblastic fibro-odontoma is no exception; a few have the capability of progressive growth and bony destruction. [4] AFO is usually treated by enucleation and curettage without recurrence. [2] The purpose of this report is to document the unusual occurrence in a 22-month-old boy in the anterior mandible.


   Case Report Top


A 22-month-old male presented with a "bump on his gum" noticed by his parents. The oral examination revealed a swelling in the anterior mandible replacing the primary left mandibular canine (M) and lateral incisor (N). The involved area had a bluish hue, appeared to be fluid-filled and was firm to palpation. An occlusal radiograph revealed a radiolucent lesion with scattered radiopaque foci [Figure 1]. The lesion was well-circumscribed and coronal to teeth M and N, which were displaced apically. The primary left mandibular central incisor (O), right mandibular central incisor (P), and lateral incisor (Q) had erupted in proper sequence. The clinical impression was an odontogenic lesion with impacted primary mandibular left canine and lateral incisor.
Figure 1: (A) A well-defined coronal border of AFO. (B) Scattered radiopaque foci within radiolucency. Primary teeth M and N identified

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An excisional biopsy was performed under general anesthesia. The lesion was enucleated with curettage of the bony walls. The primary teeth M and N were identified, elevated, and removed. The crypt was cleaned with no obvious soft tissue remnants. The bone was smoothed and the excisional site was sutured. The lesion and primary teeth were submitted for pathologic examination. Microscopically, the lesion was composed of fragments of immature connective tissue reminiscent of dental papillae with cords and islands of cuboidal to columnar odontogenic epithelial cells [Figure 2] and [Figure 3]. Irregular masses of dental hard tissue composed of tubular dentin and enamel matrix were also seen [Figure 2] and [Figure 3]. These findings were characteristic of AFO as described in the literature.
Figure 2: H and E 10×: Ameloblastic fibro-odontoma exhibiting (1) odontogenic mesenchyme, (2) odontogenic epithelium with (3) hard tissue

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Figure 3: H and E 40×: Ameloblastic fibro-odontoma exhibiting (1) odontogenic epithelium with (2) hard tissue

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The postoperative course was uneventful. The last follow-up visit was approximately 1 year post-operative. The parents were informed of the low lesional recurrence rate, but were made aware of the possibility of a delayed eruption of permanent teeth. The patient is presently lost to follow-up.


   Discussion Top


An AFO is a mixed odontogenic neoplasm most commonly found in the posterior mandible and less likely (11%), in the anterior mandible. [1] Slootweg [5] reports that, although uncommonly found in the anterior mandible, an AFO occurring at this site tends to be in a younger patient with an average age of 3.3 years. AFO and odontoma are associated with unerupted teeth and are either unilocular or multilocular well-defined radiolucencies with multiple foci of radiopacity. [2] The present case supports Slootweg's [5] findings and documents the uniqueness of this lesion given the age of the patient and location of the neoplasm [Table 1].
Table 1: Demographics of AFO in children younger than 5 years


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The differential diagnosis in this case included other benign mixed lesions of the jaws, such as adenomatoid odontogenic tumor, odontoma, and calcifying odontogenic cyst.

An adenomatoid odontogenic tumor is limited to younger patients usually between the ages of 10 and 19 years with the anterior maxilla most commonly affected. In 75% of cases, the tumor appears as an unilocular radiolucency with scattered calcifications associated with the crown of an erupted tooth, most often the canine teeth. [1],[2] An odontoma is most common in the first 2 decades with the majority discovered when a tooth fails to erupt. A complex odontoma appears as a calcified mass consistent with the radiodensity of the tooth structure, usually surrounded by a radiolucent rim with well-defined corticated borders. [1] A calcifying odontogenic cyst is a mixed lesion of the jaws found in all age groups with a mean age of 33 years. The majority are found in the anterior jaws presenting as a unilocular radiolucency with calcifications consistent with tooth structure. One third of cases are associated with an unerupted tooth, usually the canine teeth. [1]

Histologically, the specimen in this case was characteristic of an AFO, which has the same features of an ameloblastic fibroma with the addition of odontogenic hard tissues. Some authors have argued that an ameloblastic fibroma, an AFO, and a complex odontoma represent differing stages of the same lesion. [1] Slootweg [5] challenges this theory by pointing out the difference in site predilection and most importantly, the lower mean age of AFO as compared with the ameloblastic fibroma. The opposite should be true if an ameloblastic fibroma represented an immature AFO.

The management of an AFO involves enucleation and curettage as most lesions easily separate from the surrounding bone. Since most lesions are coronal to an unerupted tooth, the deciduous tooth is usually extracted along with the tumor. When associated with a permanent tooth, the tumor is enucleated in most instances allowing the involved permanent tooth to erupt. Lesional recurrence is uncommon and transformation to an ameloblastic fibrosarcoma is exceedingly rare. [1],[2],[3],[4],[5],[6],[7],[8],[9]


   Conclusion Top


The youngest reported patients with AFO were a 9-month-old boy with a lesion in the anterior maxilla followed by a 12-month-old girl with a maxillary lesion. The present case report documents the previously unreported occurrence in the anterior mandible in a 22-month-old boy and supports the theory of Slootweg that amelobastic fibroma is not an immature stage of AFO.

 
   References Top

1.Damm N, Bouquot A. Oral and Maxillofacial Pathology. 3 nd ed. St Louis, MO Saunders Elsevier Inc; 2009.  Back to cited text no. 1
    
2.Takeda Y, Tomich CE. Ameloblastic fibro-odontoma. In: Barnes L, Evenson JW, Reichart P, Sidransky D, editors. Pathology and Genetics of head and neck tumors. Lyon: IARC Press; 2005. p. 309.  Back to cited text no. 2
    
3.Ozer E, Pabuccuoglu U, Gunbay U, Sarioglu S, Aktas S. Ameloblastic fibro-odontoma of the maxilla: Case report. J Clin Pediatr Dent 1997;21:329-31.  Back to cited text no. 3
[PUBMED]    
4.Flaitz C, Hicks J. Delayed tooth eruption associated with an ameloblastic fibro-odontoma. Pediatr Dent 2001;23:253-4.  Back to cited text no. 4
    
5.Slootweg PJ. An analysis of the interrelationship of the mixed odontogenic tumors ameloblastic fibroma, ameloblastic fibro-odontoma, and the odontomas. Oral Surg Oral Med Oral Pathol 1981;51:266-76.  Back to cited text no. 5
[PUBMED]    
6.Baker WR, Swift JQ. Ameloblastic fibro-odontoma of the anterior maxilla. Oral Surg Oral Med Oral Pathol 1993;76:294-7.   Back to cited text no. 6
[PUBMED]    
7.Dhanuthai K, Kongin K. Ameloblastic fibro-odontoma: A case report. J Clin Pediatr Dent 2004;29:75-8.  Back to cited text no. 7
[PUBMED]    
8.Favia G, Di Alberti L, Scarano A, Piattelli A. Ameloblastic fibro-odontoma: Report of two cases. Oral Oncol 1997;33:444-6.  Back to cited text no. 8
    
9.Daley T, Lovas G. Ameloblastic fibro-odontoma: Report of a case. J Can Dent Assoc 1982;48:467-9.  Back to cited text no. 9
    

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Correspondence Address:
Nagamani Narayana
Department of Oral Biology, UNMC, College of Dentistry, Lincoln, Nebraska
USA
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DOI: 10.4103/0970-9290.74237

PMID: 21187639

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    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1]

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