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CASE REPORT
Year : 2018  |  Volume : 7  |  Issue : 4  |  Page : 281-284

Ameloblastic fibro-odontoma or complex adontoma: A diagnostic dilemma?


1 Department of Oral Medicine and Radiology, Mamata Dental College and Hospital, Khammam, Telangana, India
2 Department of Oral Pathology and Microbiology, Mamata Dental College and Hospital, Khammam, Telangana, India

Date of Web Publication10-Jan-2019

Correspondence Address:
Dr. Kotya Naik Maloth
Department of Oral Medicine and Radiology, Mamata Dental College and Hospital, Giriprasad Nagar, Khammam - 507 002, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JDRNTRUHS.JDRNTRUHS_45_17

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  Abstract 


Ameloblastic fibro-odontoma is a rare benign mixed odontogenic tumor exhibiting the histologic characteristics of ameloblastic fibroma and complex odontoma. Clinically, it represents a well-defined asymptomatic swelling with deformity of face, and radiographically a well-defined radiolucent area, which contains radiopaque foci of various sizes and shapes. These features overlap with other lesions such as calcifying odontogenic cyst, calcifying epithelial odontogenic tumor, and adenomatoid odontogenic tumor which makes a diagnostic dilemma for oral clinicians. Hereby we report such a rare case in a 7-year-old boy.

Keywords: Complex odontoma, mixed odontogenic tumor, odontoameloblastoma


How to cite this article:
Reddy K V, Maloth KN, Lakshmi MV, Thakur M. Ameloblastic fibro-odontoma or complex adontoma: A diagnostic dilemma?. J NTR Univ Health Sci 2018;7:281-4

How to cite this URL:
Reddy K V, Maloth KN, Lakshmi MV, Thakur M. Ameloblastic fibro-odontoma or complex adontoma: A diagnostic dilemma?. J NTR Univ Health Sci [serial online] 2018 [cited 2019 Sep 16];7:281-4. Available from: http://www.jdrntruhs.org/text.asp?2018/7/4/281/249830




  Introduction Top


Ameloblastic fibro-odontoma (AFO) is a rare benign mixed odontogenic tumor, defined by the World Health Organization (WHO) as “a neoplasm composed of proliferating odontogenic epithelium in a cellular ectomesenchymal tissue with varying degrees of inductive changes and dental hard tissue formation.”[1],[2] It is characterized by a well-defined, slow-growing, and expansible asymptomatic swelling usually detected as a result of failure of tooth eruption.[3] The frequency of occurrences reported was 1%–3%.[4] Hereby we report such a rare case of AFO in a 7-year-old boy.


  Case Report Top


A 7-year-old boy reported to the department with a complaint of swelling on the right side of the face since 2 months [Figure 1]. The patient was apparently normal 2 months back; later he noticed a swelling which was small in size which gradually increased to attain the present size. The swelling was not associated with any pain and discharge. Dental and medical history was noncontributory. On extraoral examination, a diffuse swelling was noted on the right side of the mandible in the posterior region, which was hard in consistency and nontender. On intraoral examination, a well-defined swelling was noted in relation to the right retromolar region (distal to 85) which was hard in consistency and nontender with buccal and mild lingual cortical expansion [Figure 2].
Figure 1: Extraoral swelling on right side of the face

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Figure 2: Intraoral swelling with buccal vestibular obliteration on right side

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Baseline investigations were noncontributory. Radiographic examination with mandibular occlusal radiograph revealed multiple radiopacities enveloped by a radiolucent zone, surrounded by corticated border with buccal and mild lingual cortical expansion [Figure 3], and panoramic radiograph revealed a well-defined large area with multiple radiopacities enveloped by the radiolucent zone and surrounded by corticated border, extending from the distal aspect of the right primary second molar to the anterior border of the ramus of the mandible with the presence of an impacted right lower permanent first molar [Figure 4].
Figure 3: (a) Intraoral radiograph reveals radiopaque lesion enveloped by the radiolucent zone (b) mandibular occlusal radiograph showing a well-defined radiopaque lesion enveloped by the radiolucent zone with buccal and lingual cortical expansion.

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Figure 4: Panoramic radiograph showing a well-defined radiopaque lesion enveloped by the radiolucent zone extending from #85 regions to the anterior border of the ramus of the mandible with impacted #46 is also seen

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Based on the clinical and radiographical findings, a provisional diagnosis of complex odontome with a differential diagnosis of AFO, calcifying epithelial odontogenic tumor (CEOT), and calcifying odontogenic cyst (COC) was made. Excisional biopsy was done [Figure 5] and sent for histopathological examination which revealed connective tissue stroma that showed cell-rich ectomesenchymal component and odontogenic epithelium arranged in islands and strands. Irregularly arranged calcified component of minimal enamel matrix and dentinoid material was seen in the ectomesenchymal tissue. Odontogenic islands were lined by columunar or preameloblastic-like cells were noted, which was suggestive of ameloblastic fibro-odontoma [Figure 6].
Figure 5: Excisional biopsy specimen

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Figure 6: (a) (10×): Microscopic appearance of the lesion (H and E). Islands of odontogenic epithelium with peripheral cells resembling ameloblastic and central cells (stellate reticulum cells) of the enamel organ and loose cellular connective tissue similar to dental papilla; (b) (40×): Calcified component of dentinoid matrix adjacent to the epithelial islands

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  Discussion Top


The present case meets all the clinical, radiographical, and histopathological features for the diagnosis of AFO, which is a rare mixed odontogenic tumor. According to WHO classification of odontogenic tumors, it is placed under odontogenic epithelium with odontogenic ectomesenchyme, with or without hard tissue formation. AFO is a tumor similar to ameloblastic fibroma (AF) in terms of histological features, but with inductive changes that lead to the formation of dentin and enamel.[2] The term “ameloblastic fibro-odontoma” represents a histological combination of AF and complex odontoma.[4] Cahn and Blum (1952) postulated that histologically AF least differentiated tumor initially and develops later into a moderately differentiated form, AFO, and finally into complex odontoma.[2] The majority of mixed odontogenic tumors are hamartomatous and are considered as a part of developing complex odontoma line. Complex odontoma begins with AF or ameloblastic fibrodentinoma, and further these tumors develop into the second stage called AFO and finally convert into completely mineralized complex odontoma.[1] AFO is relatively rare, with frequency of occurrence about 1%–3%.[5] Most of the lesions are usually diagnosed during the first and second decades of life without any gender predilection and occur equally in both maxilla and mandible at the posterior region.[3],[4],[5],[6] The mean age of the patients as reported by Hooker is 11.5 years.[6] The present case is in accordance with the literature. Two most common complaints reported by the affected individuals are asymptomatic swelling and failure of tooth eruption, which are usually diagnosed incidentally on radiographic examination.[4] Occasionally, the tumor exhibits marked swelling, which results in facial disfigurement,[7] as in the present case.

Radiographically, the lesion usually appears as a well-defined radiolucent area containing various amounts of radiopaque material of irregular size and form.[1],[4],[6] Most cases are associated with an unerupted tooth located in the periphery, and often it is noted that the associated tooth is displaced in an apical direction, indicating the lesion is arising above the tooth.[1] Our case presented with radiographic features in accordance with the literature.

The other lesions showing mixed radiolucent and radiopaque features such as CEOT, immature complex odontoma, COC, and adenomatoid odontogenic tumor should be differentiated with AFO. Microscopically, it is composed of strands, cords, and islands of odontogenic epithelium embedded in a cell-rich primitive ectomesenchyme, resembling dental papilla. Dentin and enamel matrix is also seen; whether or not the tissues actually resemble “teeth” is irrelevant.[8]

As the lesion is nonaggressive and well-encapsulated, the recommended treatment is generally enucleation and there is little tendency for local invasion. The recurrence rate is very low; it happens when conservative treatment, with an attempt to save associated teeth, is performed.[1],[9] Malignant transformation rate is rare, but Howell and Burkes reported two cases of AFO that showed malignant transformation to ameloblastic fibrosarcoma.[10] The present case was treated with conservative surgery with enucleation. After a follow-up of 8 months, no recurrence was observed.


  Conclusion Top


The purpose of reporting this case is to enlighten the clinical and radiographic features of AFO, as it overlaps with other lesions such as complex odontoma, CEOT, and COC. This rare lesion remains a diagnostic dilemma for the oral physicians.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Silva GCC, Jham BC, Silva EC, Horta MCR, Godinho SHP, Gomez RS. Ameloblastic fibro-odontoma. Oral Oncol Extra 2006;42:217-20.  Back to cited text no. 1
    
2.
Barnes L, Eveson JW, Reihcart P, Sidransky D. Pathology and Genetics of Head and Neck Tumours. World Health Organization Classification of Tumours. Volume 9. IARC Press; 2005.  Back to cited text no. 2
    
3.
Gupta R, Astekar M, Chittlangia RK. Ameloblastic fibro-odontoma of mandible. J Dent Sci Oral Rehab 2014;5:103-6.  Back to cited text no. 3
    
4.
Chang H, Shimizu MS, Precious DS. Ameloblastic fibro-odontoma: A case report. J Can Dent Assoc 2002;68:243-6.  Back to cited text no. 4
    
5.
Elen de Souza Tolentino, Centurion BS, Lima MC, Faria PF, Consolaro A, Santana E. Int J Dent 2010;2010:1-4.  Back to cited text no. 5
    
6.
Hooker SP. Ameloblastic odontoma: An analysis of twenty six cases. J Oral Surg 1967;24:375-6.  Back to cited text no. 6
    
7.
Reis SR. Management of ameloblastic fibro-odontoma in a 6-year-old girl preserving the associated impacted permanent tooth. J Oral Sci 2007;49:331-5.  Back to cited text no. 7
    
8.
Furst I, Pharoah M, Phillips J. Recurrence of an ameloblastic fibro-odontoma in a 9-year old boy. J Oral Maxillofac Surg 1999;57:620-3.  Back to cited text no. 8
    
9.
Tomich CE. Benign mixed odontogenic tumors. Semin Diagn Pathol 1999;16:308-16.  Back to cited text no. 9
    
10.
Howell RM, Burkes EJ Jr. Malignant transformation of ameloblastic fibro-odontoma to ameloblastic fibrosarcoma. Oral Surg Oral Med Oral Pathol 1977;43:391-401.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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