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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 27  |  Issue : 4  |  Page : 584-587

Ameloblastic fibro-odontoma: A diagnostic challenge


1 Department of Oral Medicine and Radiology, Sarjug Dental College and Hospital, Darbhanga, Bihar, India
2 Department of Psychiatry, Sri Krishna Medical College and Hospital, Muzaffarpur, Bihar, India
3 Department of Public Health Dentistry, Institute of Dental Sciences, Bareilly, Uttar Pradesh, India

Date of Submission02-Aug-2015
Date of Acceptance26-Feb-2016
Date of Web Publication19-Aug-2016

Correspondence Address:
Dr. Abhijeet Alok
Department of Oral Medicine and Radiology, Sarjug Dental College and Hospital, Hospital Road, Laheriasarai, Darbhanga - 846 003, Bihar
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.188768

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   Abstract 

Ameloblastic fibro-odontoma (AFO) is a rare mixed odontogenic tumor. It occurs predominantly in children and young adults with no sex predilection, mostly in the mandible posterior region. A painless swelling is the most common clinical sign of AFO. Radiographically, AFO shows a circumscribed radiolucency, which contains radiopaque foci of various sizes and shapes. Histological examination reveals a fibrous soft tissue, islands of odontogenic epithelium, and a disordered mixture of dental tissues. Treatment of AFOs usually consists of enucleation or surgical curettage, which is possible due to their benign biological behavior. Here, we are reporting a case of AFO in a 20-year-old male.

Keywords: Ameloblastic fibro-odontoma, benign tumor, mandible, mixed odontogenic tumor, odontoma


How to cite this article:
Alok A, Singh ID, Singh S. Ameloblastic fibro-odontoma: A diagnostic challenge. J Indian Acad Oral Med Radiol 2015;27:584-7

How to cite this URL:
Alok A, Singh ID, Singh S. Ameloblastic fibro-odontoma: A diagnostic challenge. J Indian Acad Oral Med Radiol [serial online] 2015 [cited 2021 Jul 26];27:584-7. Available from: https://www.jiaomr.in/text.asp?2015/27/4/584/188768


   Introduction Top


Ameloblastic fibro-odontoma (AFO) is a rare odontogenic tumor accompanied by tooth-like structures. [1] It may inhibit tooth eruption or displace the involved teeth, but the teeth involved are vital. [2] According to the World Health Organization (WHO), AFO is defined as a neoplasm composed of proliferating odontogenic epithelium embedded in a cellular ectomesenchymal tissue that resembles dental papilla, and with varying degrees of inductive change and dental hard tissue formation. [3] Clinically, this neoplasm behaves as a slow-growing, well-encapsulated, benign lesion, and it is frequently asymptomatic. [4] There is no sex predilection. Radiographically, the lesions present as well-circumscribed, unilocular or multilocular radiolucent lesions with radiopaque border at their periphery. Treatment of AFO includes conservative surgery with enucleation. [5] Prognosis is excellent. [6] In this article, we report a case of AFO in a 20-year-old male patient, with details on its clinical features and differential diagnosis.


   Case Report Top


An 20-year-old male patient reported to the Department of Oral Medicine and Radiology with the chief complaint of pain in lower back tooth region since 2 months. History of present illness revealed that pain was sudden in onset, severe in intensity, and intermittent. It was localized in nature and relieved by itself. He gave history of extraction in the same region 2 years back. He also had history of pus discharge from that region. On extraoral examination, a diffuse swelling was present at the angle of mandible on the right side [Figure 1]. It extended superiorly 2 cm from the tragus of ear till the angle of mandible inferiorly. Anteroposteriorly, it extended 2 cm from the corner of the mouth on the right side till 0.5 cm to the angle of mandible. The overlying surface appeared normal. Intraorally, an erythematous area was present on the right mandibular posterior alveolus area [Figure 2]. The overlying surface appeared rough. The color varied from red to yellow. Slough was present in the affected area. On palpation, it was tender. Pus discharge was present. Based on the history and clinical examination, a provisional diagnosis of chronic suppurative osteomyelitis in right mandible was made. Investigations like orthopantomogram (OPG) and histopathology examination were done. OPG [Figure 3] showed a large, well-demarcated radiolucency containing radiopaque areas of irregular shape, affecting the angle and ramus of mandible. The lesion contained multiple foci of radiopacities forming a radiopaque mass of around 2 × 4 cm in diameter. The lower second molar was displaced to the angle of mandible. A well-circumscribed radiolucency was present around the crown of the mandiblular second molar. Radiographic differential diagnosis included AFO, immature complex odontoma, calcifying epithelial odontogenic tumor, and calcifying odontogenic cyst. The lesion was surgically excised and sent for histopathological investigation. Histopathological report revealed strands and islands of odontogenic epithelium showing peripheral palisading and loosely arranged central cells, identical to stellate reticulum, embedded in a myxoid cell-rich stroma resembling the dental papilla. Dentin and enamel were also present [Figure 4]. Based on the clinical, radiographic and histopathologic findings a final diagnosis of AFO was made.
Figure 1: Extraoral view of patient

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Figure 2: Intraoral view of patient

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Figure 3: OPG showing the lesion in the right mandible region

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Figure 4: Histopathological picture of ameloblastic fibro-odontoma

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   Discussion Top


AFOs are benign mixed odontogenic tumors that contain cords and nests of odontogenic epithelium and some calcified odontogenic tissue in a myxomatous stroma. This tumor represents 2% of odontogenic tumors, but the prevalence among oral biopsies is about 1%. [7],[8] AFO usually occurs in people less than 20 years of age. Thus, age plays an important characteristic feature in differential diagnosis. Most of the patients are in the first two decades of life, with the average age of the patient being 12 years. [9] Slootweg reviewed 50 patients of AFO and the mean age for occurrence of AFO was found to be 8.1 years. [10] The proper classification of AFO is still not clear. Some consider it to be a neoplasm, while some others consider it to be a hamartoma. [11],[12] Both Slootweg [10] and Philipsen et al. [13] indicated that AFO has a hamartomatous character, but in contrast, the ameloblastic fibroma has a neoplastic nature. But most authors now agree that AFO is a separate entity. AFO provides a diagnostic challenge as it is histologically indistinguishable from immature complex odontoma. The relative arrangement of the soft tissues and the stage of development of the involved tooth are useful criteria for diagnosis. According to the revised WHO classification, AFO is a benign tumor without invasive growth; this is in contrast to the ameloblastoma. The most common complaints of the patients are swelling in the affected area which may lead to facial disfigurement and failure of tooth eruption. AFO often displaces erupted teeth. Pain and paresthesia in the affected region are uncommon findings. [2] Radiographically, the periphery of this tumor is usually well defined and sometimes corticated. Internal structure of AFO is mixed, with majority of lesions being radiolucent. Small lesions may appear as enlarged follicles with only one or two small, discrete radiopacities. Larger lesions may have a more extensive calcified internal structure. In some cases, these small calcifications have a round shape with a radiopaque enamel-like margin, giving a shape similar to a small doughnut. But in some cases, lesions appear to be completely radiolucent making it difficult to differentiate them from odontogenic lesions. Sometimes, the mineralized component predominates in the tumor and the lesion may be radiographically similar to complex odontoma. [14] Histopathologically, the lesion is controversial. It is considered to be a combination of odontome and ameloblastic fibroma. AFO is composed of strands, cords, and islands of odontogenic epithelium immersed in a cell-rich embryonic connective tissue, containing a variable amount of irregular formation of enamel and osteodentine, as seen in our patient. [10] Differential diagnosis of AFO should include immature complex odontoma, compound odontoma, calcifying epithelial odontogenic tumor, adenomatoid odontogenic tumors, ameloblastic fibroma, and calcifying odontogenic cyst [Table 1]. As AFO is well encapsulated and there is little tendency for local invasion, the treatment of choice is conservative surgery with enucleation. As it is often associated with unerupted tooth, the tooth must be removed with the mass, as done in our case. When associated with permanent tooth, the tumor is enucleated, allowing the involved permanent tooth to erupt. If the tumor is encasing the permanent tooth, both the tumor and the tooth should be removed to minimize recurrences. AFO reoccurs in those cases where inadequate surgical removal has taken place or if tumor remnants persist in the enucleated margins. Malignant transformation of AFO to ameloblastic fibrosarcoma or ameloblastic fibro-odontosarcoma is rare, but few cases have been reported. [6],[15]
Table 1: Differential diagnosis of ameloblastic fibro-odontoma


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   Conclusion Top


AFO is a rare entity. As it closely resembles immature complex odontome, diagnosis becomes difficult. The clinician should consider all the lesions which closely resemble AFO, in the differential diagnosis. But correct history, investigations, and observation can help clinicians arrive at a diagnosis. AFO can show malignant transformation, so constant follow-up of patients should be done.

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.
Piette EM, Tideman H, Wu PC. Massive maxillary ameloblastic firo-odontoma. Case report with surgical management. J Maxillofac Surg 1990;48:526-30.  Back to cited text no. 1
    
2.
Miller AS, Lopez CF, Pullon PA, Elzay RP. Ameloblastic fibroodontoma: Report of seven cases. Oral Surg Oral Med Oral Pathol 1976;41:354-65.  Back to cited text no. 2
    
3.
Baker WR, Swift JQ. Ameloblastic fibroodontoma of the anterior maxilla. Report of a case. Oral Surg Oral Med Oral Pathol 1993;76:294-7.  Back to cited text no. 3
    
4.
Reis SR, de Freitas CE, do Espirito Santo AR. Management of ameloblastic fibroodontoma in a 6-year-old girl preserving the associated impacted permanent tooth. J Oral Sci 2007;49:331-5.  Back to cited text no. 4
    
5.
Friedrich RE, Siegert J, Donath K, Jäkel KT. Recurrent ameloblastic fibro-odontoma in a 10-year-old boy. J Oral Maxillofac Surg 2001;59:1362-6.  Back to cited text no. 5
    
6.
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. 6
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7.
Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors: Analysis of 706 cases. J Oral Surg 1978;36:771-8.  Back to cited text no. 7
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8.
Daley DT, Wysocki GP, Pringle GA. Relative incidence of odontogenic tumors and oral and jaw cysts in a Canadian population. Oral Surg Oral Med Oral Pathol 1994;77:276-80.  Back to cited text no. 8
    
9.
Minderjahn A. Incidence and clinical differentiation of odontogenic tumors. J Maxillofac Surg 1979;7:142-50.  Back to cited text no. 9
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10.
Slootweg PJ. An analysis of the interrelationship of the mixed odontogenic tumors-amelobastic fibroma, ameloblastic fibro-odontoma, and the odontomas. Oral Surg Oral Med Oral Pathol 1981;51:266-76.  Back to cited text no. 10
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11.
Olech E, Alvares O. Ameloblastic odontoma. Report of a case. Oral Surg Oral Med Oral Pathol 1967;23:487-92.  Back to cited text no. 11
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12.
Schmidseder R, Hausamen JE. Multiple odontogenic tumors and other anomalies. An autosomal dominantly inherited syndrome. Oral Surg Oral Med Oral Pathol 1975;39:249-58.  Back to cited text no. 12
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13.
Philipsen HP, Reichart PA, Praetorius F. Mixed odontogenic tumors and odontomas: Considerations on interrlationship. Review of the literature and presentation of 134 new cases of odontomas. Oral Oncol 1997;33:86-99.  Back to cited text no. 13
    
14.
Hawkins PL, Sadeghi EM. Ameloblastic fibro-odontoma: Report of case. J Oral Maxillofac Surg 1986;44:1014-9.  Back to cited text no. 14
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15.
Wang S, Shi H, Wang P, Yu Q. Ameloblastic fibro-odontosarcoma of the mandible: Imaging findings. Dentomaxillofac Radiol 2011;40:324-7.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]
 
 
    Tables

  [Table 1]



 

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