|Year : 2013 | Volume
| Issue : 4 | Page : 275-278
Ameloblastic fibrodentinoma involving anterior maxilla: A rare case report
Shailaja Sankireddy1, Atul Kaushik1, Bhargavi A Krishna2, Asha Latha G Reddy2, VC Vinod1, V Sridevi1
1 Department of Oral Medicine and Radiology, SGT Dental College and Hospital, Gurgaon, Haryana, India
2 Department of Oral Pathology, Panineeya Dental College and Hospital, Hyderabad, Andhra Pradesh, India
|Date of Web Publication||21-Nov-2013|
Department of Oral Medicine and Radiology, SGT Dental College and Hospital, Gurgaon, Haryana
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Ameloblastic fibroma (AF) and related lesions comprise a complex group of mixed odontogenic tumors. Ameloblastic fibrodentinoma (AFD) is a rare tumor and is considered as a histological variant of AF showing inductive changes that lead to the formation of dentin. Although the most common site for this tumor is the posterior mandible, hereby we are reporting a case of AFD in the anterior maxillary region which is a very rare site for this group of tumors. The present case report highlights the clinical, radiological and histological presentation of AFD in order to raise awareness for the earlier diagnosis and precise management of this rare pathological entity.
Keywords: Ameloblastic fibrodentinoma, ameloblastic fibroma, odontogenic tumor
|How to cite this article:|
Sankireddy S, Kaushik A, Krishna BA, Reddy AG, Vinod V C, Sridevi V. Ameloblastic fibrodentinoma involving anterior maxilla: A rare case report. J Indian Soc Pedod Prev Dent 2013;31:275-8
|How to cite this URL:|
Sankireddy S, Kaushik A, Krishna BA, Reddy AG, Vinod V C, Sridevi V. Ameloblastic fibrodentinoma involving anterior maxilla: A rare case report. J Indian Soc Pedod Prev Dent [serial online] 2013 [cited 2020 Feb 20];31:275-8. Available from: http://www.jisppd.com/text.asp?2013/31/4/275/121832
| Introduction|| |
Ameloblastic fibroma (AF) and related lesions have been defined by World Health Organization (WHO) as "neoplasms composed of proliferating odontogenic epithelium embedded in a cellular ectomesenchymal tissue resembling the dental papilla and varying degrees of inductive changes and dental hard tissue formation."  Ameloblastic fibrodentinoma (AFD) is one of the rare entity of the above mentioned group, which is believed to be an intermediate stage between AFD and ameloblastic fibro-odontoma (AFO). However, AFD is a controversial neoplasm and its existence as an independent entity has still not been fully accepted.  AFD is predominantly a slow growing asymptomatic lesion frequently involving the mandibular posterior region.  Treatment for the initial lesion is generally conservative, with long-term follow-up evaluations indicating no recurrence. 
The present case report is an attempt to highlight various clinical, radiological and histological presentation of AFD, which will help the general and specialty practitioner in earlier diagnosis and more precise management of this set of odontogenic tumors.
| Case Report|| |
A 14-year-old male patient reported to the Department of Oral Medicine and Radiology with the complaint of a painless swelling of the right upper back face for last 6 months. Extra-oral examination revealed a diffuse swelling over the right anterior maxilla with obliteration of the nasolabial fold [Figure 1]. Intra-orally, a diffuse, firm, non-tender swelling was noted in the buccal vestibule extending from the region of the right maxillary central incisor to the first premolar [Figure 2]. A diffuse swelling was also observed on the palatal aspect of the same region. A mobile retained deciduous right maxillary canine was present. The corresponding permanent canine was missing.
|Figure 1: A diffuse swelling over the right anterior maxilla with obliteration of the nasolabial fold|
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|Figure 2: Swelling involving the buccal vestibule extending from the region of the right maxillary central incisor to the fi rst premolar|
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A set of plain radiographs was taken, which included periapical, occlusal [Figure 3] and panoramic [Figure 4] views. The radiographs revealed a well-circumscribed, unilocular radiolucency involving the impacted permanent canine. The radiolucency was triangular in shape, with the apex between the divergent roots of the lateral incisor and the first premolar and the base extending to the cervical third of the root of the impacted canine. The deciduous canine had significant root resorption. Based on the history, clinical examination and radiographic evaluation, a diagnosis of dentigerous cyst associated with 13 was arrived at.
|Figure 3: Well-circumscribed, unilocular radiolucency involving the|
impacted permanent canine
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|Figure 4: Apex of radiolucency between the roots of lateral incisor and the fi rst premolar and the base extending to the cervical third of the root of the impacted canine|
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Patient was referred to Department of Oral and Maxillofacial Surgery where an incisional biopsy was performed. The histopathological examination revealed strands and islands of odontogenic epithelium in a cell rich primitive ectomesenchyme resembling the dental papilla. The islands consisted of a peripheral row of high cubiodal to columnar ameloblast like cells. Some of the areas were highly cellular showing zone of hyalinization and formation of dentinoid at the periphery of the odontogenic epithelial islands [Figure 5]. Based on the histopathological examination, a final diagnosis AFD was arrived at.
The tumor was enucleated along with impacted 13 through intra-oral approach under general anesthesia. After enucleation and proper hemostasis, the wound was closed by 3/0 vicryl. No sign of recurrence of AFD was recorded during the 36 months follow-up period.
|Figure 5: Zone of hyalinization and formation of dentinoid at the periphery of the odontogenic epithelial islands|
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| Discussion|| |
Mixed odontogenic tumors comprise a rare group of lesions with diverse histopathological types and clinical behavior. Despite numerous efforts, there is still considerable confusion concerning the nature and inter relationship of these mixed odontogenic tumors and related lesions. In general, these lesions comprise AF, AFD and AFO.
AFD is a very rare tumor, which is considered to occupy a stage between AF and AFO by some authors based on the extent of histodifferentiation. Although the existence of AFD as a separate entity is not completely accepted, it is defined as "a neoplasm similar to AF, but also showing inductive changes that lead to the formation of dentin."  In some cases, diagnosed as AFD, dentin matrix or dentinoid tissue is an area of hyalinization around epithelial component and some authors have suggested that such hyalinized zone may not represent dentin formation. In other cases, a hyalinized area containing entrapped cells has been considered to be an abortive dentin or dentinoid tissue. 
It is also important to note that in the revised WHO classification of odontogenic tumors AFD and "dentinoma" are used synonymously. However, there are histological differences between several cases reported previously as dentinoma and AFD. Dentinoma is a very rare odontogenic neoplasm composed of odontogenic epithelium and immature connective tissue, characterized by the formation of dysplastic dentin. 
AFD presents itself as an asymptomatic, slow growing lesion commonly associated with an unerupted tooth. In the present case also, the lesion was associated with the crown of unerupted 13 which was seen during conventional radiological investigation. AFD is most commonly seen in young individuals, generally in first and second decade. However, some cases of AFD have been reported in adults as old as 60 years also. A higher male predilection is seen in AFD with a male to female ratio of 3:1.  Similar pattern of age and gender predilection was seen in our patient who was 14-year-old male. The tumors related to deciduous teeth usually arise in incisor area while those associated to permanent teeth mostly develop in molar area.  However, the present case report highlights a lesion in the anterior maxilla, which is a very rare site of occurrence for AFD.
AFD radiographically appears as a well delineated radiolucency with varying degree of radio-opacity owing to the formation of dentin. If an embedded tooth is involved, the tumor is often closely associated with the crown.  In the present case also, the radiographs revealed a well circumscribed, unilocular radiolucency involving the impacted permanent canine. Similar appearance may also be seen in AFO and odontoma. Hence a careful consideration must be given to the age and gender of the patient, location of the lesion, radiological findings and histological features while making a diagnosis of AFD.
In cases of AFD, epithelium is usually in the form of slender strands consisting of double layer of cuboidal cells and connective tissue resembles dental papilla. Presence of hard tissue similar to dentin may be seen near epithelial cells.  In the present case, the histopathological features were consistent with earlier reports. AFD has been reported to undergo malignant transformation as ameloblastic fibrodentinosarcoma where only the mesenchymal component shows malignant transformation.  Although the mechanism of conversion into malignancy is not very clearly understood, multiple surgical procedures of recurrent lesions remain one of the important risk factor. Metastasis is not common and fatal cases have been usually associated with uncontrollable local infiltration following numerous recurrence. 
Most often AFD have a distinctive fibrous connective tissue capsule. Since it is a capsulated lesion, conservative surgery like simple enucleation remains the treatment of choice.  If the lesion is aggressive and presents with erosion of the cortical plate, a radical resection should be performed. In the present case, the tumor was enucleated along with impacted 13 through intra-oral approach under general anesthesia and no signs of recurrence of were observed during the 36 months follow-up period.
| Conclusion|| |
Though rare, AFD and related lesions should be considered in the differential diagnosis of swellings of maxillofacial region. A proper clinical, radiological and histological evaluation must be established and the treatment should be accordingly planned for AFD cases. A regular follow-up protocol should be established to rule out any evidence of recurrence and malignant transformation. A detailed research is required in order to understand the relationship of AFD and related lesions, their biological behavior and management strategies.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]