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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 28  |  Issue : 3  |  Page : 310-313

An odontogenic tumor mimicking a dentigerous cyst in a 13-year-old female: A rare clinical presentation


1 Department of Oral Medicine and Radiology, Church of South India College of Dental Sciences and Research, Madurai, India
2 Department of Oral and Maxillofacial Surgery, Church of South India College of Dental Sciences and Research, Madurai, India
3 Department of Oral and Maxillofacial Pathology, Church of South India College of Dental Sciences and Research, Madurai, India

Date of Submission15-Dec-2015
Date of Acceptance29-Nov-2016
Date of Web Publication13-Dec-2016

Correspondence Address:
Dr. Leelavathy Jegadeesan
21, 5th street, Park Town, P and T Nagar, Madurai - 625 017
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.195666

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   Abstract 

Ameloblastic fibroma is a relatively rare benign odontogenic tumor in which both the epithelial and ectomesenchymal components are neoplastic. A 14-year-old girl presented with a complaint of unerupted maxillary right premolar. The panoramic radiograph revealed a well-circumscribed unilocular radiolucency involving an unerupted maxillary right first premolar. The lesion was enucleated and the material was sent for histopathologic examination. This case emphasized the importance of careful differential diagnosis of intraosseous oral lesions and reported a rare lesion as well as its atypical location.

Keywords: Ameloblastic fibroma, maxillary tumor, odontogenic tumor


How to cite this article:
Jegadeesan L, Ambrose WC, Ammamuthu SV, Ragunath ST. An odontogenic tumor mimicking a dentigerous cyst in a 13-year-old female: A rare clinical presentation. J Indian Acad Oral Med Radiol 2016;28:310-3

How to cite this URL:
Jegadeesan L, Ambrose WC, Ammamuthu SV, Ragunath ST. An odontogenic tumor mimicking a dentigerous cyst in a 13-year-old female: A rare clinical presentation. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2022 Oct 7];28:310-3. Available from: https://www.jiaomr.in/text.asp?2016/28/3/310/195666


   Introduction Top


Ameloblastic fibroma (AF) is a benign odontogenic tumor considered to be a true mixed tumor composed of neoplastic epithelium and mesenchymal tissues.[1],[2],[3],[4],[5],[6] This tumor is rare, representing only 2% of all odontogenic tumors.[1],[5],[6] AF is a tumor of childhood and adolescence which occurs exclusively in the first and second decades of life usually seen in the ages of 7 to 51 years. In the majority of cases, the lesion is usually evident in the mandible, slow-growing and painless, with or without tooth eruption.[6],[7],[8],[9] However, the tooth impaction and delayed eruption may be associated with dental developmental defects or abnormalities such as amelogenesis imperfecta. Whereas in few other cases, the tumors are asymptomatic and diagnosed only during routine oral/radiographic examination.[9]

AF consists of odontogenic ectomesenchyme, resembling the dental papilla, and epithelium, resembling dental lamina and enamel organ without dental hard tissues.[7] Knowledge of the malignant potential in the mesenchymal spindle cells of AF should aid in determining the management of these benign tumors to prevent malignant transformation to ameloblastic fibrosarcoma.[3],[7] The most appropriate treatment method for AF is still uncertain, although several authors have suggested conservative approach. However, tumors may recur following surgical removal and progress to malignancy. Thus, long-term follow-up of AF is recommended.[8] This report describes an interesting case of AF that affected the maxillary region of a young girl, which was associated with an unerupted maxillary right premolar.


   Case Report Top


A 14-year-old girl reported with a complaint of an extraoral swelling associated with unerupted permanent maxillary right premolar since 1 year. General examination revealed a healthy child. The medical, surgical, family, and social histories were unremarkable. Extraorally, a diffuse swelling of size 1 × 1 cm in diameter was evident along the right nasolabial fold region, which was firm in consistency, nontender, and fixed [Figure 1]. Intraoral examination revealed a small painless expansion of the maxillary buccal cortical plate associated with missing first premolar where the overlying mucosa was intact and the color and consistency was normal [Figure 2].
Figure 1: An extraoral swelling evident near the right nasolabial fold

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Figure 2: Expansion of the maxillary buccal cortical plate associated with missing first premolar

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Maxillary occlusal radiograph revealed a unilocular radiolucency surrounded by a well-defined radiopaque border within which there was an impacted premolar [Figure 3]. Orthopantomogram (OPG) revealed a well-defined unilocular radiolucent lesion in the right maxillary region. The crown of the permanent first premolar was apparent within the lesion. A dense radiopaque mass with prominent external margins surrounded by thin radiolucent zone was evident above the crown of the unerupted tooth, which was suggestive of odontoma [Figure 4]. The second premolar was present. Computed tomography (CT) scanning confirmed the findings of the panoramic radiograph [Figure 5]. Based on the clinical and roentgenographic findings, a presumptive provisional diagnosis of dentigerous cyst was made. Differential diagnosis included expanded follicular space, adenomatoid odontogenic tumor, and AF.
Figure 3: A unilocular radiolucency surrounded by a well-defined radiopaque border within which is an impacted premolar

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Figure 4: Well-defined unilocular radiolucent lesion in the right maxilla with the crown of the permanent first premolar apparent within the lesion. A dense radiopaque mass with prominent external margins surrounded by thin radiolucent zone is evident above the crown of unerupted tooth

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Figure 5: A heterogeneously enhancing lesion in superior alveolus

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Under local anesthesia, the lesion was completely excised through an intraoral approach, thereby protecting the definitive second premolar. The enucleated material was sent for histological examination [Figure 6]. Microscopically, the lesion consisted of tumor tissue composed of cell-rich mesenchymal tissue, resembling dental papilla. The lesion also revealed numerous proliferating odontogenic islands, which were arranged in the form of narrow cords or discrete, small islands. The histopathological diagnosis confirmed the diagnosis to be AF [Figure 7].
Figure 6: Excised specimen

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Figure 7: Cell-rich mesenchymal tissue, resembling dental papilla, and numerous proliferating odontogenic islands which were arranged in the form of narrow cords or discrete, small islands

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   Discussion Top


AF are neoplasms of odontogenic epithelium and mesenchymal tissues and are categorized as mixed odontogenic tumors. Other mixed odontogenic lesions, such as ameloblastic fibro-odontomas and odontomas share some of the clinical, radiographic and histologic similarities with AF.[1],[4] Earlier it was suggested that these lesions represent a spectrum of a single entity, with AF being the least differentiated of the tumors, maturing and developing into ameloblastic fibro-odontomas and later odontomas.[4],[9] Along with tumor associated demographics, this theory has been repudiated.

The least differentiated lesion, AF, usually occurs, on average, in an older age group rather than the more differentiated ameloblastic fibro-odontoma and odontoma.[1],[2],[3],[7] Furthermore, ameloblastic fibro-odontomas and odontomas are better categorized as hamartomas, and as a result, unlike AF, they have minimal chances of recurrence or malignant transformation.[10] Therefore, despite many similarities, it is essential to differentiate AF from other mixed odontogenic lesions because it has true neoplastic qualities.[11] Radiographically, AFs are unilocular lesions, occasionally multilocular when larger, with smooth well-demarcated borders. Cortical expansion may or may not be discernable on plane film. Because these lesions are frequently associated with unerupted teeth, initially they may be interpreted as dentigerous cysts.[2],[8]

Grossly, AF appears as firm, lobular soft tissue mass with a smooth surface. If the lesion is associated with a tooth, it may accompany the specimen. A capsule is generally not appreciated. Microscopically, an AF is composed of a connective tissue background that appears to recapitulate dental papilla, resembling stellate reticulum. This tissue is composed of spindle and angular cells with little collagen, imparting a myxomatous appearance. The epithelial component is made up of thin branching cords or small nests of odontogenic epithelium with little cytoplasm and basophilic nuclei. Larger nests may show a central area of stellate reticulum.[5],[11],[12] Mitoses should not be a feature of AF. The presence of mitosis should expand the differential diagnosis to include malignant entities and ameloblastic fibrosarcoma.[5],[9] Finally, immunohistochemistry generally does not aid in differentiating AF from other mixed odontogenic tumors. Surgical excision or thorough curettage with removal of the affected teeth is the treatment of choice. The recurrence rate varies among sources, but is considered to be low. The possibility of malignant transformation of AF into ameloblastic fibrosarcoma is well-documented, but still uncommon.


   Conclusion Top


In the case reported here, the focal point was the failure of eruption of a maxillary first premolar. This disturbance may or may not be associated with a pathology such as AF. Thus, this case emphasizes the importance of careful differential diagnosis in dentistry, while reporting a rare lesion and its atypical location.

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.
Dallera P, Bertoni F, Warchetti C, Bacchini P, Campobassi A. Ameloblastic fibroma: A follow-up of six cases. Int J Oral Maxillofac Surg 1996;25:1999-2202.  Back to cited text no. 1
    
2.
Mosby EL, Russell D, Noren S, Barker BF. Ameloblastic fibroma in a 7-week-old infant: A case report and review of the literature. J Oral Maxillofac Surg 1998;56:368-72.  Back to cited text no. 2
    
3.
Chen Y, Li TJ, Gao Y, Yu SF. Ameloblastic fibroma and related lesions: A clinicopathologic study with reference to their nature and interrelationship. J Oral Pathol Med 2005;34:588-95.  Back to cited text no. 3
    
4.
Barnes L, Eveson JW, Reichart PA, Sidransky P. Pathology and genetics of tumours of the head and neck: World Health Organization Classification of Tumours: International Histological Classification of Tumors. Lyon: IARC Press; 2005.  Back to cited text no. 4
    
5.
Ebenezer V, Ramalingam B. A cross-sectional survey of prevalence of odontogenic tumours. J Maxillofac Oral Surg 2011;9:369-74.  Back to cited text no. 5
    
6.
McGuinness NJ, Faughan T, Bennani F, Connolly CE. Ameloblastic fibroma of the anterior maxilla presenting as a complication of tooth eruption: A case report. J Orthod 2001;28:115-7.  Back to cited text no. 6
    
7.
López RA, Ortega L, Corchón MA, Sández AB. Ameloblastic fibroma of the mandible: Report of the two cases. Med Oral 2003;8:150-3.  Back to cited text no. 7
    
8.
Dimitrakopoulos I, Psomaderis K, Zaramboukas T. Ameloblastic fibroma of the mandible associated with root resorption and unerupted teeth: A case report. Quintessence Int 2008;39:523-7.  Back to cited text no. 8
    
9.
Kim SG, Jang HS. Ameloblastic fibroma: Report of a case. J Oral Maxillofac Surg 2002;60:216-8.  Back to cited text no. 9
    
10.
Chen Y, Wang JM, Li TJ. Ameloblastic fibroma: A review of published studies with special reference to its nature and biological behavior. Oral Oncol 2007;43:960-9.  Back to cited text no. 10
    
11.
Canger EM, Çelenk P, Yenisey M, Odyakmaz SZ. Amelogenesis imperfecta, hypoplastic type associated with some dental abnormalities: A case report. Braz Dent J 2010;21:170-4.  Back to cited text no. 11
    
12.
Philipsen HP, Reichart PA, Praetorius F. Mixed odontogenic tumors and odontomas. Considerations on interrelationship. Review of the literature and presentation of 134 new cases of odontomas. Oral Oncol 1997;33:86-99.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]


This article has been cited by
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Human Pathology: Case Reports. 2020; 20: 200372
[Pubmed] | [DOI]



 

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