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 Table of Contents  
Year : 2020  |  Volume : 32  |  Issue : 1  |  Page : 65-68

Extrafollicular AOT presenting as a gingival overgrowth: A rare case report

1 Department of Oral Medicine and Radiology, Dayananda Sagar College of Dental Sciences, Bengaluru, Karnataka, India
2 Department of Oral Medicine and Radiology, SDM College of Dental Sciences, Dharwad, Karnataka, India
3 Department of Orthodontics and Dentofacial Orthopedics, Dayananda Sagar College of Dental Sciences, Bengaluru, Karnataka, India

Date of Submission09-Jul-2019
Date of Decision31-Aug-2019
Date of Acceptance28-Dec-2019
Date of Web Publication17-Apr-2020

Correspondence Address:
Dr. Preeti Patil
Senior Lecturer, Department of Oral Medicine and Radiology, Dayananda Sagar College of Dental Sciences, Bangalore, Karnataka
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jiaomr.jiaomr_128_19

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Adenomatoid odontogenic tumor (AOT) is an uncommon, benign, asymptomatic, slow-growing tumor with marked predilection for occurrence in the anterior maxilla. It represents 3% of all odontogenic tumors. There are three clinicopathological variants of AOT, namely, follicular, extrafollicular, and peripheral. This paper describes in detail a rare case of an extrafollicular adenomatoid odontogenic tumor occurring in mandible and presenting as gingival growth.

Keywords: AOT, extrafollicular AOT, mandible

How to cite this article:
Patil P, Nandimath K, Burde K, Kabbur KJ. Extrafollicular AOT presenting as a gingival overgrowth: A rare case report. J Indian Acad Oral Med Radiol 2020;32:65-8

How to cite this URL:
Patil P, Nandimath K, Burde K, Kabbur KJ. Extrafollicular AOT presenting as a gingival overgrowth: A rare case report. J Indian Acad Oral Med Radiol [serial online] 2020 [cited 2022 Jan 27];32:65-8. Available from: https://www.jiaomr.in/text.asp?2020/32/1/65/282603

   Introduction Top

Adenomatoid odontogenic tumor (AOT) is a relatively uncommon distinct odontogenic neoplasm that was first described by Steensland in 1905.[1] Some authors consider AOTs to be benign and noninvasive neoplasms; however, others describe them as developmental hamartomas odontogenic growths. AOT usually appears in the age group of 5–50 years; two-thirds of the cases are diagnosed in the second decade of life, with an average age of 16 years. There is a predilection of AOT in females (female to male ratio = 1.9: 1). At least 75% of lesions occur in the anterior maxilla followed by the anterior mandible.[2] Three clinicopathologic variants of the tumor have been described, namely, intraosseous follicular (73% of all AOT cases), intraosseous extrafollicular (24%), and (3%) peripheral.[3] In this paper, we describe in detail a case of extrafollicular AOT in the mandibular canine premolar region presenting as a gingival swelling along with the literature review of extrafollicular AOT.

   Case Report Top

A 16-year-old female reported with a complaint of painless swelling in gingiva in the lower left back region since 4 months. The swelling was associated with bleeding during brushing and interfered with mastication. Dental history and medical history were noncontributory. On intraoral examination, there was gingival overgrowth extending from 32 to 35 region measuring 3 × 5 cm in size [Figure 1]. The growth extended to cover the occlusal surface of 34 and 35. The surface of the swelling was lobulated. On palpation, the swelling was firm in consistency and nontender. There was an expansion of buccal and lingual cortices. There was grade II mobility with 33 and grade I mobility with 34. 34 was lingually displaced. Clinical diagnosis of ameloblastoma, odontogenic keratocyst, adenomatoid odontogenic tumor (AOT), and calcifying epithelial odontogenic tumor (CEOT) was given. OPG revealed the presence of well corticated bilocular radiolucency from 32 to 35 regions causing crestal bone perforation [Figure 2]. There was displacement of 33 and 34 and extrusion of 32, 33, and 34. There was a loss of lamina dura in relation to 32, 33, and 34. Occlusal radiograph showed expansion and thinning of buccal and lingual cortices and displacement of 33 and 34 [Figure 3]A. IOPA revealed fine calcifications in the centre of the lesion [Figure 3]B. Radiographic differential diagnosis included AOT and CEOT. The lesion was surgically exposed [Figure 4]A and biopsy samples were collected. Histopathological sections revealed densely arranged cuboidal to spindle shaped odontogenic epithelial cells in variable duct-like and rosette-like structures [Figure 5]. Based on clinical findings and histopathologic features, the final diagnosis of extrafollicular type of AOT was given. The tumor was surgically removed [Figure 4]B and 33 and 34 were extracted.
Figure 1: Gingival growth covering 34 and 35. Lingually displaced 34

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Figure 2: Well corticated bilocular radiolucency with the displacement of 33 and 34

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Figure 3: A: expansion and thinning of cortices with the displacement of 33 and 34. Figure 3B: IOPA shows fine calcification in the center of lesion and crestal perforation

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Figure 4: A: surgical exposure of the lesion, B: surgically removed tumor

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Figure 5: Cuboidal to columnar cells arranged in the form of nests and duct-like pattern

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   Discussion Top

Extrafollicular AOT is an uncommon variant of AOT and represents 24% of all AOT. It is a central lesion that is not related to the embedded teeth.[4] The most common site of extrafollicular AOT is anterior region of maxilla (incisor to canine) and anterior region of mandible being the second common site.[5] However, in our case, the AOT was located in mandibular canine premolar region. The extrafollicular type is subdivided into E1- no relation to tooth structure either erupted or unerrupted, E2- intraradicular adjacent roots diverge apically due to tumor expansion, E3- superimposed on the root apex, E4- superimposed on the midroot level.[6] AOT often causes expansion of surrounding bone and displacement of adjacent teeth;[7] the similar features were seen in our patient.

Radiographically, central AOT presents as well-defined, almost always unilocular radiolucency.[8] Fine intralesional calcification is seen within the lesion and is seen in approximately 78% of AOT.[9] Expansion of the cortical plate can be presented. However, Patil and Mishra reported a case of extrafollicular AOT with multilocular radiolucency.[10] In the present case, the lesion presented as bilocular radiolucency. As a result of tumor expansion, adjacent teeth may be displaced. Tooth displacement is more common than root resorption.[11] In our case, there was expansion of cortices with displacement of adjacent teeth. The most common treatment modality for the tumor is enucleation and curettage.[5] However, Patil and Mishra in their case treated extrafollicular AOT by surgical decompression.[10] In the present case, the lesion was treated with curettage. [Table 1] summarizes the literature review of extrafollicular AOT.
Table 1: Summary of the literature review of extrafollicular AOT

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   Conclusion Top

Extrafollicular AOT is an uncommon tumor and occurs less often than the follicular variant. Extrafollicular AOT is not associated with unerupted or impacted tooth and it should be included in the differential diagnosis of other odontogenic tumors appearing as unilocular radiolucencies. In this article we have presented uncommon exrafollicular variant of AOT that presented as gingival growth.

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.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Steensland HS. Epithelioma adamantinum. J Exper Med 1905;6:377-89.  Back to cited text no. 1
White S, Pharoah M. Oral Radiology Principles and Interpretation. 7th ed. Elsevier, Pennsylvania, Mosby; 2014.  Back to cited text no. 2
Reichart PA, Philipsen HP. Adenomatoid odontogenic tumor. In: Reichart PA, editor. Textbook of Odontogenic Tumors and Allied Lesion. London: Quintessence Publishing Co. Ltd.; 2004. p. 105-15.  Back to cited text no. 3
Yilmaz N, Acikgoz A, Celebi N, Zengin AZ, Gunhan O. Extrafollicular adenomatoid odontogenic tumor of the mandible: Report of a case. Eur J Dent 2009;3:71-4.  Back to cited text no. 4
Philipsen HP, Reichart PA. Adenomatoid odontogenic tumor: Facts and figures. Oral Oncol 1998;35:1-7.  Back to cited text no. 5
Philipsen HP, Samman N, Ormiston IW, Wu PC, Reichert PA. Variants of the adenomatoid odontogenic tumor with a note on tumor origin. J Oral Pathol Med 1992;21:348-52.  Back to cited text no. 6
Bravo M, White D, Miles L, Cotton R. Adenomatoid odontogenic tumor mimicking a dentigerous cyst. Int J Pediatr Otorhinolaryngol 2005;69:1685-8.  Back to cited text no. 7
Shafer HL. Textbook of Oral Pathology. 5th ed. Elsevier Publication, Mosby, Pennsylvania; 2006.  Back to cited text no. 8
Toida M, Hyodo I, Okuda T, Tatematsu N. Adenomatoid odontogenic tumor: Report of two cases and survey of 126 cases in Japan. J Oral Maxillofac Surg 1990;48:404-8.  Back to cited text no. 9
Patil PM, Mishra M. A rare case of extrafollicular adenomatoid odontogenic tumor in the posterior mandible and ramus treated by decompression. J Cranio Maxi Dis 2014;4:148-52.  Back to cited text no. 10
Neville BW. Update on current trends in oral and maxillofacial pathology. Head Neck Pathol 2007;1:75-80.  Back to cited text no. 11
Ninan T, Khanum N, Prathibha Rani RM, Basappa S. Extrafollicular adenomatoid tumour- A case report. RRJDS 2017;5:42-4.  Back to cited text no. 12
Mosavat F, Rashtchian R, Zeini N, Pour DG, Charlie SM, Mahdavi N. An extrafollicular adenomatoid odontogenic tumor mimicking a periapical cyst. Case Rep Radiol 2018. Article ID 6987050:1-5.  Back to cited text no. 13
Shivali V, Khanna VD, Khanna P, Singh A, Pandey A, Ahuja T. A rare case of extrafollicular adenomatoid odontogenic tumour in the posterior region of the mandible: Misdiagnosed as residual cyst. J Int Oral Health 2013;5:123-7.  Back to cited text no. 14
Khalid I, Devarakonda V, Raju KP, Ansari MA. Extrafollicular adenomatoid odontogenic tumour. J Contemp Dent 2012;2:25-8.  Back to cited text no. 15
Eroglu CN, Tunc SK, Gunhan O. A case of extrafollicular adenomatoid odontogenic tumor with long-term follow-up. J Case Rep Images Dent 2016;2:6-9.  Back to cited text no. 16
Kandasamy M, Mathew P, Dineshshankar J, Yoithapprabhunath TR. Extrafollicular adenomatoid odontogenic tumor of the maxilla: A case report. BAOJ Dent 2016;2:008.  Back to cited text no. 17
Sargolzaei S, Atarbashi-Moghadam S, Aghdashi F, Mansouri Nejad S. Extrafollicular cystic adenomatoid odontogenic tumor of the maxilla: A rare challenging case report with review of the literature. J Dent Mater Tech 2018;7:141-4.  Back to cited text no. 18


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

  [Table 1]


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