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Year : 2022  |  Volume : 65  |  Issue : 3  |  Page : 728-730
Extrafollicular adenomatoid odontogenic tumor: An infrequent case report

1 Regional Medical Advisor, Abbott India Limited, Faridabad, Haryana, India
2 Senior lecturer, Manav Rachna Dental College, Faridabad, Haryana, India
3 Consultant Periodontist, Delhi, India
4 Senior Resident, Safdarjung Hospital, Delhi, India

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Date of Submission19-Apr-2021
Date of Decision20-Aug-2021
Date of Acceptance24-Aug-2021
Date of Web Publication21-Jul-2022

How to cite this article:
Sharma J, Varshney A, Raina AB, Chopra D. Extrafollicular adenomatoid odontogenic tumor: An infrequent case report. Indian J Pathol Microbiol 2022;65:728-30

How to cite this URL:
Sharma J, Varshney A, Raina AB, Chopra D. Extrafollicular adenomatoid odontogenic tumor: An infrequent case report. Indian J Pathol Microbiol [serial online] 2022 [cited 2022 Aug 15];65:728-30. Available from: https://www.ijpmonline.org/text.asp?2022/65/3/728/351601

Dear Editor,

Adenomatoid odontogenic tumor (AOT) is an uncommon, hamartomatous, and asymptomatic benign noninvasive lesion of odontogenic origin that was first described by Driebaldt in 1907 as a pseudo-adenoameloblastoma.[1] In 1948, Stafne considered AOT as a distinct entity whereas others believed it to be a variant of ameloblastoma.[2] Philipsen and Birn in 1969 declined this thought and suggested the name “adenomatoid odontogenic tumor.”[3] The World Health Organization (WHO) in 1971 adopted the term “adenomatoid odontogenic tumor.” In 2003, Max and Stern coined the name “adenomatoid odontogenic cyst.”[4] There are three clinical variants: the follicular variant (F) is intraosseous and is associated with impacted and displaced tooth; the extrafollicular variant (subtype: E1, E2, E3, and E4) is within the bone but is not associated with unerupted tooth, rather in between erupted tooth mimicking a radicular cyst or lateral periodontal cyst; and the peripheral (epulis-like) variant, which exhibits a periodontal bone defect or ectopic growth.[5]

A 33-year-old male patient reported with a chief complaint of painless swelling in the upper-left front-tooth region since 1–2 months. The swelling increased in size within a short span of time. The patient revealed no history of pain or pus discharge associated with the swelling. Extraorally, the swelling was not obvious and did not produce any facial asymmetry. On palpation, it was nontender, firm in consistency, noncompressible, nonreducible, and nonfluctuant. Intraoral examination revealed a swelling in the left maxillary front region, extending from 21 to 24 (FDI tooth numbering system), and approximately 2 × 2 cm in size. The overlying mucosa was of normal color and appearance [Figure 1]. On palpation, the swelling was firm, nontender, nonmobile, and nonpulsatile. An incomplete root canal treatment was done in relation to 21, 22, and 23 but the swelling did not decrease, which was observed for at least one month. A provisional diagnosis of the radicular cyst and differential diagnosis of odontogenic keratocyst, unicystic ameloblastoma, and adenomatoid odontogenic tumor was given.

Radiographic examination revealed a large unilocular radiolucent area with a well-defined border extending from 21 to 24 region with slight root resorption [Figure 2]. An incisional biopsy was done and the specimen was sent for histopathological examination. Macroscopic examination revealed a tan-colored, roughly spherical-shaped tumor mass, which was firm in consistency. Histopathological examination revealed a solid nodule surrounded by a fibrous capsule with minimal connective tissue stroma [Figure 3]. The odontogenic columnar epithelial cells were arranged in the form of numerous rosettes, cords, duct-like structures, and central lumen was filled with eosinophilic amorphous material (often described as “tumor droplets”) [Figure 4] and [Figure 5]. The histopathological features confirmed the diagnosis of AOT. The patient was under follow-up for 2 years without recurrence.
Figure 1: Normal overlying mucosa

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Figure 2: Well-defined border extending from 21 to 24 region with slight root resorption

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Figure 3: Solid nodule surrounded by a fibrous capsule with minimal connective tissue stroma (H&E)

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Figure 4: The odontogenic columnar epithelial cells were arranged in the form of numerous rosettes, cords, duct-like structure

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Figure 5: Central lumen was filled with eosinophilic amorphous material

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Adenomatoid odontogenic tumor (AOT) is an uncommon tumor of odontogenic origin that represents 3% of all odontogenic tumors and is often misdiagnosed as an odontogenic cyst.[6] It is predominantly found in young female patients, located more often in the maxilla. In most cases, it is associated with an unerupted permanent tooth,[7] but in our case, the patient was a 33-year-old man. Its occurrence in the maxilla is twice as frequent as that in the mandible, and the anterior part of the jaw is more frequently involved than the posterior part, as in the present case. An unerupted maxillary canine is the tooth most commonly associated with AOT.[8] However, in our case, it was not associated with an unerupted tooth. The AOT has three clinicopathologic variants, namely intraosseous follicular, intraosseous extrafollicular, and extraosseous/peripheral. The intraosseous follicular type (accounting for 73% of all AOT cases) is associated with an impacted tooth, whereas intraosseous extrafollicular type (accounting for 24%) has no relation with an impacted tooth, as in the case we present here; the peripheral variant (accounting for 3%) is attached to the gingival structures.[9] The tumors are usually asymptomatic and are small (1.5–3 cm),[7] as in the present case.

Radiographically, unilocular radiolucency with a distinct radiopaque border is a characteristic feature of AOT. It is usually associated with the displacement of teeth. Root resorption is seldom seen,[10] but slight resorption was reported in our case. The histological findings for AOT are remarkably similar in the literature. The histological features of the tumor were described as a tumor of odontogenic epithelium with duct-like structures and with varying degrees of inductive changes in the connective tissue. The tumor may be partly cystic and in some cases, the solid lesion may be present only as masses in the wall of a large cyst. The tumor may contain pools of amyloid-like material and globular masses of calcified material.[7] Our case was consistent with these common features reported in the literature. Conservative surgical enucleation and curettage of the lesion are the treatment of choice. Recurrence of the tumor is very rare with an excellent prognosis.[10]

The purpose of this case study is to increase the existing knowledge of this rare entity as it is often misdiagnosed by clinicians due to low incidence and uncommon presentation of the disease and should be confirmed by histopathological examination.

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

Batra P, Prasad S, Parkash H. Adenomatoid odontogenic tumour: Review and case report. J Can Dent Assoc 2005;71:250-3.  Back to cited text no. 1
Harbitz F. On cystic tumors of the maxilla, and especially on adamantine cystadenomas (adamantomas). Dent Cosmos 1915;15:1081-93.  Back to cited text no. 2
Stafne EC. Epithelial tumors associated with developmental cysts of the maxilla: Report of 3 cases. Oral Surg Oral Med Oral Pathol 1948;1:887-94.  Back to cited text no. 3
Philipsen HP, Birn H. Adenomatoid odontogenic tumour, Ameloblastic Adenomatoid tumor or adenoameloblastoma. Acta Pathol Microbiol Scand 1969;75:375-98.  Back to cited text no. 4
Pati P, Debata T, Das BK, Das SN. Atypical presentation of extra-follicular adenomatoid odontogenic tumor of anterior maxilla: A clinical predicament. Int J Sci Stud 2016;4:261-3.  Back to cited text no. 5
Philipsen HP, Srisuwan T, Reichart PA. Adenomatoid odontogenic tumor mimicking a periapical (radicular) cyst: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:246-8.  Back to cited text no. 6
Reddy Kundoor VK, Maloth KN, Guguloth NK, Kesidi S. Extrafollicular adenomatoid odontogenic tumor: An unusual case presentation. J Dent Shiraz Univ Med Sci 2016;17:370-4.  Back to cited text no. 7
Philipsen HP, Reichart PA. Adenomatoid odontogenic tumour: Facts and figures. Oral Oncol 1999;35:125-31.  Back to cited text no. 8
Swasdison S, Dhanuthai K, Jainkittivong A, Philipsen HP. Adenomatoid odontogenic tumors: An analysis of 67 cases in a Thai population. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:210-5.  Back to cited text no. 9
Panjwani S, Bagewadi A, Keluskar V. An unusual site of Adenomatoid Odontogenic Tumor: A rare case report. J Int Oral Health 2010;2:71-6.  Back to cited text no. 10

Correspondence Address:
Jaishree Sharma
856, Dr Mukherjee Nagar, Delhi - 110 009
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijpm.ijpm_388_21

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


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