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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 29  |  Issue : 4  |  Page : 329-332

Extrafollicular Adenomatoid Odontogenic Tumor: A Case Report


1 Department of Oral Medicine and Radiology, Chhatrapati Shahu Maharaj Shikshan Sanstha Dental College and Hospital, Aurangabad, Maharashtra, India
2 Department of Oral Medicine and Radiology, Kusum Devi Sunderlal Dugar Jain Dental College and Hospital, Kolkata, West Bengal, India

Date of Submission13-Sep-2016
Date of Acceptance07-Dec-2017
Date of Web Publication15-Feb-2018

Correspondence Address:
Dr. Sneha H Choudhary
Department of Oral Medicine and Radiology, Kusum Devi Sunderlal Dugar Jain Dental College and Hospital, Kolkata, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.JIAOMR_116_16

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   Abstract 


Odontogenic tumors are a group of heterogeneous lesions. Adenomatoid odontogenic tumor (AOT) is a benign, hamartomatous, slow-growing lesion, which is usually located in the anterior maxilla and is more prevalent in females. Although the follicular type is more frequently described in literature, here we report a case of extrafollicular type of AOT in the maxillary anterior region in a 16-year-old female.

Keywords: Adenomatoid odontogenic tumor, anterior maxillary swelling, extrafollicular adenomatoid odontogenic tumor


How to cite this article:
Zaidi NS, Kale LM, Choudhary SH, Kadam V. Extrafollicular Adenomatoid Odontogenic Tumor: A Case Report. J Indian Acad Oral Med Radiol 2017;29:329-32

How to cite this URL:
Zaidi NS, Kale LM, Choudhary SH, Kadam V. Extrafollicular Adenomatoid Odontogenic Tumor: A Case Report. J Indian Acad Oral Med Radiol [serial online] 2017 [cited 2019 May 22];29:329-32. Available from: http://www.jiaomr.in/text.asp?2017/29/4/329/225465




   Introduction Top


Adenomatoid odontogenic tumor (AOT) is a relatively uncommon distinct odontogenic neoplasm.[1] AOT is also called as “master of disguise.”[2] Steensland was the first who described AOT in 1905. Initially, various terminologies such as adenoameloblastoma, adamantinoma, ameloblastic adenomatoid tumor, ameloblastic adenomatoid tumor or epithelioma adamantinum were used for AOT.[1] However, Philipsen and Birn in 1969 coined the term AOT, which was adopted by the first edition of the World Health Organization classification of odontogenic tumors in 1971.[3] Three types of AOT have been described – follicular, extrafollicular, and peripheral.[4] The follicular type accounts for 73% of AOT cases and is associated with an unerupted tooth. The extrafollicular type is not associated with an impacted tooth, and accounts for approximately 24% of AOT cases. The peripheral type accounts for 3% of the cases and affects the gingival mucosa.[5]


   Case Report Top


A 16-year-old female patient reported to the department of oral medicine and radiology with the chief complaint of swelling in her upper front region of jaw since 2 years. Patient's history revealed that the swelling was smaller in size at initial presentation and had gradually increased in size over the period of 2 years to the present condition. Patient gave no history of trauma or pain in the same region. Extraoral examination revealed facial asymmetry due to presence of bulging of upper lip on left side [Figure 1]. Single, mobile, nontender, and firm submandibular lymph node palpable on left side of size approximately 1.5 × 2 cm was noted.
Figure 1: Extraoral clinical picture showing bulging of upper lip on left side

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Intraoral examination revealed a single well-defined round-to-ovoid shaped swelling present on the labial and palatal aspect of 21 and 22 involving the interdental area of size approximately 2.5 × 3 cm extending mesiodistally from the mesial aspect of 21 to the mesial aspect of 23 and superoinferiorly from the labial vestibule region till the middle one third of crown of 21 and incisal one-third of crown of 22 involving marginal gingiva, interdental papilla, and attached gingiva. Overlying mucosa appeared normal pink in color [Figure 2]a and [Figure 2]b. On palpation, the swelling was nontender, firm-to-hard in consistency on the labial aspect of 21 and 22, and soft to firm in consistency on the inferior aspect. Midline shifted to the right side, mesially displaced and slightly extruded 21, and distally displaced and rotated 22 [Figure 3] were noted. However, both 21 and 22 were not mobile. Electric pulp testing was done with 21, 22, 23, and 11 teeth, which revealed delayed response with 21 and 22, whereas 11 and 23 showed normal response.
Figure 2: (a) Intraoral clinical picture showing swelling on labial aspect of 21 and 22, and (b) Showing swelling on palatal aspect of 21 and 22

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Figure 3: Intraoral clinical picture showing midline shift to right side, mesially displaced and slightly extruded 21, and distally displaced and rotated 22

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On the basis of history and clinical examination, a provisional diagnosis of peripheral ossifying fibroma in 21 and 22 region was made. Differential diagnosis considered were AOT (extrafollicular type), compound odontome, developmental periodontal cyst, dentigerous cyst with supernumerary teeth, and calcifying odontogenic cyst. Investigations advised were fine needle aspiration, intraoral periapical radiograph of 21 and 22 region, and maxillary occlusal radiograph. Aspiration from the swelling was nonproductive. Intraoral periapical radiograph (IOPA) of 21 and 22 region showed single well-defined corticated radiolucency present in the interdental area of 21 and 22 with multiple small radiopaque foci, which was suggestive of calcification. Mesial displacement of 21 was seen along with distal displacement of 22. Severe bone loss was seen in the distal aspect of root of 21 and mesial aspect of root of 22 up to the periapex area. Ghost image of the nose ring was seen as a single radiopacity in the periapical area mesial to the root of 22 [Figure 4]. Maxillary anterior topographic occlusal radiograph revealed a solitary well-defined round-shaped corticated radiolucency in the interdental area of 21 and 22 region. Multiple small radiopaque foci were seen within the radiolucency suggestive of calcification. Labial cortical plate expansion was seen in 21 and 22 region. Mesially displaced 21 and distally displaced 22 were seen. Severe bone loss was seen in the distal aspect of root of 21 and mesial aspect of root of 22 up to the periapex area. Ghost image of the nose ring was seen as single radiopacity overlapping the crown of 22 [Figure 5].
Figure 4: IOPA of 21 and 22 region

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Figure 5: Maxillary anterior topographic occlusal radiograph

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Thus, a working diagnosis of AOT (extrafollicular type – E2)[6] involving 21 and 22 region was established. Local surgical excision of the lesion with extraction of 21 and 22 was done because both these teeth showed delayed response on electric pulp testing and the radiograph revealed severe bone loss. The excised specimen was sent for histopathological examination. The histologic section showed capsulated tumor consisting of variable size of solid nodules of odontogenic epithelium, forming nests with minimal stromal connective tissue. Cellular areas showed duct-like structures lined by a single row of columnar epithelial cells along with areas of calcification [Figure 6]. Thus, a diagnosis of AOT (Extrafollicular type- E2) involving 21 and 22 region was confirmed. Patient is on regular follow-up, and there is no sign of recurrence [Figure 7].
Figure 6: Histopathological picture of the lesion

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Figure 7: Postoperative follow-up

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   Discussion Top


AOT was first described in 1905 by Steensland,[1],[7] when it was named pseudo-adenoameloblastoma.[5] Staphne in 1948 recognized that the lesion shows different pathological structures.[5] Various terms have been used in literature to describe AOT such as adenoameloblastoma, adamantinoma, ameloblastic adenomatoid tumor, ameloblastic adenomatoid tumor, or epithelioma adamantinum. In 1969, Philipsen and Birn suggested that AOT is not a variant of ameloblastoma but a separate entity and proposed the term AOT.[1] It accounts for 2.2–7.1% of all odontogenic tumors. Till date, more than 750 cases have been reported in literature.[3]

AOT is an uncommon, asymptomatic, benign, slow growing tumor, which has marked predilection for anterior maxillary region. It is more commonly found in young patients with two-third of all cases occurring between the age group 10 and 19 years of age. It is more common in females than males with incidence of almost 2:1.[1] This tumor is sometimes referred to as “two-thirds tumor” because it occurs in the maxilla in about two-third cases, about two-third cases arise in young females, two-third cases are associated with an unerupted tooth, and two-third affected teeth are canines.[3] Here, we have reported a case of extrafollicular type of AOT in a 16-year-old female in the anterior maxillary region, which was not associated with impacted tooth.

The origin of AOT is still controversial. Most of the authors believe it to be odontogenic in origin as it occurs within the tooth bearing areas of the jaws and has cytological features similar to enamel organ, dental lamina, reduced enamel epithelium, and/or their remnants suggesting it to be a neoplastic growth; whereas others support the idea that the lesion is a developmental outgrowth or hamartoma due to the relative size of the tumor and lack of recurrence.[3]

Mostly the lesions are asymptomatic and measure 1.5–3 cm, however, larger lesions of more than 7 cm may occur.[3] In the present case, the lesion was almost 3 cm in size. The main types of AOT include follicular, extrafollicular, and peripheral, among which the follicular is the most common that appears as well-defined round or ovoid-shaped radiolucency associated with the crown of unerupted tooth, most often canine. The radiolucency of the extrafollicular type of AOT is located either between, above, or superimposed upon the roots of erupted permanent teeth.[3],[7] Similarly, in the present case, the well-defined radiolucency of the lesion was seen in the interdental area of 21 and 22. It also contains a minimal amount of radiopacities described as flecks, snowflake, as was seen in the present case.[3] Often the peripheral type show slight erosion of the alveolar bone crest, however, radiographic changes are difficult to detect. Displacement of the adjacent teeth due to tumor expansion is much more common than root resorption,[3] as was seen in the present case.

Conservative surgical excision is the treatment of choice for AOT because of the presence of capsule.[3],[5] In the present case, local surgical excision along with extraction of 21 and 22 was done. Recurrence of AOT is rare. Only three cases in Japanese patients are reported, in which the recurrence of this tumor occurred; therefore, the prognosis is excellent when the lesion is completely removed.

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.
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;5:124-8.  Back to cited text no. 1
    
2.
Mutalik VS, Shreshtha A, Mutalik SS, Radhakrishnan R. Adenomatoid odontogenic tumor: A unique report with histological diversity. J Oral Maxillofac Pathol 2012;16:118-21.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Shah H, Parikh N, Nandini C, Pate A. Extrafollicular adenomatoid odontogenic tumor of Maxilla: A case report. Int J Oral Res 2015;1:27-34.  Back to cited text no. 3
    
4.
Philipsen HP, Reichart PA, Zhang KH, Nikai H, Yu QX. Adenomatoid odontogenic tumor: Biologic profile based on 499 cases. J Oral Pathol Med 1991;20:149-58.  Back to cited text no. 4
[PUBMED]    
5.
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. 5
    
6.
Friedrich RE, Scheuer HA, Zustin J. Adenomatoid odontogenic tumor (AOT) of maxillary sinus: Case report with respect to immunohistochemical findings. In Vivo 2009;23:111-6.  Back to cited text no. 6
[PUBMED]    
7.
Katpar S, Mehwash K, Naghma A, Rashid QN. Maxillary Adenomatoid odontogenic tumour-An uncommon oral pathology, Reported locally. J Liaquat Univ Med Health Sci 2010;9:155-8.  Back to cited text no. 7
    


    Figures

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



 

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