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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 29  |  Issue : 4  |  Page : 325-328

Adenomatoid Odontogenic Tumor: Intraosseous Variant–A Case Series and Review of Literature


1 Department of Oral Medicine and Radiology, Faculty of Dental Sciences, Shree Guru Gobind Singh Tricentenary University, Gurgaon, India
2 Department of Pedodontics, Faculty of Dental Sciences, Shree Guru Gobind Singh Tricentenary University, Gurgaon, India
3 Department of Oral Medicine and Radiology, Government Dental College, Rohtak, Haryana, India
4 Department of Oral Medicine and Radiology, Dental College and Research Centre, Teerthanker Mahaveer University, Moradabad, Uttar Pradesh, India

Date of Submission06-Apr-2016
Date of Acceptance15-Jan-2018
Date of Web Publication15-Feb-2018

Correspondence Address:
Dr. Lavina Taneja
Department of Oral Medicine and Radiology, Faculty of Dental Sciences, Shree Guru Gobind Singh Tricentenary University, Gurgaon, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.JIAOMR_40_16

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   Abstract 


Adenomatoid odontogenic tumor is an uncommon odontogenic lesion with a predilection for adolescent females and anterior maxilla. There is a controversy about it being a tumor or hamartoma. It appears in three clinicopathological variants – follicular, extrafollicular, and peripheral. Here, we report three cases of intraosseous adenomatoid odontogenic tumor.

Keywords: Adenomatoid odontogenic tumor, extrafollicular, follicular, intraosseous


How to cite this article:
Taneja L, Arya V, Gupta A, Sunil MK. Adenomatoid Odontogenic Tumor: Intraosseous Variant–A Case Series and Review of Literature. J Indian Acad Oral Med Radiol 2017;29:325-8

How to cite this URL:
Taneja L, Arya V, Gupta A, Sunil MK. Adenomatoid Odontogenic Tumor: Intraosseous Variant–A Case Series and Review of Literature. J Indian Acad Oral Med Radiol [serial online] 2017 [cited 2019 Aug 21];29:325-8. Available from: http://www.jiaomr.in/text.asp?2017/29/4/325/225556




   Introduction Top


Adenomatoid odontogenic tumor (AOT) accounts for approximately 3–7% of all odontogenic tumors. It was first described by Dreibladt in 1907 as a pseudoameloblastoma. In 1948, Stafne considered it as a separate entity. Earlier this tumor was represented by many nomenclatures such as adenoameloblastoma, ameloblastic adenomatoid tumor, adamantinoma, epithelioma adamantium, or teratomatous odontoma. Unol et al. published a list containing all nomenclatures for AOT.[1] Philipsen and Birn proposed the name adenomatoid odontogenic tumor in 1969 and suggested it as a distinct entity than ameloblastoma.[2] Interestingly, AOT is called a two-third tumor because two-third tumors occur in the maxilla, about two-third cases are reported in young females, and two-third cases are associated with unerupted canine.[3] Here, we present a case series of intraosseous AOT.


   Case Reports Top


Case report 1

A 15-year-old female patient reported to the department with the chief complaint of swelling in mouth for 2 months. The swelling was initially small with a gradual increase in size and was painless. On examination, there was a swelling on the palate extending from the maxillary right lateral incisor (12) to the maxillary right first premolar (14) region measuring approximately 1 × 2 cm in diameter [Figure 1]a. The overlying mucosa was normal and was noncompressible with no pulsations with minimal buccal cortical plate expansion, and over-retained deciduous right canine (53) was present which was discoloured with missing right maxillary permanent canine.
Figure 1: (a) Swelling on the palate extending from 11 to 14 region, (b) OPG revealing a well-defined radiolucency with respect to impacted 13 extending distally from coronal aspect of 13 and mesially apical to CEJ, and (c) Histopathological picture showing characteristic duct like structures lined by cuboidal cells with hyaline ring in the lumen

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The lesion was provisionally diagnosed as dentigerous cyst in relation to 13 with differential diagnosis of radicular cyst in 53. The orthopantomogram (OPG) [Figure 1]b revealed a well-defined radiolucency with respect to impacted maxillary right canine (13) extending distally from the coronal aspect of 13 and mesially apical to cementoenamel junction (CEJ) measuring approximately1 × 0.5 cm. The radiolucency was extending in the interdental region between 11 and 12 with tipping of root of 12 and resorption at the apical third. The radiographic diagnosis was follicular type of AOT with differential diagnosis of dentigerous cyst. The lesion was subjected to surgical enucleation and sent for histopathological evaluation, which confirmed the final diagnosis of AOT [Figure 1]c.

Case report 2

A 23-year-old male patient presented with a chief complaint of swelling in the upper left front region for 1.5 years. The swelling was slowly growing, and the patient reported history of nasal irritation of 1.5 months. There was no history of nasal discharge. On clinical examination, left side of the nose and cheek were swollen with obliteration of nasolabial fold with flaring of the nostril [Figure 2]a. The overlying skin was normal. There was a bulge in the left nostril. There were no neurologic deficiencies. Intraorally, a hard, nontender swelling obliterating the buccal vestibule with respect to the left maxillary lateral incisor to canine (22 to 24) region was observed along with mild palatal expansion. A missing maxillary left canine (23) and over-retained 63 were noted [Figure 2]b.
Figure 2: (a) Extraoral swelling on left side of nose and cheek with deviation of nose to right side, (b) Intraoral picture showing swelling obliterating the buccal vestibule with respect to 22-24 region and missing 23, and (c) The OPG showing a well-defined unilocular radiolucency in relation to impacted 23 and over-retained 63 with tipping of roots of 22

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A working diagnosis of dentigerous cyst with a differential diagnosis of follicular variant of AOT, keratocystic odontogenic tumor (KCOT), unicystic ameloblastoma, and ameloblastic fibroma were made. The OPG revealed a well-defined unilocular radiolucency in relation to impacted 23 and over-retained 63 with tipping of roots of 22 [Figure 2]c. Computed tomography (CT) revealed an expansile, lytic lesion on the left side of the maxilla causing cortical thinning and discontinuity in the lateral aspect of maxilla. Medially, the lesion appeared to involve the nasal cavity with erosion of nasal bone. Impacted right canine was noted with respect to the superior medial aspect of the lesion extending towards the lower margin of the nasal cavity [Figure 3]. The lesion was enucleated along with removal of impacted 23 and was sent for histopathological examination [Figure 4]. The final diagnosis of intraosseous AOT was thus made.
Figure 3: CT revealing an expansile, lytic lesion on left side of maxilla causing cortical thinning and discontinuity in the lateral aspect of maxilla. Medially involvement of the nasal cavity with the erosion of nasal bone can be seen. Impacted right canine was noted with respect to superomedial aspect of lesion

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Figure 4: Histopathological picture showed many duct-like structures lined by cuboidal or columnar cells with eosinophilic rimming towards the lumen

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Case report 3

A 14-year-old female patient presented with a chief complaint of swelling in the left upper front gum region since 1.5 months, which was gradually increasing in size. Patient also gave a history of increasing gap between the teeth since 1.5 years and mobile teeth in that region for 1 month. There was history of extraoral swelling of 4 days. There was no history of trauma. Extraoral examination revealed a diffuse swelling on the left middle-third of the face and upper lip region. Intraorally, swelling obliterating the buccal vestibule showed blanched mucosa, erythematous marginal gingiva and spacing between 21 and 22 [Figure 5]. The swelling was tender and firm in consistency. The maxillary left lateral incisor (22) showed grade 2 mobility with deep periodontal pockets. A provisional diagnosis of lateral periodontal abscess with differential diagnosis of dentigerous cyst with supernumerary teeth and extrafollicular AOT were made.
Figure 5: Intraoral picture of swelling obliterating the buccal vestibule and spacing between 21 and 22

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Intraoral periapical radiograph (IOPA), anterior occlusal, and OPG radiographs were advised. The IOPA revealed pear-shaped well-defined radiolucency between diverging roots of 21 and 22 with superior corticated border [Figure 6]a. The anterior occlusal radiograph revealed tipping of 22 but no cortical expansion [Figure 6]b. OPG also revealed the same changes along with other findings i.e., congenitally missing 41 and over-retained 81 with otherwise normal chronological dental pattern [Figure 6]c. Excisional biopsy along with curettage was done and subjected to histopathological evaluation [Figure 7], which confirmed the final diagnosis of extrafollicuar AOT.
Figure 6: (a) IOPA revealing pear shaped well-defined radiolucency between diverging roots of 21 and 22 with superior corticated border, (b) Anterior occlusal radiograph revealing tipping of 22 but no cortical expansion, and (c) OPG also revealing the same changes along with other findings i.e., congenitally missing 41 and over-retained 81 with otherwise normal chronological dental pattern

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Figure 7: Histopathological picture showing proliferating glandular epithelium with duct-like structure lined by cells showing reversal of polarity

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   Discussion Top


AOT is a benign, slow growing noninvasive odontogenic tumor of epithelial origin. It is encountered mostly in young patients, especially in the second decade with a female predilection.[4] Anterior maxillary region is the most predominant site of occurrence. In our cases, all three were in the maxillary anterior region. There are three variants of AOT, i.e., central or intraosseous type which radiographically can be divided as follicular (73%) associated with impacted tooth and extrafollicular type (24%) or which have no connection with tooth and extraosseous or peripheral type (3%).[5] In our cases, 2 were follicular and one was extrafollicular.

The origin of AOT is controversial. There is consensus that it is odontogenic in origin as it occurs in the tooth bearing area and cytological features are the same as various components of enamel organ, dental lamina, reduced enamel epithelium or their remnants.[6] There is an unresolved controversy about it being a true neoplasm or a hamartoma. The point in favor of its neoplastic origin include considerable size of the lesion attained in some of the reported cases [7],[8],[9] similar to case 2 of ours which attained large size, and greater histopathological variation in the arrangement of odontogenic apparatus as would be expected from a dental anomaly. However, the lack of recurrence and limited size attained in a majority of cases goes in favor of hamartoma.[10],[11],[12] None of our cases showed recurrence and cases 1 and 3 were of limited size.

Radiographically, most AOTs are well-demarcated with almost always unilocular radiolucency of smooth corticated and sometimes sclerotic borders. Most follicular variants show pericoronal or juxtracoronal radiolucency which may extend apical to CEJ on atleast one side of the root.[13] Occasionally, scalloped border is observed. Maxillary canine is the most commonly involved tooth. Root resorption is rarely seen though divergence of adjacent roots and displacement of teeth may be observed as seen in case 1. The differential diagnosis given in such cases is dentigerous cyst but the attachment apical to CEJ can give a clue to the diagnosis. Both of cases 1 and 2 were of the follicular variety with involvement of an impacted canine. Case 2 showed huge size with a rare presentation of displacement of the impacted canine till the nasal wall along with perforation of the anterior wall of maxillary sinus.

The extrafollicular type usually present as a unilocular, well-defined radiolucency between or above or superimposed on the roots of erupted teeth. Differential diagnosis given in such case can be residual cyst, radicular cyst, or lateral periodontal cyst. Discrete or flocculent radiopaque foci are observed in two-third cases of AOT and IOPA can show these changes superior to OPG.[14] However, none of our cases showed the flocculence.


   Conclusion Top


AOT should be considered in the differential diagnosis of unilocular radiolucency in anterior jaw in young adults and careful clinical and radiographic evaluation can lead to correct diagnosis of this commonly mimicking entity.

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.
Lucas RB. Pathology of Tumours of Oral Tissues. 4th edition. Edinburgh, Scotland: Churchill Livingstone; 1984. p. 66.  Back to cited text no. 1
    
2.
Philipsen HP, Birn H. The adenomatoid tumour, ameloblastic adenomatoid tumour or adenoameloblastoma. Acta Pathol Microbiol Scand 1969;75:375-98.  Back to cited text no. 2
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3.
Marx RE, Stern D. Oral and Maxillofacial Pathology: A Rationale for Diagnosis and Treatment. Hanover Park: Quintessence Publishing; 2003. p. 609-12.  Back to cited text no. 3
    
4.
Rick GM. Adenomatoid odontogenic tumour. Oral Maxillofac Surg Clin North Am 2004;16:333-54.  Back to cited text no. 4
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5.
Chettopadhyay A. Adenomatoid odontogenic tumour; Review of literature and report of 30 cases from India. Indian J Dent Res 1994;5:89-95.  Back to cited text no. 5
    
6.
Garg D, Palaskar S, Shetty VP, Anju B. Adenomatoid odontogenic tumour - Hamartoma or true neoplasm: A case report. J Oral Sci 2009;51:155-9.  Back to cited text no. 6
    
7.
Lee JK, Lee KB, Hwang BN. Adenomatoid odontogenic tumour: A case report. J Oral Maxillofac Surg 2000;58:1161-4.  Back to cited text no. 7
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8.
Raubenheimer EJ, Seeliger JE, van Heerden WF, Dreyer AF. Adenomatoid odontogenic tumour: A report of two large lesions. Dentomaxillofac Radiol 1999;120:43-5.  Back to cited text no. 8
    
9.
Shetty K, Vastardis S, Giannini P. Management of an unusually large adenomatoid odontogenic tumour. Oral Oncol Extra 2005;41:316-8.  Back to cited text no. 9
    
10.
Philipsen HP, Reichert PA. Adenomatoid odontogenic tumour: Facts and figures. Oral Oncol 1998;35:125-31.  Back to cited text no. 10
    
11.
Rajendran R, Sivapadasundaram B. Shafer's Text Book of Oral Pathology. 6th edition. Noida, India: Churchill Livingstone, Elsevier Publications; 2009. p. 282-3.  Back to cited text no. 11
    
12.
Kramer IRH, Pindborg JJ, Shear M. WHO International Histological Classification of Tumour. Histological typing of odontogenic tumours. 2nd edition. Berlin: Springer Vering; 1992.  Back to cited text no. 12
    
13.
Reichart PA, Philipsen HP. Odontogenic Tumour and Allied Lesions. London: Quintessence; 2004.  Back to cited text no. 13
    
14.
Dare A, Yamaguchi A, Yoshiki S, Okano T. Limitation of panoramic radiography in diagnosing adenomatoid odontogenic tumour. Oral surg Oral Med Oral Pathol Oral Radiol Endod 1994;77:662-8.  Back to cited text no. 14
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    Figures

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



 

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