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Journal of Indian Society of Pedodontics and Preventive Dentistry Official publication of Indian Society of Pedodontics and Preventive Dentistry
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CASE REPORT
Year : 2016  |  Volume : 34  |  Issue : 3  |  Page : 291-293
 

An adenomatoid odontogenic tumor in disguise


1 Department Oral and Maxillofacial Surgery, Hitkarini Dental College and Hospital, Jabalpur, Madhya Pradesh, India
2 Department Pedodontics, Hitkarini Dental College and Hospital, Jabalpur, Madhya Pradesh, India

Date of Web Publication25-Jul-2016

Correspondence Address:
Ankit Sharma
Department Oral and Maxillofacial Surgery, Hitkarini Dental College and Hospital, Jabalpur, Madhya Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.186752

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   Abstract 

Adenomatoid odontogenic tumor (AOT) is a slowly growing benign tumor of the oral cavity. It accounts of 3-7% of all odontogenic tumors. It is seen to occur commonly in the anterior maxilla. The tumor is usually associated with an impacted tooth with maxillary canine being the most common tooth. AOT is seen in a younger group, especially below 20 years with a female preponderance. AOT occurs in two main variants: Central or intraosseous which is more common and second is peripheral which is rare. Radiologically, it represents as a radiolucent lesion with radiopaque foci usually an impacted or a supernumerary tooth. With a close clinical and radiographic resemblance to dentigerous cyst correct diagnosis and treatment is necessary. AOT being benign in nature requires a conservative management with enucleation. This is a case report of a 13-year-old girl with an AOT occurring in the anterior maxillary region.


Keywords: Adenomatoid odontogenic tumor, anterior maxilla, dentigerous cyst


How to cite this article:
Dhirawani RB, Pathak S, Mallikaarjuna K, Sharma A. An adenomatoid odontogenic tumor in disguise. J Indian Soc Pedod Prev Dent 2016;34:291-3

How to cite this URL:
Dhirawani RB, Pathak S, Mallikaarjuna K, Sharma A. An adenomatoid odontogenic tumor in disguise. J Indian Soc Pedod Prev Dent [serial online] 2016 [cited 2021 Jan 21];34:291-3. Available from: https://www.jisppd.com/text.asp?2016/34/3/291/186752



   Introduction Top


Adenomatoid odontogenic tumor (AOT) is a benign neoplasm with a female preponderance and occurs commonly in maxillary bone. It occurs in two forms: Intraosseous and peripheral.[1] It is seen in dentate segment of the jaw bone and usually shows cytologic resemblance with the dental organ component mainly enamel organ and the remnant of Hertwig's epithelial root sheath. Most commonly associated tooth with the tumor is maxillary cuspid.[2] We report a case of AOT which is mistaken as a dentigerous cyst in a 13-year-old girl.


   Case Report Top


A 13-year-old girl reported to the Department of Oral and Maxillofacial Surgery with swelling on the left side of the face for the past 3 months. The swelling was slowly growing but was not associated with pain. Extraorally, swelling was moderate and diffuse on the left side of nasolabial fold region extending from 1 cm below lower eyelid to left upper lip superioinferiorly and from ala of the nose to the middle of ala-tragal line anteroposteriorly. The size of swelling was approximately 3 cm × 2 cm. The color of overlying skin swelling was normal, and there was the obliteration of nasolabial fold [Figure 1]. On palpation, swelling was firm, nontender and smooth. Intraorally, there was an expansion of labial cortical plate, along with the presence of over-retained deciduous lateral incisor and canine. Intraorally, swelling extended from left central incisor to second premolar region anteroposteriorly [Figure 2]. An orthopantomogram was done which revealed a radiolucent area with an impacted lateral incisor and a canine superior to radiolucency. The radiolucency was having a radioopaque border along with extension from distal of central incisor to mesial of first premolar region [Figure 3].
Figure 1: Swelling with left side of face at nasolabial fold region

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Figure 2: Swelling on left side with obliteration of labial vestibule

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Figure 3: Panoramic radiograph showing radioleucency on the left side with an impacted lateral incisor

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Aspiration with an 18-gauge needle was done, and straw-colored fluid was seen. A provisional diagnosis of the dentigerous cyst was made, and an incisional biopsy was done with marsupialization of the lesion. The histopathological report of the lesion revealed it to be an AOT. Under general anesthesia, a crevicular triangular flap with the distal release was raised and the lesion was excised along with the lateral incisor [Figure 4]. The involved canine was also removed and peripheral ostectomy was done [Figure 5]. Hemostasis was achieved and primary closure was done using a resorbable 3-0 vicryl.
Figure 4: Tumor mass encapsulating lateral incisor and extracted maxillary cuspid

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Figure 5: Operated site after enucleation and peripheral ostectomy

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   Discussion Top


AOT was first reported by Harbitz from Norway in year 1915 and named it “adamantoma.” Later Philipsen and Birn proposed the name “AOT” in 1969.[3] AOT is slow growing and a noninvasive neoplasm of odontogenic origin with low recurrence rate. It accounts of 3-7% of all odontogenic tumors.[4] It has been reported to occur from 3 to 82 years of age with more than two-thirds (69%) are diagnosed between the ages of 10 and 19 years; more than half (53%) are diagnosed in teenagers; 21% are diagnosed between ages 20 and 29; and altogether, 88% are diagnosed in the second and third decades;[3] and with a female to male ratio approximately 1.9:1. AOT is also known as “tumor of two-thirds” as its incidence is two-thirds cases in the maxilla, of all cases 2/3 females are involved and in 2/3 cases it is associated with the unerupted tooth.[4]

AOT occurs in two forms: Intraosseous and peripheral forms. Intraosseous forms are further of two types radiologically: Follicular and extrafollicular types.[3]

Intraosseous forms account for 96% of all AOT.[1]

Follicular type exists as a smooth round radiolucent lesion with an impacted tooth associated with it. It may initially be misdiagnosed as dentigerous cysts. Approximately 77% of the follicular variant are provisionally seen to be diagnosed as dentigerous cysts.

Extrafollicular type is not associated with an impacted tooth and present between the two roots.[3] Apart from the dentigerous cyst, AOT should be differentially diagnosed from other lesions including odontogenic keratocyst, ameloblastic fibroma, odontogenic myxoma, or a central giant cell lesion. With radiopaque calcifications seen on radiographs, this lesion may be suggestive of ameloblastic fibro-odontoma, ossifying fibroma, or calcifying epithelial odontogenic tumor (CEOT). However, CEOT is uncommon among first and second decades of life.[4]

Distinguishing features between adenomatoid odontogenic tumor, and dentigerous cyst

Both AOT and dentigerous cyst arise in association with teeth as pericoronal radiolucency. Dentigerous cyst encloses only a coronal part of the impacted tooth more commonly in the third molar region. Whereas, AOT may enclose both a coronal and radicular part of the tooth and sometimes the tooth as a whole.[5] About 30% of the intraosseous AOTs are not pericoronal. Rather they demonstrate a radicular relationship to adjacent teeth which may range from lateral or periapical to no relationship at all and 89% extracoronal cases are associated with maxillary canines.[3] AOT are usually seen in young teens with female predilection usually below 20 years, whereas a dentigerous cyst seen in second to the third decade of life with male predilection.[2] AOT is usually 1-3 cm rarely may enlarge up to 7 cm. A normal dental follicular space is approximately 3-4 mm and a follicular space of 5 mm or more is suspicious of a dentigerous cyst. Furthermore, AOT may displace adjacent teeth whereas dentigerous cyst may not displace the adjacent teeth until attain a large size.[5] AOT is mostly associated with an impacted canine and commonly in anterior maxilla,[1] whereas dentigerous cyst are associated mostly with impacted third molars and maxillary canine.[6]

AOT usually presents as a well-encapsulated mass with a central solid mass or a small cyst like space. Marsupialization has been considered for the lesion as an attempt to allow impacted tooth to erupt but as it originates from Hertwig's epithelial root sheath, the tooth is completely encapsulated in the lumen of the lesion. That is why surgically it becomes impossible as no or little radicular bone support is present with the impacted tooth.[4] In only three out of 750 cases reported by Philipsen and Reichart recurrence has been seen.[7] Because of its low recurrence rate, fibrous encapsulation and benign nature, enucleation with peripheral ostectomy provides with success in treating these lesion.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Bartake AR, Punnya VA, Sudeendra P, Rekha K. Two adenomatoid odontogenic tumours of the maxilla: A case report. Br J Oral Maxillofac Surg 2009;47:638-40.  Back to cited text no. 1
    
2.
Rajendran R. Shafer's textbook of oral pathology. 7th ed. Elsevier India; 2009.  Back to cited text no. 2
    
3.
Rick GM. Adenomatoid odontogenic tumor. Oral Maxillofac Surg Clin North Am 2004;16:333-54.  Back to cited text no. 3
    
4.
Marx RE, Stern D. Odontogenic and nonodontogenic cysts. Oral and maxillofacial pathology: A rationale for diagnosis and treatment. Quintessence Publishing Chicago; Hanover Park, Ill, USA. “Odontogenic and nonodontogenic cysts,” 2003. p. 607.  Back to cited text no. 4
    
5.
Lee JK, Lee KB, Hwang BN. Adenomatoid odontogenic tumor: A case report. J Oral Maxillofac Surg 2000;58:1161-4.  Back to cited text no. 5
    
6.
Patel V, Sproat C, Samani M, Kwok J, McGurk M. Unerupted teeth associated with dentigerous cysts and treated with coronectomy: Mini case series. Br J Oral Maxillofac Surg 2013;51:644-9.  Back to cited text no. 6
    
7.
Reichert PA, Philipensen HP. Ossifying fibroma. Odontogenic tumor and allied lesions. Surrey: Quintessence; 2004. p. 273-80.  Back to cited text no. 7
    


    Figures

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



 

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