|Year : 2013 | Volume
| Issue : 1 | Page : 50-53
A rare occurrence of adenomatoid odontogenic tumor arising from cystic lining in the mandible: Review with a case report
Harish Saluja1, Vikrant Kasat2, Prafful Gaikwad1, Uma Mahindra1, Vipin Dehane1
1 Department of Oral and Maxillofacial Surgery, Rural Dental College, Loni, Ahmednagar, India
2 Department of Oral Medicine and Radiology, Rural Dental College, Loni, Ahmednagar, India
|Date of Web Publication||20-Jun-2013|
Department of Oral and Maxillofacial Surgery, Rural Dental College, Loni, Tal - Rahata, District Ahmednagar, Maharashtra - 413 736
Source of Support: None, Conflict of Interest: None
Adenomatoid odontogenic tumor (AOT) is a rare tumor generally found in young females. It is more commonly seen in anterior maxilla in association with an unerupted tooth. Dentigerous cyst is a odontogenic cyst commonly seen arising from the impacted mandibular third molars. We present a case of simultaneous occurrence of both these lesions. A female in the second decade of life reported with the complaint of swelling in the anterior region of lower jaw. Clinical and radiographic findings were suggestive of a dentigerous cyst. Cystic lesion was enucleated and the specimen was sent for histopathologic analysis. It showed AOT in association with a dentigerous cyst. This case is worth discussing as it is rare to find the simultaneous occurrence of two pathologies in the same region of jaw. The common site for AOT is anterior maxilla and for dentigerous cyst is posterior mandible. But in our case, it was found in the anterior mandible in association with an impacted canine.
Keywords: Adenomatoid odontogenic tumor, dentigerous cyst, impacted, mandible, maxilla
|How to cite this article:|
Saluja H, Kasat V, Gaikwad P, Mahindra U, Dehane V. A rare occurrence of adenomatoid odontogenic tumor arising from cystic lining in the mandible: Review with a case report. J Orofac Sci 2013;5:50-3
|How to cite this URL:|
Saluja H, Kasat V, Gaikwad P, Mahindra U, Dehane V. A rare occurrence of adenomatoid odontogenic tumor arising from cystic lining in the mandible: Review with a case report. J Orofac Sci [serial online] 2013 [cited 2020 Sep 20];5:50-3. Available from: http://www.jofs.in/text.asp?2013/5/1/50/113695
| Introduction|| |
The adenomatoid odontogenic tumor (AOT) is a rare tumor generally found in young females (f:m = 2.3:1).  It is more commonly seen in anterior maxilla (maxilla:mandible = 2.6:1)  in association with an unerupted tooth. It is generally seen in the second decade of life.  Dentigerous cysts are the most common type of developmental odontogenic cysts arising from the crowns of impacted, embedded, or unerupted teeth. They constitute the second most common cystic lesion of the jaws, after radicular cysts. They are commonly seen in the second and third decades of life and show a male predilection. With patient's consent, management of a case of simultaneous occurrence of these two lesions in an 18-year-old female is described in this article.
| Case Report|| |
An 18-year-old female patient reported to our department with the complaint of swelling in the anterior region of lower jaw since 6-8 months. The swelling was initially small and had gradually increased in size. The medical history was unremarkable. On extraoral examination, there was a diffuse swelling seen on left side of chin [Figure 1]. Intraorally, a firm, slightly tender swelling was present in lower labial/buccal vestibule, extending from 35 to 42 regions and obliterating the vestibular depth. The lower left permanent canine was missing and an over-retained deciduous canine was seen in the same region.
|Figure 1: Extraoral photograph showing diffuse swelling on left side of chin|
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Orthopantomogram (OPG) revealed a well-defined corticated radiolucency associated with an impacted 33. It was extending anteroposteriorly from 42 to 35 and vertically from apices of 41, 31, 32, 73, 34, and 35 to the inferior border of the mandible, causing displacement of the root of 32. External root resorption with 34 was evident [Figure 2]. Computed Tomography (CT) scan revealed a 2.9 × 2.2 cm expansile lesion associated with an impacted 33 [Figure 3]a and b. On transverse section, the impacted tooth was located close to the outer cortex and lingual cortex was seems to be intact on this axis, while in coronal section lesion was seems to be extended to the interdental area leading to resorption of mandibular anterior teeth. On aspiration, a straw-colored fluid was seen. Correlating clinical and radiographic findings, a diagnosis of dentigerous cyst associated with impacted 33 was made. Enucleation of cystic lesion was carried out under general anesthesia after the root canal treatment of involved teeth was completed. The lesion was enucleated in toto along with the impacted 33 [Figure 4]. The enucleated lesion was sent for histopathologic examination which revealed a cystic cavity lined by thin nonkeratinized stratified squamous epithelium resembling reduced enamel epithelium and a tumor in the capsule. The connective tissue was fibrocellular with myxomatous areas and showed distinct tumor tissue composed of nodular proliferation of odontogenic epithelial cells arranged in different patterns like ducts, whorls, rosettes, nests, and strands with eosinophilic coagulum and many microcysts. Cribriform or lace-like pattern showed cords of cuboidal cells with associated hyaline material surrounded by a loose edematous vascular stroma. Areas of amyloid, dysplastic dentin and calcified globules were also noticed. These histopathologic features were suggestive of AOT in the capsule of dentigerous cyst [Figure 5]. Hence, a final diagnosis of AOT with dentigerous cyst was made. Postoperative healing was uneventful and the patient is being followed up for last 1 year and is asymptomatic [Figure 6].
|Figure 2: OPG showing a well-defined corticated radiolucency containing impacted 33|
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|Figure 3: (a and b) Coronal and axial computed tomography scan showing the dimensions of the cyst|
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|Figure 5: Photomicrograph shows lining epithelium of dentigerous cyst and adenomatoid odontogenic tumor in the capsule (H and E Stain 10X magnification)|
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| Discussion|| |
Dentigerous cyst is a type of odontogenic cyst. It encloses the crown of an unerupted tooth and is attached to the cementoenamel junction (CEJ). This cyst is commonly seen in the second and third decades of life, but can occur at any age. These cysts are generally not detected until they are enlarged sufficiently to produce cortical plate expansion. Pain is not a presenting sign unless it is secondarily infected. Most of these cysts are detected at the time of routine radiographic examination because of unerupted or missing tooth. Dentigerous cysts are more common in the mandible than in maxilla and show male predilection. Dentigerous cysts may affect impacted, unerupted, supernumerary teeth, and rarely odontomes. They most frequently involve the mandibular third molar, followed by maxillary canine, mandibular second premolar, and maxillary third molar. Normal follicular space is 3-4 mm thick. A dentigerous cyst can be suspected when the follicular space is more than 5 mm. Radiographically dentigerous cysts are seen as unilocular radiolucent areas having a well-defined sclerotic margin, associated with the crown of an unerupted tooth. The margins are poorly defined in cases of infected cysts.
AOT is an uncommon benign epithelial lesion of odontogenic origin. It was first described by Dreibaldt in 1907 as pseudo-adenoameloblastoma.  It was later described by Ghosh as adamantinoma of the maxilla.  Adenoameloblastoma, ameloblastic adenomatoid tumor, adamantinoma, epitheloma adamantinum, and teratomatous odontoma are the terms that are used to define what is currently known as AOT.  Stafne in 1948 considered it as a distinct entity.  Philipsen and Birn proposed the term AOT, indicating that it is not a variant of ameloblastoma.  In 1971, AOT was accepted in the first WHO classification of odontogenic tumors. 
There are three variants of AOT. They are: (i) follicular type (73%); (ii) extrafollicular type (24%); and (iii) peripheral type. , AOTs are usually solid, but occasionally cystic. Because neoplastic and hamartomatous lesions can occur at any stage of odontogenesis, odontogenic tumors with combined features of epithelial and mesenchymal components may arise within the odontogenic cyst [Table 1]. There is uncertainty whether the lining of an associated cyst represents a true dentigerous cyst, cystic change within an AOT, or a distinct entity. Also, it is unclear whether this entity has a more aggressive potential. Development of odontogenic neoplasms and hamartomas can occur at any stage of tooth development.  Odontogenic tumors and odontogenic cysts are two distinct pathologies seen in tooth-bearing areas of the jaws. Association of these two pathologies is rarely noticed. Santos  reported a case of AOT being developed from the fibrous capsule of the dentigerous cyst.
It is known that the lining epithelium of dentigerous cyst can give rise to some neoplasms like ameloblastoma, squamous cell carcinoma, and mucoepidermoid carcinoma. The secondary development of AOT is also reported in the literature. It is uncertain whether the dentigerous cyst forms first or the AOT. If dentigerous cyst forms in the beginning and then AOT develops in the wall of cyst, then we have cyst attached to the neck of the tooth and tumor in the capsule, as seen in our case. If AOT formation takes place before cystic change, then AOT will fill the follicular space and present as a solid tumor. Sometimes AOT develops as a mural growth in a dentigerous cyst. AOT is benign, hamartomatous, non-invasive, and has a low recurrence rate. Therefore, it is unnecessary to carry out aggressive and extensive surgery. AOT and dentigerous cyst are both benign, encapsulated lesions, and conservative surgical enucleation or curettage is the treatment of choice.
| Conclusion|| |
We have reported a rare case with simultaneous occurrence of dentigerous cyst and AOT.
| References|| |
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]