|Year : 2022 | Volume
| Issue : 3 | Page : 723-728
Adenomatoid odontogenic tumor: “Quiet but not Quiet” – A rare case report
Soumi Samuel1, Biju Thomas2, Saurabh Pillai1, Harish Kumar1, Abel Abraham1, Deeyah Miriam Deepak3
1 Department of Oral and Maxillofacial Surgery, A B Shetty Memorial Institute of Dental Sciences (ABSMIDS), Nitte University (Deemed to be University), Deralakatte, Mangalore, India
2 Department of Periodontics, A B Shetty Memorial Institute of Dental Sciences (ABSMIDS), Nitte University (Deemed to be University), Deralakatte, Mangalore, India, India
3 Undergraduate Dental Intern, A B Shetty Memorial Institute of Dental Sciences (ABSMIDS), Nitte Deemed to be University, Mangalore, Karnataka, India
|Date of Submission||29-Jan-2021|
|Date of Decision||20-Feb-2022|
|Date of Acceptance||16-Jun-2022|
|Date of Web Publication||2-Nov-2022|
Dr. Soumi Samuel
Department of Oral and Maxillofacial Surgery, A B Shetty Memorial Institute of Dental Sciences (ABSMIDS), Nitte University (Deemed to be University), Deralakatte - 575 018, Mangaluru
Source of Support: None, Conflict of Interest: None
The adenomatoid odontogenic tumor (AOT) is a hamartomatous benign neoplasia of odontogenic origin. It appears to be, mostly prevalent in young female patients, mostly during the second decade of life. The maxillary region is most affected when compared to the mandible. Its presentation is slow-growing, asymptomatic, and quiet but not quite a lesion. It is related to canines which are unerupted. Here, we report a case of the AOT in the left maxillary region of a female patient aged 13 years. We approached with a surgical enucleation resulting in an excellent outcome.
Keywords: Adenomatoid odontogenic tumor, benign neoplasia, hamartomatous, maxilla, rare
|How to cite this article:|
Samuel S, Thomas B, Pillai S, Kumar H, Abraham A, Deepak DM. Adenomatoid odontogenic tumor: “Quiet but not Quiet” – A rare case report. J Datta Meghe Inst Med Sci Univ 2022;17:723-8
|How to cite this URL:|
Samuel S, Thomas B, Pillai S, Kumar H, Abraham A, Deepak DM. Adenomatoid odontogenic tumor: “Quiet but not Quiet” – A rare case report. J Datta Meghe Inst Med Sci Univ [serial online] 2022 [cited 2023 Feb 8];17:723-8. Available from: http://www.journaldmims.com/text.asp?2022/17/3/723/360217
| Introduction|| |
An adenomatoid odontogenic tumor (AOT) is a rare tumor of odontogenic origin. It can be described as a benign, epithelial tumor with a slow growth rate in the presence or absence of teeth or pain, which causes gradual expansion of bone and results in deformity of the face.
The term AOT adopted by the WHO in 2005, was formerly introduced by Philpsen and Birn. With a controversial etiology, it is considered a hamartoma by a few authors, whereas others consider it as a neoplastic growth of odontogenic epithelium.,
Defined as “a tumor of odontogenic epithelium with duct-like structures and with varying degrees of inductive change 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” by the WHO.
It is classified as a mixed odontogenic neoplasm by the WHO as it often consists of odontogenic hard tissue originating from the dental epithelium and the ectomesenchyme. Currently, it is classified as an epithelial tumor devoid of odontogenic mesenchyme and categorized as a tumor with 1%–10% frequency.,
A pure epithelial odontogenic tumor when assessed histopathologically, it exhibits a distinct consistency which makes the diagnosis unambiguous.,
Review of literature
B4-Steensland described AOT as a relatively uncommon neoplasm of odontogenic origin in 1905.
B5-in 1907, Dreibaltt proposed the term “pseudo-adenoma-ameloblastoma.” Harbitz reported it as a cystic adamantoma in 1915 and Ghosh in the early 1930s called it an adamantinoma of the maxilla.
Stephanie in 1948 recognized it as a distinct pathological entity. Bernier and Tiecke coined the term adenoameloblastoma.
Gorlin et al. in 1961 introduced the term ameloblastic adenomatoid tumor. Moreover, Shafer et al. supported this claim. Philipsen and Birn in 1969 proposed the name “AOT” and shortly after, it was adopted in the initial edition of the WHO histological typing of odontogenic tumors, jaw cysts, and allied lesions in 1971 and was retained in the second edition in 1992.
Unal et al. produced a list of all the nomenclature such as adenoameloblastoma, ameloblastic adenomatoid tumor, adamantinoma, epithelioma adamantinum, or teratomatous odontoma.
The AOT accounts for 1%–9% of all odontogenic tumors.
| Case Report|| |
A 13-year-old girl presented with left anterior maxillary swelling of about 2-month duration [Figure 1] and [Figure 2] and a slow-growing swelling with no other symptoms. On clinical examination, the left cheek was grossly deformed with normal skin overlying the swelling, and there was no neurological deficit over the affected area and no nasal discharge [Figure 3] and [Figure 4]. The palatal vault showed no deformity, and eggshell crackling was palpable in the buccal aspect of the swelling. The oral mucosa over this area appeared healthy and asymptomatic, without any evidence of infection. A canine was seen to be missing.
There was an expansion of the buccal cortical plate of the left maxilla from the central incisor to the first molar on the same side.
The radiograph with orthopantomogram and cone-beam computed tomography showed a well-defined, unilocular radiolucency in the maxilla with expansion and thinning of all its bony walls with the left upper canine tooth and without any evidence of calcifications [Figure 5] and [Figure 6]. It also showed displacement of the tooth and root resorption of the lateral incisor and the first premolar. A clinical diagnosis of dentigerous cyst and adenomatoid odontogenic tumor (AOT) was made. Radiolucent lesions worthy of consideration are keratocystic odontogenic tumor, ameloblastic fibroma, odontogenic myxoma, or central giant cell tumor, as well as unicystic ameloblastoma as the age increases beyond 14 years.
Fine-needle aspiration cytology contained yellowish-brown-colored fluid [Figure 7]. The enucleation of the cyst was done under local anesthesia, sparing the lateral incisor and adjacent premolar [Figure 8].
The histopathological report was suggestive of AOT [Figure 9] and [Figure 10]. The solid areas show multinodular proliferation of spindle and cuboidal and columnar cells in a scattered duct-like structure [Figure 11] and rosette pattern [Figure 12]a and [Figure 12]b. Connective tissue stroma in loose areolar tissue with numerous vascular components extravasated red blood cells, eosinophilic droplets, anastomosing cord, and duct-like structure with eosinophilic rim were seen [Figure 13], [Figure 14], [Figure 15]. Areas presenting with cystic components show dentigerous-like epithelial lining and tumor cells seen proliferating in a solid pattern into the lumen.
The dead space was packed with betadine-soaked cotton gauze which was changed every 72 h for 4 weeks [Figure 16].
| Discussion|| |
The AOT is an uncommon cause of jaw swelling. The tumor has three clinicopathologic variants, namely, intraosseous follicular, intraosseous extrafollicular, and peripheral. The extrafollicular type (24%) has no relation with an impacted tooth, whereas the follicular type (73% of all AOT cases) is associated with an unerupted tooth as in the case we presented here, and the peripheral variant (3%) is attached to the gingival structures. Follicular and extrafollicular types are more common in the maxilla than in the mandible,, and most of the tumors involve an anterior aspect of the anterior maxilla.,
In our case, the tumor was a follicular intraosseous type and also found in the anterior region of the maxilla. There is a slight female over male predilection, almost 2:1.
Asian AOT cases (reported from Japan, India, China, Thailand, Taiwan, Sri Lanka, and Malaysia) show a female: male ratio of 2.3:1. If cases reported from Sri Lanka and Japan are considered separately, they show ratios of 3.2:1 and 3.0:1, respectively,, and this appears most often in the second decade of life., The sex and the age of the patient we described in this article were consistent with the literature. The lesions are typically asymptomatic but may cause cortical expansion and displacement of the adjacent teeth, as in the case reported here. The origin of the AOT is controversial., Due to its predilection for tooth-bearing bone, it is thought to arise from odontogenic epithelium.
Although larger lesions have been reported in the literature,, the tumors are usually in the dimensions of 1.5–3 cm. Radiographically, they usually appear unilocular and, may contain fine calcifications, and irregular root resorption is rare. The differential diagnosis can also be made with ameloblastoma, ameloblastic fibroma, and ameloblastic fibro-odontoma.
Histologically our case reports the features were suggestive of AOT. Interestingly, there are a few reports of AOT to occur with many types of cysts and neoplasms including dentigerous cyst, periapical cyst, calcifying odontogenic cyst, odontoma, ameloblastoma, and so forth, and also about pigmented cells in AOT. However, all of these reported lesions did not show macroscopically visible pigmentation.
The tumor is well encapsulated and showed an identical benign behavior. Therefore, conservative surgical enucleation produces an excellent outcome. Recurrence is very rare (0.2%).
| Conclusion|| |
The tumor presentation was well encapsulated and benign in nature; conservative surgical enucleation was the treatment of choice. In our cases, the lesions were associated with the tooth favoring orthodontic movement. Our patient has been under follow-up for the past 6 months and is planned for rehabilitation in coordination with the department of orthodontics for the protraction of the canine.
- The AOT presents a very quiet presentation which is asymptomatic, along with a facial asymmetry accompanied by an unerupted tooth
- The female: male ratio of 2.3:1 and mostly seen in the second decade of life
- Clinical presentation, radiographic interpretation, and histological examination are very important for treatment planning
- In the younger age group with a regular follow-up, the patient can be planned for orthodontic rehabilitation if the unerupted tooth is not sacrificed during the surgical intervention.
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
Conflicts of interest
There are no conflicts of interest.
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