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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 17  |  Issue : 3  |  Page : 723-728

Adenomatoid odontogenic tumor: “Quiet but not Quiet” – A rare case report


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 Submission29-Jan-2021
Date of Decision20-Feb-2022
Date of Acceptance16-Jun-2022
Date of Web Publication2-Nov-2022

Correspondence Address:
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
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdmimsu.jdmimsu_40_21

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  Abstract 


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 4];17:723-8. Available from: http://www.journaldmims.com/text.asp?2022/17/3/723/360217




  Introduction Top


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.[1] With a controversial etiology, it is considered a hamartoma by a few authors, whereas others consider it as a neoplastic growth of odontogenic epithelium.[2],[3]

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.[1]

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.[1],[4]

A pure epithelial odontogenic tumor when assessed histopathologically, it exhibits a distinct consistency which makes the diagnosis unambiguous.[3],[5]

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[6] and Ghosh in the early 1930s called it an adamantinoma of the maxilla.[7]

Stephanie in 1948 recognized it as a distinct pathological entity.[8] Bernier and Tiecke coined the term adenoameloblastoma.[9]

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.[10]

Unal et al. produced a list of all the nomenclature such as adenoameloblastoma, ameloblastic adenomatoid tumor, adamantinoma, epithelioma adamantinum, or teratomatous odontoma.[11]

The AOT accounts for 1%–9% of all odontogenic tumors.[12]


  Case Report Top


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.
Figure 1: Frontal View

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Figure 2: Left Lateral View

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Figure 3: Preop Lateral view

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Figure 4: Preop Frontal View

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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.[13]
Figure 5: OPG

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Figure 6: CBCT

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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].
Figure 7: FNAC

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Figure 8: Post Excision

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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.
Figure 9: Gross specimen

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Figure 10: Gross specimen after sectioning

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Figure 11: Duct like structures

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Figure 12: (a) Rosette like pattern (b) Rosette pattern

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Figure 13: Eosinophilic droplets

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Figure 14: Anastomising Cord

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Figure 15: Duct like structure with eosinophilic rim

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The dead space was packed with betadine-soaked cotton gauze which was changed every 72 h for 4 weeks [Figure 16].
Figure 16: One month Follow up

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  Discussion Top


The AOT is an uncommon cause of jaw swelling.[13] The tumor has three clinicopathologic variants, namely, intraosseous follicular, intraosseous extrafollicular, and peripheral. The extrafollicular type (24%) has no relation with an impacted tooth,[14] 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.[14] Follicular and extrafollicular types are more common in the maxilla than in the mandible,[15],[16] and most of the tumors involve an anterior aspect of the anterior maxilla.[17],[18]

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.[19]

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,[17],[20] and this appears most often in the second decade of life.[2],[21] 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,[22] as in the case reported here. The origin of the AOT is controversial.[23],[24] Due to its predilection for tooth-bearing bone, it is thought to arise from odontogenic epithelium.[9]

Although larger lesions have been reported in the literature,[25],[26] the tumors are usually in the dimensions of 1.5–3 cm.[27] Radiographically, they usually appear unilocular and[25],[28] may contain fine calcifications,[17] and irregular root resorption is rare.[28] The differential diagnosis can also be made with ameloblastoma, ameloblastic fibroma, and ameloblastic fibro-odontoma.[29]

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,[27] periapical cyst,[14] calcifying odontogenic cyst, odontoma, ameloblastoma, and so forth[10],[26] 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.[15] Therefore, conservative surgical enucleation produces an excellent outcome. Recurrence is very rare (0.2%).


  Conclusion Top


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.

Learning points

  • 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

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Kramer IR, Pindborg JJ. WHO international histological classification of tumors no. 5. In: Histological Typing of Odontogenic Tumours, Jaw Cysts and Allied Lesions. Berlin: Springer, Verlap; 1971.  Back to cited text no. 1
    
2.
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. 2
    
3.
Philipsen HP, Reichart PA. Adenomatoid ododntogenic tumour: Facts and figures. Eur J Cancer B Oral Oncol 1998;35:125-31.  Back to cited text no. 3
    
4.
Mosqueda-Taylor A. New findings and controversies in odontogenic tumors. Med Oral Patol Oral Cir Bucal 2008;13:E555-8.  Back to cited text no. 4
    
5.
Tahsinoglu M. Odontogenic tumors. Dis Hek Fak Derg 1980;14:91-9.  Back to cited text no. 5
    
6.
Harbitz F. On cystic tumours of the maxilla, and especially on adamantine cystaadenomas (adamantomas). Dent Cosmos 1915;57:1081-93.  Back to cited text no. 6
    
7.
Ghosh LS. Adamantinoma of the upper jaw: Report of a case. Am J Pathol 1934;10:773-790.  Back to cited text no. 7
    
8.
Stafne EC. Epithelial tumors associated with developmental cysts of the maxilla; a report of three cases. Oral Surg Oral Med Oral Pathol 1948;1:887-94.  Back to cited text no. 8
    
9.
Bravo M, White D, Miles L, Cotton R. Adenomatoid odontogenic tumor mimicking a dentigerous cyst. Int J Pediatr Otorhinolaryngol 2005;69:1685-8.  Back to cited text no. 9
    
10.
Rick GM. Adenomatoid odontogenic tumor. Oral Maxillofac Surg Clin North Am 2004;16:333-54.  Back to cited text no. 10
    
11.
Unal T, Cetingul E, Gunbay T. Peripheral adenomatoid odontogenic tumor: Birth of a term. J Clin Pediatr Dent 1995;19:139-42.  Back to cited text no. 11
    
12.
Handschel JG, Depprich RA, Zimmermann AC, Braunstein S, Kübler NR. Adenomatoid odontogenic tumor of the mandible: Review of the literature and report of a rare case. Head Face Med 2005;1:3.  Back to cited text no. 12
    
13.
Nigam S, Gupta SK, Chaturvedi KU. Adenomatoid odontogenic tumor – A rare cause of jaw swelling. Braz Dent J 2005;16:251-3.  Back to cited text no. 13
    
14.
Philipsen HP, Srisuwan T, Reichart PA. Adenomatoid odontogenic tumor mimicking a periapical (radicular) cyst: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:246-8.  Back to cited text no. 14
    
15.
Philipsen HP, Reichart PA. Adenomatoid odontogenic tumour: Facts and figures. Oral Oncol 1999;35:125-31.  Back to cited text no. 15
    
16.
Yilmaz N, Acikgoz A, Celebi N, Zengin AZ, Gunhan O. Extrafollicular adenomatoid odontogenic tumor of the mandible: Report of a case. Eur J Dent 2009;3:71-4.  Back to cited text no. 16
    
17.
Toida M, Hyodo I, Okuda T, Tatematsu N. Adenomatoid odontogenic tumor: Report of two cases and survey of 126 cases in Japan. J Oral Maxillofac Surg 1990;48:404-8.  Back to cited text no. 17
    
18.
Swasdison S, Dhanuthai K, Jainkittivong A, Philipsen HP. Adenomatoid odontogenic tumors: An analysis of 67 cases in a Thai population. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:210-5.  Back to cited text no. 18
    
19.
Ajagbe HA, Daramola JO, Junaid TA, Ajagbe AO. Adenomatoid odontogenic tumor in a black African population: Report of thirteen cases. J Oral Maxillofac Surg 1985;43:683-7.  Back to cited text no. 19
    
20.
Mendis BR, MacDonald DG. Adenomatoid odontogenic tumour. A survey of 21 cases from Sri Lanka. Int J Oral Maxillofac Surg 1990;19:141-3.  Back to cited text no. 20
    
21.
Vera Sempere FJ, Artes Martínez MJ, Vera Sirera B, Bonet Marco J. Follicular adenomatoid odontogenic tumor: Immunohistochemical study. Med Oral Patol Oral Cir Bucal 2006;11:E305-8.  Back to cited text no. 21
    
22.
Batra P, Prasad S, Parkash H. Adenomatoid odontogenic tumour: Review and case report. J Can Dent Assoc 2005;71:250-3.  Back to cited text no. 22
    
23.
Giansanti JS, Someren A, Waldron CA. Odontogenic adenomatoid tumor (adenoameloblastoma). Survey of 3 cases. Oral Surg Oral Med Oral Pathol 1970;30:69-88.  Back to cited text no. 23
    
24.
Tajima Y, Sakamoto E, Yamamoto Y. Odontogenic cyst giving rise to an adenomatoid odontogenic tumor: Report of a case with peculiar features. J Oral Maxillofac Surg 1992;50:190-3.  Back to cited text no. 24
    
25.
Larsson A, Swartz K, Heikinheimo K. A case of multiple AOT-like jawbone lesions in a young patient – A new odontogenic entity? J Oral Pathol Med 2003;32:55-62.  Back to cited text no. 25
    
26.
Khot K, Vibhakar PA. Mural adenomatoid odontogenic tumor in the mandible – A rare case. Int J Oral Maxillofac Pathol 2011;2:35-9.  Back to cited text no. 26
    
27.
Sandhu SV, Narang RS, Jawanda M, Rai S. Adenomatoid odontogenic tumor associated with dentigerous cyst of the maxillary antrum: A rare entity. J Oral Maxillofac Pathol 2010;14:24-8.  Back to cited text no. 27
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28.
Dayi E, Gürbüz G, Bilge OM, Ciftcioğlu MA. Adenomatoid odontogenic tumour (adenoameloblastoma). Case report and review of the literature. Aust Dent J 1997;42:315-8.  Back to cited text no. 28
    
29.
Shetty K, Vastardis S, Giannini P. Management of an unusually large adenomatoid odontogenic tumor. Oral Oncol Extra 2005;41:316-8.  Back to cited text no. 29
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14], [Figure 15], [Figure 16]



 

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