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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 28  |  Issue : 4  |  Page : 453-457

Adenomatoid odontogenic tumor with dentigerous cyst: Report of an unusual Case


Department of Oral Medicine and Radiology, Maulana Azad Institute of Dental Sciences, New Delhi, India

Date of Submission19-Aug-2015
Date of Acceptance20-Dec-2016
Date of Web Publication21-Feb-2017

Correspondence Address:
Dr. Shikha Gupta
Department of Oral Medicine and Radiology, Maulana Azad Institute of Dental Sciences, New Delhi - 110 002
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.200624

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   Abstract 

Adenomatoid odontogenic tumor (AOT) is a relatively uncommon benign odontogenic tumor which affects females in the second decade of life, with a predilection for the anterior region of the maxilla, usually associated with impacted canine. In this paper, we present a case of follicular AOT associated with dentigerous cyst affecting the anterior mandible in a 12-year-old male with review of the cases reported in literature till date.

Keywords: Adenomatoid, cyst, dentigerous


How to cite this article:
Gupta S, Singh K, Ghosh S, Gupta S. Adenomatoid odontogenic tumor with dentigerous cyst: Report of an unusual Case. J Indian Acad Oral Med Radiol 2016;28:453-7

How to cite this URL:
Gupta S, Singh K, Ghosh S, Gupta S. Adenomatoid odontogenic tumor with dentigerous cyst: Report of an unusual Case. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2021 Jan 28];28:453-7. Available from: https://www.jiaomr.in/text.asp?2016/28/4/453/200624


   Introduction Top


Adenomatoid odontogenic tumor (AOT), an uncommon benign odontogenic tumor, constituting 2–3% of all odontogenic tumors was first described by Dreibaldt in 1907 as pseudoadenoameloblastoma. However, Staphne in 1948 first recognized this as a distinct pathological entity.[1] It is usually found in young patients, especially in the second decade of life, occurring predominantly in females with a male:female ratio of 1:1.9.[2] Philipsen et al. has subdivided it into three groups, namely, follicular, extrafollicular and peripheral.[3] They all share a common histologic characteristic that indicate a common origin being remnant of dental lamina. The most common is the central variant (97.2%), of which 73% is the follicular type. Peripheral variant is rare with only 18 cases reported so far.[4] The follicular variant is associated with an impacted tooth, most commonly maxillary canine (40%). Therefore, 77% of follicular variant is often diagnosed as dentigerous cyst based on the clinical and radiographic findings.[4] Very rarely, AOT is associated with an odontogenic cyst. Till date, there are 14 cases of AOT associated with dentigerous cyst. Here, we report an uncommon case of AOT associated with dentigerous cyst in anterior mandible.


   Case Report Top


A 12-year-old male patient reported to the outpatient department of a tertiary care hospital with the chief complaint of swelling in the anterior mandible for the past 4 months. Swelling was initially small and gradually increased to its present size over a period of 4 months. It was associated with dull, intermittent pain without any history of pus discharge. Medical and personal histories were noncontributory. On extraoral examination, there was left submandibular lymphadenopathy. On inspection, face was asymmetrical with swelling in the anterior mandible extending superiorly from lower lip to 0.5 cm below the inferior border of the mandible inferiorly and medially from a point 1–1.5 cm lateral to the midline on the right side up to left corner of mouth laterally [Figure 1]. On palpation, it was roughly ovoid in shape, being 4 × 3 cm in size and was firm in consistency. The overlying skin was afebrile to touch with normal color and texture.
Figure 1: Extraoral facial photograph of the patient showing facial asymmetry with swelling over the anterior mandible

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On intraoral examination, dome-shaped expansion of buccal cortex was seen extending from the distal surface of the right mandibular canine mesially to the mesial surface of the left mandibular second premolar distally, and from the gingival margins of these teeth to buccal vestibule obliterating the vestibule [Figure 2]. The expansion was approximately 5 × 2 cm in size and the overlying mucosa was normal in color and texture with a smooth surface. There was no lingual expansion [Figure 3]. On palpation, it was firm in consistency with well-defined margins; fluctuant areas were present with respect to the buccal cortex of the left mandibular central and lateral incisor. Left mandibular canine was missing and there was displacement of the left and right mandibular central and lateral incisor with no apparent mobility. Based on the clinical findings, a provisional diagnosis of central giant cell granuloma was made because it is common in younger individuals, presenting anterior to premolars in mandible and usually crossing the midline. Other lesions, including dentigerous cyst, AOT and keratocystic odontogenic tumor, were considered as differential diagnoses. Pulp vitality was done, and teeth associated with the swelling were tested to be vital. All blood investigations including complete blood count, serum calcium, phosphorus, and parathormone levels were within normal limits.
Figure 2: Intraoral photograph of the patient showing dome shaped expansion of the buccal cortex along with displacement of teeth

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Figure 3: Intraoral photograph of the patient with no lingual expansion and missing left canine

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Mandibular cross-sectional occlusal radiograph revealed thinning with expansion of buccal cortex extending from 41 to 35. Impacted canine was seen along the lingual cortex close to the inferior border of the mandible. Orthopantomogram was suggestive of a well-defined unilocular radiolucent lesion surrounded by a thin radiopaque margin in anterior mandible approximately 5 × 3 cm in size extending from the right canine to the left second premolar with expansion of inferior border of mandible. There was displacement of the right central and lateral incisor distally, left central and lateral incisor mesially, and of the left first and second premolar distally, with loss of lamina dura of these teeth. There was apical root resorption of left central and lateral incisor, first and second premolar. Left canine was impacted within the lesion and pushed towards the inferior border of mandible [Figure 4]. Based on radiographic findings, a provisional diagnosis of dentigerous cyst was made, with AOT as the differential diagnosis. Aspiration of the swelling was done, which revealed clear yellow-colored fluid; the smear was suggestive of paucicellular fluid with few red cells.
Figure 4: Orthopantomogram showing well-defined unilocular corticated radiolucency in the anterior mandible with associated impacted canine and its root reaching up to the inferior border of the mandible

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An incisional biopsy was done and the specimen was submitted for histopathological examination. Section showed cystic wall lined by nonkeratinized reduced enamel epithelium-like flattened cystic lining without rete ridges. Epithelium showed whirling and cell rich nodules, duct-like spaces and rosette-like pattern with proliferating interlacing cords at places. Areas of calcification were seen within the epithelium. The underlying connective tissue showed loosely arranged collagen fibres and fibroblasts with few myxoid areas [Figure 5] and [Figure 6]. Overall picture was suggestive of AOT with dentigerous cystic lining. The patient was referred to the department of oral and maxillofacial surgery where enucleation of the lesion was done under local anesthesia. The patient is under regular follow up and no recurrence has been noted during 1 year of follow up.
Figure 5: Photomicrograph (40×) showing flattened epithelial cystic lining

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Figure 6: Photomicrograph (10×) showing whirling and cell rich nodules, duct-like spaces, and rosette-like pattern with proliferating interlacing cords

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   Discussion Top


AOT is a slow growing lesion, constituting only 3% of all odontogenic tumors with a predilection for the anterior maxilla usually associated with impacted canine among young females in the second decade of life.[5] In the present case, the patient was a male in the second decade with a lesion in the anterior mandible associated with impacted canine. Radiographically, this lesion usually surrounds an unerupted tooth and is seen as a corticated radiolucency with small radiopacities. An AOT often appears to envelop the crown as well as the root unlike the dentigerous cyst which does not envelop the roots.[5] In our case, the lesion was seen as well-defined unilocular radiolucency with corticated margins and the lesion was enveloping the crown as well as the root of impacted canine. Root resorption seen in the present case is not a usual feature associated with AOT; this might suggest aggressive nature of the tumor associated with dentigerous cyst. Because odontogenic cysts showing aggressive growth with neoplastic potential have been reported by many authors, it would be interesting to speculate whether dentigerous cyst has the potential of developing into a more aggressive odontogenic neoplasm. The clinical and radiographic differential diagnosis of benign odontogenic tumor has been described in [Table 1].
Table 1: Clinical and radiographic differential diagnosis of benign odontogenic tumor

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According to the second edition of the WHO “Histological Typing of Odontogenic Tumors,” AOT is 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.''[6] When the pathogenesis of AOT is considered, the origin of this tumor is controversial. Some believe that they originate from the odontogenic epithelium of a dentigerous cyst.[1] Therefore, the hypothesis that follicular AOTs arise from the reduced enamel epithelium that lines the follicles of unerupted teeth is fairly conclusive. This is further supported by both morphological and immunocytochemical evidence. According to this hypothesis, the lesions grow next to or into a nearby dental follicle leading to the the “envelopmental theory.”[1]

It has been reported that AOT occurs with many types of cysts and neoplasms including dentigerous cyst, calcifying odontogenic cyst, odontoma and ameloblastoma.[7] This is because neoplastic and hamartomatous lesions can occur at any stage of odontogenesis, and as a result, odontogenic tumors with combined features of epithelial and mesenchymal components may arise within the odontogenic cyst.[1],[8] In relation with a dentigerous cyst, the AOT may demonstrate, grossly and microscopically, one or more associated cystic cavities. Some of these cysts are lined by nonkeratinized stratified squamous epithelium, which is similar to the lining of the dentigerous cyst, as was seen in the present case.[7],[9] Clinical, radiographic and macroscopic findings in this case were consistent with earlier descriptions of the lesion in the literature.

Whether a lesion of the type shown in this case has the potential to develop into a frank AOT is unknown. Whether the origin of the follicular variant occurs before or after cystic expansion is yet to be fully explained. If the tumor grows after cystic expansion, then this confirms its origin from a dentigerous cyst, however, if it occurs before cystic expansion, then the tumor tissue fills the follicular space and the AOT presents as a solid tumor.[1] It is reasonable to assume that, given enough time, even those originating from a cyst may grow and fill the lumen completely. It cannot be ruled out that the dentigerous cyst with an impacted canine developed first followed by development of AOT in the cyst wall.[1] Radiographically, it should be differentiated from dentigerous cyst, which most frequently occurs as a pericoronal radiolucency in the jaws. Dentigerous cyst encloses only the coronal portion of the impacted tooth, whereas AOT shows radiolucency usually surrounding both the coronal and radicular aspects of the involved tooth. However, in cases where AOT grows from dentigerous cyst, like in this case, the radiographs are inconclusive. The irregularity in the wall of cyst may indicate the development of AOT.

Very few cases of AOT arising in association with a dentigerous cyst have been described. A systematic search of English language medical literature results showed only 14 such cases documented till date [Table 2]. Of these, 9 patients were females and 5 were males, with the exception of 3 patients who were in the second decade. Eleven cases presented as a swelling in maxilla with only 3 cases reported in the mandible. The most commonly associated impacted tooth was canine followed by premolars and incisor. Radiographically, all cases presented as well-defined unilocular radiolucency with corticated borders and associated impacted teeth. Only two cases showed scattered radiopacities within the radiolucency. AOTs and dentigerous cysts are both benign, encapsulated lesions and conservative surgical enucleation or curettage is the treatment of choice. Prognosis for a dentigerous cyst is good, and recurrences are very rare after complete removal of the lesion.[1],[6]
Table 2: Review of literature showing characteristics of cases of AOT associated with dentigerous cyst

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   Conclusion Top


Meticulous histopathological evaluation is thus required from all enucleated cysts, which could contribute to the accurate diagnosis and appropriate management. Further research into the possibility of fourth type of a “hybrid” kind of AOT, apart from the already established three types of AOTs, is required.

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.[17]

 
   References Top

1.
Singh V, Goyal S, Sheikh S, Shambulingappa P, Singh B, Singh R. Adenomatoid odontogenic tumor with dentigerous cyst: Report of a rare case with review of literature. Contemp Clin Dent 2012;3:S244-7.  Back to cited text no. 1
    
2.
Philipsen HP, Reichart PA, Siar CH, Ng KH, Lau SH, Zhang X, et al. An updated clinical and epidemiological profile of the adenomatoid odontogenic tumour: A collaborative retrospective study. J Oral Pathol Med 2007;36:383-93.  Back to cited text no. 2
    
3.
Philipsen HP, Reichart PA, Zhang KH. Adenomatoid odontogenic tumor: Biologic profile based on 499 cases. J Oral Pathol Med 1991;20:149-58.  Back to cited text no. 3
    
4.
Sreenivas SD, Lalita CS, Harsha G, Rao VC. Multiple pathology in a single lesion: AOT associated with dentigerous cyst. J Maxillofac Oral Surg 2015;14:215-21.  Back to cited text no. 4
    
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John BJ, John RR. Adenomatoid odontogenic tumor associated with dentigerous cyst in posterior maxilla: A case report and review of literature. J Oral Maxillofac Pathol 2010;14:59-62.  Back to cited text no. 5
[PUBMED]  Medknow Journal  
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Sandhu SV, Narang RS, Jawanda M, Rai S. Adenomatoid odontogenic tumor associated with dentigerous cyst of maxillary antrum: A rare entity. J Oral Maxillofac Pathol 2010;14:24-8.  Back to cited text no. 6
[PUBMED]  Medknow Journal  
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Agarwal A, Giri KY, Alam S. The interrelationship of adenomatoid odontogenic tumor and dentigerous cyst: A report of a rare case and review of the literature. Case Rep Pathol 2012;2012:358609.  Back to cited text no. 7
    
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Garcia-Pola Vallejo M, Gonzalez Garcia M, Lopez-Arranz JS, Herrero Zapatero A. Adenomatoid odontogenic tumor arising in a dental cyst: Report of unusual case. J Clin Pediatr Dent 1998;23:55-8.  Back to cited text no. 8
    
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Rick GM. Adenomatoid odontogenic tumor. Oral Maxillofac Surg Clin North Am2004;16:333-54.  Back to cited text no. 9
    
10.
Valderrama LS. Dentigerous cyst with intracystic adenomatoid odontogenic tumor and complex odontoma. J Philipp Dent Assoc 1988;41:35-41.  Back to cited text no. 10
    
11.
Warter A, George-Diolombi G, Chazal M, Ango A. Melanin in a dentigerous cyst and associated adenomatoid odontogenic tumor. Cancer 1990;66:786-8.  Back to cited text no. 11
    
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Nonaka CFW, De Souza LB, Quindere LB. Adenomatoid odontogenic tumour associated with dentigerous cyst– Unusual case report. Braz J Otorhinolaryngol 2007;73:135-7.  Back to cited text no. 12
    
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Chen YK, Hwang IY, Chen JY, Wang WC, Lin LM. Adenomatoid odontogenic tumor arising from a dentigerous cyst– A case report. Int J Pediatr Otorhinolaryngol 2007;2:257-63.  Back to cited text no. 13
    
14.
Manjunatha BS, Shah V, Pateel D, Tiwari H. Adenomatoid odontogenic tumor with tooth like structure arising from a dentigerous cyst: Report of a unique case. Acta Stomatol Croat 2011;45:276-9.  Back to cited text no. 14
    
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Manjunatha BS, Mahajan A, Mody BM, Shah V. Adenomatoid odontogenic tumor arising from a dentigerous cyst: Literature review and report of a case. J Maxillofac Oral Surg 2015;14:393-7.  Back to cited text no. 15
    
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Morelli HDA, Queiroz SC, Padilha SMW, Santos NDJ, Flores CP. Adenomatoid odontogenic tumor associated to dentigerous cyst. Chirurgia 2013;26:121-3.  Back to cited text no. 16
    
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Latti BR, Kalburge JV. Dentigerous cyst associated with adenomatoid odontogenic tumor- A rare case report & review of literature. Medical Science 2013;1:44-7.  Back to cited text no. 17
    


    Figures

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

  [Table 1], [Table 2]



 

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