akerzoul n et al.: case report - ijohmr odontogenic tumor- an unco… · international journal of...

International Journal of Oral Health and Medical Research | ISSN 2395-7387 | MARCH-APRIL 2016 | VOL 2 | ISSUE 6 70

CASE REPORT Akerzoul N et al.: Adenomatoid Odontogenic Tumor

Correspondence to: Dr. Narjiss.Akerzoul, Résidence Ryad Al Majd n°8,

App1 RDCH, Hay El Fath, Rabat, Morocco. Contact Us: www.ijohmr.com

Adenomatoid Odontogenic Tumor: An

Uncommon Mandibular Case Report Narjiss Akerzoul1, Saliha Chbicheb2, Wafaa El Wady3

The adenomatoid odontogenic tumor is a hamartomatous benign neoplasia of odontogenic origin. It appears mostly in

young patients and females, the maxillary region being the most affected. It is a slow-growing, asymptomatic lesion. It

is related to non-erupted teeth, mainly canines. Lesions of this type can be clinically classified as a follicular,

extrafollicular and peripheral lesions.The treatment for these lesions is enucleation and curettage of affected area. Here

we report a case of the adenomatoid odontogenic tumor (AOT) in the mandibular right region of in a young man aged

20 years, and no recurrence has been observed after one-month follow-up.

KEYWORDS: Adenomatoid, Odontogenic Tumor, Mandibular, Uncommon, Benign Neoplasia

AA aaaasasasss The adenomatoid odontogenic tumor was first described,

in 1907 by Dreibladt, as a pseudo adenoameloblastoma.1

Over years, a variety of terminologies has been used to

designate this extremely fascinating entity like

adenoameloblastoma, adenoameloblastic odontoma,

epithelial tumor associated with an ameloblastic

adenomatoid tumor, developmental cysts, and

adenomatoid or pseudo adenomatous ameloblastoma.

Philipsen and Ben suggested the name of adenomatoid

odontogenic tumour in 1969 and that it won’t be

considered as a variant of ameloblastoma because of its

different behaviour. 1,2

The adenomatoid odontogenic tumor is also called «A

two-thirds tumor », because 2/3rd of the adenomatoid

tumors occur in young females, 2/3rd of the adenomatoid

tumors occur in the maxilla, 2/3rd of the cases of these

tumors are associated with unerupted teeth, and two-

thirds of the affected teeth are canines.3

There are 3 variants of adenomatoid odontogenic tumor.

The follicular type (accounting for 73% of cases), which

has a central lesion associated with an embedded tooth,

the extrafollicular type (24% of case), which has a central

lesion and no connection with the tooth, and the

peripheral variety (accounting for 3% of cases). The aim

of this paper is to describe a case of the adenomatoid

odontogenic tumor (AOT) in the mandibular right region

of in a young man aged 20 years, and no recurrence has

been observed after one-month follow-up.

A 20-year-old young male reported to the Oral Surgery

Department complaining from a swelling in the right

inferior front tooth region evolving since 5 of months.

The medical history of the lesion revealed that initially

the swelling was small in size, and gradually it increased

to reach up to the present size. It was not associated with

any pain or discharge, with no history of trauma.

The Intraoral examination revealed a distinct swelling in

the anterior region of the mandible from the 45 to 33. The

overlying mucosa was normal, and there was no

paresthesia in the mandibular region. Because of the

swelling, the central and lateral mandibular incisors were

deviated to the right, and the patient had retained his right

canine (Figure 1).

Panoramic radiograph and computed tomography

incidences were performed, and which revealed a well

defined unilocular radiolucency of the anterior

How to cite this article: Akerzoul N, Chbicheb S, ElWady W. Adenomatoid Odontogenic Tumor: An Uncommon Mandibular Case Report. Int J Oral Health Med Res 2016;2(6):70-74.

INTRODUCTION

1-Resident Oral Surgeon, Department Of Oral Surgery, C.C.D.T, Faculty Of Dentistry, University Mohamed V, Rabat, Morocco. 2-Professor, and Chief Department Of Oral Surgery, C.C.D.T, Faculty Of Dentistry, University Mohamed V, Rabat, Morocco. 3-Professor, And Chief Service Of Oral Surgery, C.C.D.T, Faculty Of Dentistry, University Mohamed V, Rabat, Morocco.

ABSTRACT

CASE REPORT

Fig 1 : Intraoral examination revealing the Presence Of a mandibular Swelling extended from the 45 to 41 with a right deviation of the

lateral mandibular incisors



mandibular region, which contained an impacted right

mandibular canine with radiopaque foci. (Figure 2 and 3)

The differential diagnosis was based on the clinical and

radiographic findings included a dentigerous cyst, uni

cystic ameloblastoma, and Adenomatoid Odontogenic

Tumor (AOT).

The surgical procedure consisted on firstly elevate a

mucoperiosteal flap in order to expose the lesion (Figure

4).The bone window was performed to have access to the

lesion (Figure 5). The lesion was separated from the

surrounding bone and then, the lesion and impacted

canine were enucleated (Figure 6, and 7). Intraoperati-

vely, the specimen exhibited a solid and sand-like texture

inside lesion (Figure 8).

The specimen was then sent to the department of

pathology for histopathological study.

The complete enucleation of the lesion was done under

local anesthesia with the removal of impacted 43, and

then the cystic cavity was completely cleaned (Figure 9).

Fig 2 (A+B) : Panoramic view showing a unilocular radiolucency lesion extended from the mesial side of 46 to the mesial side of 33 and

impacted right canine.

A

B

Fig 3 : CT Scan showing the blister of both the buccal and lingual bone tables, with more thiness of the buccal bone table compared to the

lingual one

Fig 4 : Surgical Flap elevation

Fig 5 : Per-operative view showing the Bone window time

Fig 6 : Per-operative Tumor Enucleation



Postoperative sutures were performed to finish the

surgical intervention (Figure10). 3 months Postoperative

follow-up did not show any signs of recurrence or

complications.

The histopathology of lesional tissue revealed a

multilobular proliferation of spindle cells in sheets, duct-

like pattern, and whorled arrangement of darkly staining

epithelial cells suggestive of an odontogenic epithelial

cells. The cuboidal to columnar cells arranged in the form

of nests and rosettes. The duct-like structures with lumina

of varying size were lined by columnar cells with

palisading appearance. A few basophilic calcifications

were also observed. The surrounding connective tissue

stroma was less cellular in nature. Based on these

findings, the final diagnosis was an adenomatoid

odontogenic tumor (Figures 11).

AOT is a rare benign odontogenic neoplasm with

predilection of occurrence in females and occurs mainly

in the second decade of life as seen in our case.1,2

The

lesions usually are characterized by the presene of an

Fig 7 : Extraction of the impacted canine (43)

Fig 8 : Tumor specimen measuring 5cm approximately + removed impacted 43

Fig 9 : Clean Cystic cavity after enucleation of the tumor and removal of the 43

Fig10: Post-operative view after sutures

Fig 11 : Histological section : presence of a solid part containing epithelial cells that form rosettes and tubes, some of these tubes

contained a calcified substance

DISCUSSION



asymptomatic jaw swelling, relatively small in size, and

not exceeding 1-3 cm in diameter, as seen in our case.3

AOT exists in three clinical subtypes: follicular type (in

73% of AOT) which is intraosseous in location and is

associated with an unerupted tooth (usually canine); the

extra-follicular variant (24%) which is located

intraosseously but is not associated with unerupted tooth,

and the peripheral form which is the most rare (3%) and

occurs within gingival mucosa.4

On radiographs, the intraosseous follicular variant of

AOT shows a well delineated, unilocular radiolucency

surrounding the crown of a retained tooth ; a picture very

similar to dentigerous cysts. Minor radiopacities around

the retained tooth may be found in AOT and are

considered a characteristic but not pathognomonic

finding. About 2/3 AOT show distinct radiopaque

calcification on radiographs.5,6

The extra-follicular variant presents as a unilocular, well-

defined radiolucency seen between, above, or

superimposed on the roots of erupted teeth and often

resembles a cystic lesion.

Our case presented as an extra-follicular AOT that was

diagnosed clinically as a cystic lesion. AOT affects more

commonly the maxilla than the mandible in a ratio of

2.1:1.7 The case reported here is considered uncommon

as it involved the mandible.

According to Philipsen et al., the fact that all AOT

variants show identical histologic characteristics points to

a common origin. The pathogenesis of this odontogenic

tumor appears to involve persistence of remnants of the

dental lamina, especially after odontogenesis of the

successional lamina. These remnants give rise to

epithelial rests that proliferate in response to an unknown

stimulus.8

The radiographic findings of AOT frequently resemble

other odontogenic lesions such as dentigerous cysts,

calcifying odontogenic cysts, calcifying odontogenic

tumors, ameloblastomas, odontogenic keratocystic tumor,

and periapical disease.9,10

Comparing diagnostic accuracy

between intraoral periapical and panoramic radiographs,

Dare et al. 11

found that intraoral periapical radiographs

allow perception of the radiopacities in AOT as discrete

foci having a flocculent pattern within radiolucency even

with minimally calcified deposits while panoramic often

do not. Those calcified deposits are seen in approximately

78% of AOT.

The diagnosis will especially be based on histological

findings . At this stage also, this tumor must be

differentiated from the calcified epithelial odontogenic

tumor or Pindborg’s tumor which radiologically appears

as a uni- or multilocular radiolucent image without

peripheral dense border, scattered with radiopaque

images of variable sizes and associated in 60 % of the

cases to an impacted tooth or an odontoma

.Macroscopically, these tumors are perfectly limited by a

fibrous connective capsule, having a smooth surface and

a rather firm consistency, a tooth can be attached or

included in the tumor. In the biopsy, a schematic liquid is

frequently noted, and the significant growth gives an

irregular shape to the cyst.

Histologically, the adenomatoid odontogenic tumor is

perfectly well-defined with the common presence of

lobes and cystic cavities. Epithelial cells are closely

aligned against the cyst border. There is little of the

stroma. Cells have a vesicular nucleus and are arranged in

nodules, spirals, and rosettes. They are crossed by

canalicular structures lined with a cylindrical epithelium

with rows of nucleus situated on the opposite side. Inside

these canalicular cells, an eosinophilic amorphous

material is found in contact with the apical pole of the

cells. Calcifications are present in some places

representing a sign of enamel formation. They are often

in association with the groups of cylindrical cells

morphologically identical to ameloblasts.12,13,14

Conservative surgical enucleation is the treatment

modality of choice. For periodontal intrabody defects

caused by AOT, guided tissue regeneration with

membrane technique is recommended after complete

removal of the tumor.15,16

Recurrence of AOT is

exceptionally rare. Therefore, the prognosis is excellent.

No recurrence was seen in our present case.

AOT is a relatively uncommon distinct odontogenic

neoplasm, and is rightfully called as Perfect imitator of

dentigerous cyst. It should be a part of differential

diagnosis whenever we encounter a lesion in young

patients with unerupted tooth (Two Third Tumor).

1. Philipsen HP, Birn H. Th e adenomatoid odontogenic

tumour. Ameloblastic adenomatoid tumour or adeno-

ameloblastoma. Acta Pathol Microbiol Scand

1969;75:375-98.

2. Shivali V, Pandey A, Khanna VD, Khanna P, Singh A,

Ahuja T. A rare case of extrafollicular adenomatoid

odontogenic tumour in the posterior region of the

mandible: misdiagnosed as residual cyst. J Int Oral Health

2013;5:124-8.

3. Philipsen HP, Nikai H. Adenomatoid odontogenic tumour.

In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors.

In: Pathology and Genetics of Head and Neck Tumors.

Lyon, France: IARC Press; 2005 ;2 : 304-5.

4. Narayanan VS, Naidu G, Ragavendra R, Mhaske-Jedhe S,

Haldar M. Adenomatoid odontogenic tumor of the

mandible with unusual radiographic features: A case

report. Imaging Sci Dent 2013;43:111-5.

5. Ide F, Mishima K, Kikuchi K, Horie N, Yamachika S,

Satomura K, et al. Development and growth of

adenomatoid odontogenic tumor related to formation and

eruption of teeth. Head Neck Pathol 2015;5:123-32.

6. More CB, Das S, Gupta S, Bhavsar K. Mandibular

adenomatoid odontogenic tumor: Radiographic and

pathologic correlation. J Nat Sci Biol Med 2013;4:457-62

7. Bhullar RP, Brar RS, Sandhu SV, Bansal H, Bhandari R.

Mandibular adenomatoid odontogenic tumor: A report of

an unusual case. Contemp Clin Dent 2011;2:230-3.

CONCLUSION

REFERENCES



8. Kurra S, Gunupati S, Prasad PR, Raju Y S, Reddy BV. An

adenomatoid odontogenic cyst (AOC) with an assorted

histoarchitecture: A unique entity. J Clin Diagn Res

2013;7:1232-5.

9. More CB, Das S, Gupta S, Bhavsar K. Mandibular

adenomatoid odontogenic tumor: Radiographic and

pathologic correlation. J Nat Sci Biol Med 2015;4:457-62.

10. 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.

11. Saluja R, Kaur G, Singh P. Aggressive adenomatoid

odontogenic tumor of mandible showing root resorption:

A histological case report. Dent Res J (Isfahan)

2014;10:279-82.

12. Mutalik VS, Shreshtha A, Mutalik SS, Radhakrishnan R.

Adenomatoid odontogenic tumor: A unique report with

histological diversity. J Oral Maxillofac Pathol. 2014;16:

118-21.

13. Philipsen HP, Reichart PA. Adenomatoid odontogenic

tumour: facts and figures. Oral Oncol 1999;35:125-31.

14. Sato D, Matsuzaka K, Yama M, Kakizawa T, Inoue T.

Adenomatoid odontogenic tumor arising from the

mandibular molar region: a case report and review of the

literature. Bull Tokyo Dent Coll 2014;45:223-7.

15. Vitkus R, Meltzer JA. Repair of a defect following the

removal of a maxillary adenomatoid odontogenic tumor

using guided tissue regeneration. A case report.

J Periodontol 2006;67:46-50.

16. Chaisuparat R, Sawangarun W, Scheper MA. A

clinicopathological study and treatment of malignant

odontogenic tumours. Histopathology 2015;61:107-112.

Source of Support: Nil

Conflict of Interest: Nil

akerzoul n et al.: case report - ijohmr odontogenic tumor- an unco… · international journal of...

Documents