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Journal of Oral and Maxillofacial Radiology / May-August 2014 / Vol 2 | Issue 2 68

An unusually large adenomatoid odontogenic


tumor with involvement of the ethmoidal sinus:
A rare case report
Puneet Kalra, Shalu Rai
1
, Siddharth Sharma, Shashank Tripathi, Afzal Ali
Departments of Oral Surgery and
1
Oral Medicine and Radiology, Institute of Dental Studies and Technologies, Kadrabad, Modinagar,
Uttar Pradesh, India
INTRODUCTION
Considerable variation in the clinicopathologic presentation
of epithelial odontogenic tumors can sometimes be
confusing and increase the chances of misdiagnosis. The
differential diagnoses of radiolucencies that occur in the
maxilla and mandible include a broad spectrum of cysts
and tumors of odontogenic and nonodontogenic origin.
Adenomatoid odontogenic tumor (AOT) is a distinct
benign (hamartomatous), noninvasive slow growing
odontogenic tumor.
[1]
It accounts to about 2.2-7.1% of all
odontogenic tumors.
[1]
It was rst described by Steensland
in 1905.
[2]
In 1969, Philipsen and Birn
[3]
proposed the term
AOT and were rst adopted by the 1
st
edition of WHO
classication of odontogenic tumor.
According to the 2
nd
edition of WHO Histological
Typing of Odontogenic Tumors, Jaw Cyst and Allied
Lesions in 2005
[4]
AOT is defined as A tumor of
odontogenic epithelium with duct-like structures and
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. There are three variants of AOT:
Follicular, extra follicular, and peripheral. Follicular type is
associated with an impacted tooth and accounts for 70%
of cases. Extrafollicular type is not related to impacted
tooth and accounts for 25% cases. Peripheral type is rare
and arises only in the gingival tissues.
This case paper aims to describe an unusual case of
AOT, originating in the maxillary sinus of a 17-year-old
female.
A B S T R A C T
Adenomatoid odontogenic tumor (AOT) is an uncommon nonaggressive tumor of the odontogenic epithelium with duct-like structures
and varying degrees of inductive changes in the stroma. It comprises only 0.1% of tumors of the jaw and 3% of all odontogenic tumors.
The majority of the cases (88%) are diagnosed in the second and third decades of life. The incidence is higher in males than in females.
The tumor has a predilection for the anterior maxilla, most commonly associated with maxillary canine; however, involvement of the
maxillary antrum is extremely rare. We report an unusually large AOT of the maxilla along with the computed tomography images in
a 17-year-old female patient encroaching the maxillary sinus, orbital oor, nasal septum, ethmoidal sinus on the right side. It was also
associated with an impacted canine. The patient was subjected for complete enucleation along with the removal of the impacted tooth.
To the best of our knowledge, it is the rst time that such a large AOT with involvement of the ethmoidal sinus has been reported.
Key words: Adenomatoid odontogenic tumor, computed tomography scan, ethmoidal sinus, maxillary sinus
Address for correspondence: Dr. Shalu Rai, Department of Oral Medicine and Radiology, Institute of Dental Studies and Technologies,
Kadrabad, Modinagar - 201 201, Uttar Pradesh, India. E-mail: drshalurai@gmail.com
Case Report
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DOI:
10.4103/2321-3841.138654
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Kalra, et al.: An unusually large adenomatoid odontogenic tumor with involvement of the ethmoidal sinus
Journal of Oral and Maxillofacial Radiology / May-August 2014 / Vol 2 | Issue 2 69
CASE REPORT
A 17-year-old female patient reported with the chief complaint
of swelling of the right side of the face since 3 years. The
swelling had been present from past 3 years and had gradually
increased to attain the present size. Past dental history revealed
extraction of maxillary anterior tooth. Extraorally, a diffuse
swelling, resulting in facial asymmetry was present in the
right maxillary region lateral to the nose extending up to the
infraorbital region. It was irregular in shape and measured
approximately 2-3 cm in diameter. Intraorally, buccally, the
swelling extended from the distal surface of the right maxillary
central incisor to the right maxillary rst premolar, obliterating
the buccal vestibule [Figure 1]. Palatally, swelling extended up
to the midline of the hard palate.
Patient was subjected for radiological investigations.
Paranasal sinus view and axial computed tomography (CT)
scan showed hyperdense expansile area in right antrum
causing the expansion and thinning of the maxillary sinus
walls with an unerupted tooth near the lower border of the
orbit [Figure 2a and 2b]. Coronal CT showed hypodense
area in the right antrum causing thinning of the lateral
wall and perforation of the medial wall of the maxillary
sinus, extending superiorly into the ethmoidal sinus with
deviation of the nasal septum and complete obliteration
of the nasal fossa [Figure 2c].
An incisional biopsy was performed, and the patient
underwent surgery under general anesthesia. A
mucoperiosteal ap was raised; resorption of the buccal
cortical plate was noted; the whole tumor mass was
enucleated along with the impacted teeth [Figure 1a
and b] and the specimen was sent for histopathological
examination. Healing was uneventful. The radiographic
follow-up examination after 6 months revealed no local
recurrence. Microscopically the lesion showed a highly
cellular tissue made up of cuboidal and tall columnar cells
arranged in whorl-like pattern. Microcystic areas with
homogeneous eosinophilic material could be appreciated
in some regions. Connective tissue was scanty showing
blood vessels with extravasated blood. The overall picture
conrmed the diagnosis of AOT [Figure 3].
DISCUSSION
The origin of AOT is controversial. Due to its predilection for
tooth-bearing bone, it is thought to arise from odontogenic
epithelium.
[5,6]
Recent reports indicate that the cells of
AOT usually differentiate toward an apparent ameloblastic
phenotype, but fail to achieve further functional maturation.
[5]
Some features of AOT suggest it to be a hamartoma,
[6]

whereas some a neoplasm
[7]
WHO classication in 1971 stated
that the lesion is generally believed not to be a neoplasm. Some
Figure 3: H and E section showing a highly cellular tissue made up of cuboidal;
and tall columnar cells arranged in whorl-like pattern along with microcystic
areas with homogeneous eosinophilic material
Figure 2: (a) Paranasal sinus view showing haziness in right maxillary antrum
with maxillary canine displaced superiorly near the lower border of the orbit.
(b) Axial computed tomography (CT) scan showing hyperdense expansile area
in right antrum causing the expansion and thinning of the sinus walls along
with an unerupted tooth near the lower border of the orbit. (c) Coronal CT
showing hyperdense area in the right antrum causing thinning of the lateral
wall of and perforation of the medial wall of the maxillary sinus, extending
superiorly into the ethmoidal sinus with deviation of nasal septum and
complete obliteration of nasal fossa
a b
c
Figure 1: (a) Extra oral swelling on right mid facial region (b) Excisional
biopsy performed under general anaesthetic. (c) Postoperative specimen
b
c
a
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Kalra, et al.: An unusually large adenomatoid odontogenic tumor with involvement of the ethmoidal sinus
Journal of Oral and Maxillofacial Radiology / May-August 2014 / Vol 2 | Issue 2 70
authors consider AOT a neoplasm due to its considerable size
that goes unnoticed and untreated for many years.
[5,8]
Clinical features generally focus on complaints regarding a
missing tooth. The lesion usually presents as asymptomatic
swelling, which is slowly growing and often associated
with an unerupted tooth, diagnosed in the second decade
of life and women are twice as commonly affected than
men.
[9]
Highest ratios are found in Asian populations, highest
incidence in Sri Lanka (3.2:1) and Japan (3:1)
[10]
and twice
more common in maxilla than mandible, with the anterior
aspect twice more commonly affected than the posterior.
The radiographic ndings of AOT frequently resemble other
odontogenic lesions such as dentigerous cysts, calcifying
odontogenic cysts, calcifying odontogenic tumors, globulo-
maxillary cysts, ameloblastomas, odontogenic keratocysts
and periapical pathosis.
[5,8]
The usual radiographic feature is a
well-dened radiolucency with corticated or sclerotic border.
Internally, radiopacities develop in two-third of cases. One
tumor may be completely radiolucent, other may contain
faint radioapaque foci, and some may show dense clusters
of radiopacities, sometimes appearing as small pebbles.
[9]

Displacement of neighboring teeth due to tumor expansion
is much more common than root resorptions.
[5,8,9]
To acquire
additional information of AOT, CT, ultrasonography, and
magnetic resonance imaging, should be performed, as these
imaging modalities correspond to the macroscopic ndings
of the histopathological examination.
Adenomatoid odontogenic tumor is composed of odontogenic
epithelium in a variety of histoarchitectural patterns, and
characterized by slow but progressive growth.
[5,10]
Immunohistochemically, the classical AOT phenotype
is characterized by a cytokeratin (CK) prole similar to
follicular cyst and/or oral or gingival epithelium based
on positive staining with CK5, CK17, and CK 19. On the
other hand, the classical AOT is negative for CK4, CK10,
CK13, and CK18.
[8]
Takahashi et al.
[11]
observed a positive
staining for iron-binding proteins (transferring, ferritin)
and proteinase inhibitor (alpha-one-antitrypsin) in various
cells of AOT indicating their role to the pathogenesis of
AOT. Finally, Gao et al.
[12]
studied the expression of bone
morphogenic protein.
Treatment includes complete surgical excision because the
tumor is noninvasive, encapsulated, and is separated easily
from the bone. Recurrence rate is 0.2%. The treatment
of choice is enucleation and simple curettage, but in case
of large tumors partial resection, en bloc of jaw has been
suggested. Use of lyophilized bone and guided tissue
regeneration has been indicated in cases where removal of
the lesion leaves a large exposed bony cavity.
Many cases have been reported affecting maxilla,
[5]
mandible,
[8]

and few cases affecting maxillary sinus,
[7]
but none so far has
been reported with the extension into ethmoidal sinus along
with the involvement of antrum. Normally the tumors do
not exceed 1-3 cm in greatest diameter.
[10]
Our case revealed
a large, gigantic tumor in 17-year-old female patient which
initially was slow growing, but exhibited noticeable growth
since the last 2 months. The CT scan revealed destruction
of the medial wall of right antrum with involvement of
the ethmoidal sinus on the ipsilateral side, deviation of the
nasal septum and complete obliteration of the nasal cavity.
Complete excision of the tumor along with the tooth did
not show any resorption of roots. Some large tumor has
been associated with a recurrence which might indicate its
neoplastic nature. Taking all the controversies surrounding
it into account, our patient are under regular 8 months
follow-up with no recurrence observed till date.
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Cite this article as: Kalra P, Rai S, Sharma S, Tripathi S, Ali A. An unusually
large adenomatoid odontogenic tumor with involvement of the ethmoidal sinus:
A rare case report. J Oral Maxillofac Radiol 2014;2:68-70.
Source of Support: Nil. Conict of Interest: None declared.
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