Oral Oncology EXTRA (2005) 41, 316318

http://intl.elsevierhealth.com/journal/ooex

CASE REPORT

Management of an unusually large

adenomatoid odontogenic tumor
a,*
Kishore Shetty , Sotirios Vastardis b, Peter Giannini c

a
Medically Complex Patient Clinic, University of Texas Dental Branch, Houston, Texas, USA
b
Department of Periodontics, LSU School of Dentistry, Louisiana, USA
c
Department of Oral Biology, College of Dentistry, University of Nebraska Medical Center, USA

Received 6 August 2005; accepted 9 August 2005

KEYWORDS Summary The present case is a rare report of an adenomatoid odontogenic tumor
Adenomatoid; (AOT) in the maxilla of a 12-year-old African-American male involving an impacted
Odontogenic; canine and bicuspid and its subsequent management.
Tumor; c 2005 Elsevier Ltd. All rights reserved.
Management

Case report togenic cyst, calcifying epithelial tumor, odonto-

A 12-year-old African-American male was referred
by his orthodontist for evaluation of a maxillary
radiolucent lesion noticed on a panoramic radio-
graph (Fig. 1). The medical history was insignificant
and the patient was in good general health. The pa-
tient was asymptomatic and clinical exam did
expansion of both vestibular and lingual osseous
cortical plates. The patient had no nerve deficit
or adenopathy in the face or neck. An orthopanto-
mogram revealed the presence of a significant uni-
locular radiolucent area with well-defined sclerotic
borders, involving an embedded upper right perma-
nent canine and first bicuspid with the displace-
ment of second bicuspid. The differential
diagnosis was of dentigerous cyst, calcifying odon-
genic keratocyst, and unicysticameloblastoma.
The patient underwent an incisional biopsy
under local anesthesia. Histological exam revealed
well-developed fibrous connective tissue capsule
with a partially cystic lesion and tumor lobules
(Fig. 2). A profuse complex of rounded, rosette-like
and stream-like aggregates of spindle-shaped
odontogenic epithelial cells, similar in size and
shape to the stratum intermedium cells of the
developing tooth germ with few areas of eosino-
philic material could be seen (Fig. 3). The histolog-
ical sign-out diagnosis was consistent with
adenomatoid odontogenic tumor (Figs. 2 and 3).
Discussion

* Corresponding author. Tel.: +1 713 500 4210. AOT had been initially described and classified as a
E-mail address: orasmile@gmail.com (K. Shetty). variant of the ameloblastoma1,2 having been


1741-9409/$ - see front matter c 2005 Elsevier Ltd. All rights reserved.
doi:10.1016/j.ooe.2005.08.002
Management of an unusually large adenomatoid odontogenic tumor
Figure 1 Panoramic radiograph of the patient reveal-
ing impacted canine and first bicuspid teeth with a
relatively well-defined radiolucent lesion on the right
maxilla.
Figure 2 A hemotoxylin and eosin stained lower mag-
nification view of a well-developed fibrous connective
tissue capsule with a partially cystic lesion and tumor
lobules.
named a adenoameloblastoma, adenoameloblastic
odontoma, pseudoadenomatous ameloblastoma,
cystic complex composite odontoma, unusual pleo-
morphic adenoma-like tumor, ameoloblastic ade-
nomatoid tumor, odonto-ameloblastic tumor,
odonto-ameloblastic odontoma, tumor of enamel
organ, ameloblastic epithelial tumor, and tumor
connected to development cysts3 Pindborg named
it of adenomatoid odontogenic tumor (AOT), classi-
fying it as an odontogenic epithelial tumor present-
ing inductive effect at the connective tissue.4
317
Figure 3 The characteristic rounded and rosette form
lined with a single layer of polarised cuboidal or
columnar epithelium cells giving it the adenomatoid
appearance (H&E 400).
AOT occurs as a well-circumscribed lesion
around the crowns of unerupted anterior teeth of
young patients and consists of epithelium in swirls
and ductal patterns interspersed with spherical cal-
cifications. The more common variant is the folli-
cular type and is often mistaken for a dentigerous
cyst. Histopathologic examination demonstrates
cuboidal or spindle-shaped epithelial cells forming
aggregates or rosette-like structures with minimal
connective tissue, and cuboidal or low columnar
cells forming glandular duct-like structures.
Management of adenomatoid odontogenic
tumor
A review of the literature depicts a lesion in which
conservative management produces a uniformly
excellent outcome without recurrence.5,6 Since
the adenomatoid odontogenic tumor (AOT) is a
benign tumor that presents with a non-aggressive
biologic behavior, progressive growth, small fre-
quency of recurrence, absence of invasion, and
the frequent presence of a connective tissue cap-
sule, the treatment should consist of enucleation
and curettage.710
In the presently reported case the lesion was re-
moved by surgical enucleation. After reflection of
an ample mucosal flap and widened entrance
through the usually thinned and expanded cortical
bone, the connective tissue capsule of the lesion
was encountered. Enucleation was achieved by
separation of the lesion from bone without perfo-
rating the capsule. Inspection, irrigation, and gen-
tle curettage of the resultant cavity remove any
residual lesion. Both the impacted canine and
318
bicuspid incorporated within the lesion were re-
moved. The upper right lateral incisor and the sec-
ond bicuspidwhose radicular bone cover had been
resorbed by the lesion, were endodontically trea-
ted. The healing was uneventful, and there was no
evidence of recurrence 1 year after the surgery.
It should however be noted that in cases when
the tooth involved by the tumor presents itself in
a favorable position for orthodontic movement,
with the objective of preservation of the tooth,
we recommend marsupialization or decompression
as the initial modality of treatment. However,
when the tumor does not decrease in size, or con-
tinues growing during treatment by marsupializa-
tion, immediate surgical removal should be
performed. The position of the impacted tooth,
as well as its localization in maxilla or at the ramus
or body of the mandible are other data that must
be considered when interpreting the radiograph
and selecting the modality of treatment.
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