International Journal of Medical and Dental Case Reports (2015), Article ID 010215, 3 Pages

CASE REPORT

Adenomatoid odontogenic tumor associated with

dilacerated root: An unusual case report
Bijayata Shrestha1, Sushil Subedi2, Sushma Pandey3
1
Department of Oral Pathology, Chitwan Medical College and Hospital, College of Dental Sciences, Bharatpur, Chitwan, Nepal, 2Department of Oral and
Maxillofacial Surgery, Chitwan Medical College and Hospital, College of Dental Sciences, Bharatpur, Chitwan, Nepal, 3Department of Oral Medicine and
Radiology, Chitwan Medical College and Hospital, College of Dental Sciences, Bharatpur, Chitwan, Nepal

Correspondence
Dr. Bijayata Shrestha, Department of Oral
Pathology, College of Dental Science, Bharatpur,
Chitwan, Nepal. Phone: +977-01-9841439711,
Fax: +977-056-532963.
Email: bijayata_shr@yahoo.com
Received 01 January 2015;
Accepted 20 February 2015
doi: 10.15713/ins.ijmdcr.20
Abstract
Adenomatoid odontogenic tumor (AOT) is a rare benign neoplasm of epithelial
origin. AOT occurs mainly in the second decade of life, and the most common location
is anterior maxilla. It rarely affects the mandible. Most of the AOTs may be located
intraosseously but few have been reported to occur within gingival structures. AOTs
located intraosseously may be seen associated with unerupted tooth (follicular variant)
or may not (extra-follicular variant) be associated with unerupted tooth. Here, we report
a rare case of presumably follicular variant of AOT associated with mandibular canine
that presented as an extra-follicular variant leading to root dilaceration of the mandibular
canine after eruption of the tooth.

How to cite the article: Keywords: Adenomatoid, extra-follicular, follicular, odontogenic, tumor
Shrestha B, Subedi S, Pandey S. Adenomatoid
odontogenic tumor associated with
dilacerated root: An unusual case report. Int J
Med Dent Case Rep 2015;1-3.

Introduction Case Report

In 1969, Philipsen and Birn proposed the widely accepted and the
currently used name adenomatoid odontogenic tumor (AOT),
which was included in the first classification of odontogenic
tumor by histologic typing of World Health Organization
(WHO) in 1971.[1,2] In 2005, WHO had recategorized AOT
as benign odontogenic tumor of epithelial origin with mature,
fibrous stroma without Odontogenic ectomesenchyme.[3] Some
of the investigators even believe it to be a hamartoma.[2,4]
AOT is an uncommon benign odontogenic tumor occurring
with a relative frequency of 2.2-7.1%.[5] It exists as three clinical
variants: Follicular (related to unerupted tooth), extra-follicular
(not related to unerupted tooth), and peripheral (attached to
the gingival structures).[6] AOT is referred to as “2/3rd tumor”
because 2/3rd of the cases occur in the maxilla, 2/3rd of the cases
occur in young woman, 2/3rd of the cases occur in unerupted
teeth and 2/3rd of the cases have been found to arise in relation
to anterior tooth particularly canine.[7]
Here we report a rare case of presumably follicular AOT in
15-year-old female which arose in association of left mandibular
canine and later with eruption of the tooth presented as an
extra-follicular variant leading to the root dilaceration of left
mandibular canine.
A 15-year-old female patient reported with the chief complaint
of the painless swelling in the mandibular anterior region since
6 months. The swelling was not associated with any history
of trauma, pain or discharge. On extra-oral examination,
lymphadenopathy was not observed. On extra-oral examination,
labial cortical plate expansion was seen in relation to tooth
33 with apparently normal overlying mucosa [Figure 1]. On
palpation, the swelling was hard and non-tender. Electrometric
pulp testing of tooth 33 suggested that the tooth was vital.
Orthopantomogram showed 33 with dilacerated root and
periapical radiolucency [Figure 2]. The provisional diagnosis of
lateral periodontal cyst was made. On surgical exploration, there
was the buccal cortical plate resorption and the lesion was totally
enucleated. The gross examination of the specimen showed
a single spherical greyish white soft tissue measuring 1 cm ×
1.5 cm and cut surface revealed a solid grayish-white tumor
mass with well-defined capsule [Figure 3]. Microscopy revealed
multisized solid nodules of cuboidal and columnar epithelial
cells forming nests or rosette-like structure with minimal stromal
connective tissue and spindle shaped cells in the spaces between
the epithelial nodules which are encapsulated with a thick,

1
Shrestha, et al.
inflamed fibrous capsule [Figure 4a]. Ameloblast-like epithelial
cells forming duct-like structures, eosinophilic amorphous
tumor droplets amongst the epithelial cells; leisegang ring
formation and eosinophilic amorphous amyloid like deposits
were observed [Figure 4b]. The final histopathological diagnosis
of AOT was obtained. The patient was kept under regular
follow-up. The patient has not shown any signs of recurrence in
the 6 months follow-up period after surgery.
Discussion
AOT is a rare benign odontogenic neoplasm with predilection
for occurrence in females and occurs mainly in the second
decade of life as seen in our case.[2,6-8] The lesions usually present
as asymptomatic jaw swelling and are relatively small in size not
exceeding 1-3 cm in diameter, as seen in this case.[8]
AOT exists as 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
Figure  1: Intraoral photograph showing labial cortical plate
expansion on the mandibular anterior alveolus in relation to
teeth  33
Figure 2: Orthopantomogram showing periapical radiolucency in
relation to dilacerated tooth 33
2
Adenomatoid odontogenic tumor
unerupted tooth, and the peripheral form which is the most rare
(3%) and occurs within gingival mucosa.[6,9]
On radiographs, the intraosseous follicular variant of AOT
shows a well-delineated, unilocular radiolucency surrounding
the crown of a retained tooth, a picture indistinguishable from
dentigerous cysts. Minute radiopacities around the retained
tooth may be found in AOT and are considered a characteristic
but not pathognomonic finding. About two out of every three
AOT show distinct radiopaque calcification on radiographs.[6,9]
The extra-follicular variant presents as a unilocular, well-
defined radiolucency found between, above, or superimposed on
the roots of erupted teeth and often resembles a cystic lesion.[6]
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.[10] The case reported here is considered
uncommon as it involved the mandible. Our case is also unique as
it is associated with dilacerated tooth and root dilaceration within
Figure  3: Cut surface of the gross specimen that revealed a solid
grayish-white tumor mass with well-defined capsule
a b
Figure  4: (a) Multisized solid nodules forming and amyloid like
eosinophilic deposits that are encapsulated with a thick, regular
inflamed fibrous connective tissue (×10). (b): Solid nodules of
cuboidal and columnar epithelial cells forming nests and rosette-
like structure along leisegang ring formation and spindle-shaped
cells in the spaces between the epithelial nodules (×40)
Adenomatoid odontogenic tumor
the lesion are only infrequently reported in the literature.[11] We
hypothesize that the AOT in this patient evolved as a follicular
variant associated with 33. After the eruption of 33, there could
be shift of position of the lesion, which impeded and changed the
pathway of development of the root resulting in root dilaceration.
Considerable amount of debate is going on regarding the
hamartomatous, neoplastic or cystic nature of AOT.[7] It was
considered to be a hamartoma clinically and histologically, due
to small size of the tumor at the time of diagnosis and the lack of
recurrences after removal. However, it had also been described
as a slow growing neoplasm that is detected at an early stage,
before it reached a clinically noticeable size.[7,11]
Amyloid like material resembling that of calcifying epithelial
odontogenic tumor (CEOT) are also present and the presence
of which has led some workers to propose the existence of
combined AOT and CEOT. However, CEOT like areas were
considered to be within normal histopathological spectrum
of AOT.[12] The calcified materials seen in AOT have been
considered to be an abortive form of enamel, dentin, enamel
and dentin, cementum, dentin, and cementum or dystrophic
calcifications, but their exact nature remains a controversy.[13]
Owning to its benign behavior, slow growth and clear
delimitation, as well as its low tendency to recur, the treatment
of choice for AOTs is conservative surgical excision.[14]
Conclusion
We conclude that the reported case presumably evolved as
follicular variant that later presented as extra-follicular variant
resulting in the dilacerations of associated erupted tooth.
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