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Histopathological Insight of an Adenomatoid Odontogenic Lesion - A Cyst,

Tumor or Hamartoma?

Article · September 2014

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Karandeep Singh Arora

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 Histopathological Insight of an Adenomatoid
Odontogenic Lesion - A Cyst, Tumor or Hamartoma?
Dr. Prabhpreet Kaur, Dr. Madhusudan Astekar, Dr. Karandeep Singh Arora

Abstract
What in the past had been referred to as adenomatoid odontogenic tumor (AOT) is now recognized to consistently represent
a lumen lined with a specific type of epithelial proliferation arising from Hertwig’s Epithelial Root Sheath (HERS),
and is therefore now identified as an adenomatoid odontogenic cyst (AOC). This paper presents a case of adenomatoid
odontogenic cyst in a 16 year old female and explains various histopathological variants of the lesion as analysed by
the oral pathologist. Emphasis has been laid on determining its various terminologies in the past, and thus analysing the
dilemma in classifying this interesting lesion as a true neoplasm, an anomalous hamartomatous growth or a cystic lesion
justifying our present knowledge about the lesion.
Keywords: Adenomatoid odontogenic tumor, adenoameloblastoma, cyst, hamartoma, histopathology, tumor.

Introduction
The odontogenic tumors comprise a group of rare neoplasias
that corresponds to about 1% of all tumors.[1] It is believed that
they are derived from the remnant tissues of tooth formation.
[2]
Adenomatoid odontogenic tumor (AOT), rightfully called
the master of disguise was first documented in literature
by Steensland (1905) as epitheliomaadamantinum.[3-5] It
represents approximately 3-7% of all odontogenic tumors.[6,7]
Over the years a variety of terminologies have been used to
designate this extremely fascinating entity.[3]
Terminologies: In 1950, Bernier and Tiecke were the first
to publish a case using the term adenoameloblastoma. It
described what they concluded to be a histologic variant of the
ameloblastoma.[8] In 1957, Lucas questioned the relationship
of this tumor to the ameloblastoma. Through increased
knowledge of its behaviour and clinical presentation, it
became apparent that the “adenoameloblastoma” was
indeed a separate entity.[9,10] In 1958, Gorlin and Chaudry,
in their discussion of adenoameloblastoma, emphasized
the inappropriateness of this term and pointed out distinct
differences between adenomatoid lesion and ameloblastoma.
[9]
In 1969, Philipson and Birn proposed the name adenomatoid
odontogenic tumor.[3,7] To distance adenomatoid lesion from
ameloblastoma, the terms odontogenic adenomatoid tumor
and adenomatoid odontogenic tumor were introduced.[9]
Later AOT was adopted in the initial edition of World Health
Organisation (WHO)’s histological typing of odontogenic
tumors, jaw cysts and allied lesion in 1971 and retained in the
second edition of WHO in 1992.[7]
The WHO histological typing of odontogenic tumors, jaw
cyst and allied lesions (2005) has defined AOT as a tumor
of odontogenic epithelium with duct-like structures and with
varying degree of inductive changes in the connective tissue.
The tumor may be partly cystic in its presentation, and in
some cases the solid lesion may be present only as masses in
the wall of a large cyst.[3,5]
John Keynes stated that ‘The difficulty lies not in new ideas but
in escaping old ones.’ Today we recognise that adenomatoid
odontogenic tumor is not a tumor at all but rather a cyst that
has a hamartomatous intraluminal proliferation of epithelial
cells derived from HERS. While at times this proliferation
may fill the lumen to give impression of a solid tumor, a close
inspection will reveal its emergence from an epithelial lining.
Calcifications seen in these cysts, which represent attempts
of the root sheath epithelium to induce root dentin, have
been identified as dentinoid material. Therefore, the more
appropriate term is adenomatoid odontogenic cyst or AOC.[9]
Here we present a case of AOC analysing and describing its
various histopathological diversities which are evident in this
particular case.
Case Report
A 16 year old female patient reported to the clinic with a chief
complaint of swelling in upper front tooth region since two
months. Patient was undergoing orthodontic treatment from
last seven to eight months. There was history of accidental
trauma with respect to the region four years back. Extra oral
examination revealed diffuse swelling involving right anterior
maxilla with moderate obliteration of naso-labial fold causing
facial asymmetry. Intraoral examination revealed solitary,
unilateral, well-circumscribed swelling approximately 2
x 3 cm2 with well-defined margins obliterating the labial
vestibule. On palpation, the swelling was firm in consistency,
non-tender in nature with well-defined borders. It was not
associated with any pain or tenderness but was causing
discomfort to patient.
Radiographic examination showed a well-circumscribed
radiolucency measuring approximately 2.5 cm in diameter

26 ASIA PACIFIC DENTAL JOURNAL, Vol. 1, issue 2, Sept to Dec 2014

having a well-defined radioopaque border seen in respect to
periapical region of tooth # 12 (Fig 1).
Differential Diagnosis: Adenomatoid odontogenic cysts that
appear without radiographic evidence of calcification will
be most suggestive of the more common dentigerous cyst.
In this young age group, other strictly radiolucent lesions
worthy of consideration include an odontogenic keratocyst,
an ameloblastic fibroma, an odontogenic myxoma or a central
giant cell tumor as well as an ameloblastoma as the age
increases beyond 14 years.
Cysts in which calcifications can be observed resemble
a calcifying odontogenic cyst. Other mixed radiolucent‐
radiopaque lesions possible in this young age group include an
ameloblastic fibro-odontoma and an ossifying fibroma. As age
increases beyond 14 years, a calcifying epithelial odontogenic
tumor (CEOT) may also be considered even though a CEOT
is uncommon at any age and even less common in individuals
younger than 25 years.
Fine needle aspiration cytology from the lesion yielded 1 ml
of straw coloured fluid mixed with blood. On examination,
lesion showed proteinaceous fluid with few red blood cells,
polymorphonuclear lymphocytes and macrophages. However,
no definitive diagnosis could be made.
Complete surgical excision was done under local anaesthesia
and tissue was sent for histopathological examination.
Macroscopic features of excisional biopsy received showed
two bits of soft tissue, roughly ovoid in shape, blackish grey
in colour having irregular surface texture, measured about 1.5
x 1.0 cm2 and showed no colour changes on pressing.
The Haematoxylin and Eosin stained soft tissue section under
scanner view showed numerous tumor cells arranged as
solid areas, whorled nodules, rosettes and streams. Dentine
or cementum like eosin stained material was seen which
indicated AOC’s histogenesis from Hertwig root sheath.
A thick fibrous capsule was seen surrounding the lesion
(Fig 2a). A well-demarcated cystic lining was present with
amorphous eosinophilic material present within the cystic
lumen. The epithelial lining was proliferative in nature with
exophytic epithelial cell growth into the lumen (Fig 2b).
Excessive proliferative growth of this cystic lining may lead
to complete obliteration of the lumen at a later stage, thus
giving it a typical tumor-like appearance without the cystic
characteristics.
Low power view showed duct-like structures or cyst-
like spaces of varying size along with lattice work pattern
characteristic of AOC. Numerous small and large blood
vessels, extravasated RBC’s were also present indicating its
high vascularity. Fine, fibrillar, eosinophilic material at the
epithelium-connective tissue interface was visible (Fig 2c). A
typical rosette was appreciated with cribriform pattern at its
periphery. A smaller rosette with an obliterating lumen had
few cells arranged in a plexiform pattern surrounding it. Pale,
ASIA PACIFIC DENTAL JOURNAL, Vol. 1, issue 2, Sept to Dec 2014
homogenous eosin stained calcifications were also seen (Fig
2d).
High power view showed cuboidal or columnar cells lining the
tubular or duct-like structures. The nuclei were polarised, that
is, they were present towards the basement membrane, away
from the lumen. A typical eosinophilic rim of varying thickness
at the periphery of the lumen was evident (Fig 2e and 2f).
Anastomosing strands of basaloid epithelial cells resembling
cell rests of dental lamina were appreciated arranged in a
rosette like configuration (Fig 2g). Homogenously stained,
pale, eosinophilic calcifications present in varying quantities
with brightly stained outlines were seen within the cystic
lumen which was lined by cuboidal and columnar epithelial
cells thus suggesting its cystic characteristics (Fig 2h).
Interestingly, present case showed many histomorphological
diversities that could be appreciated and are of diagnostic
value for AOC. Overall features were thus suggestive of
Adenomatoid Odontogenic Cyst.
Discussion
Adenomatoid odontogenic tumor is an uncommon benign
odontogenic lesion that affects young patients associated with
an impacted tooth, usually canine.[7,11] The origin of AOT
is controversial. It occurs within the tooth bearing areas of
jaws and is often found in close association with embedded
teeth, having cytological features similar to those of various
components of the enamel organ, dental lamina, reduced
enamel epithelium and/or their remnants.[12]
Glickman et al stated that whether AOT is a hamartomatous
growth or a true neoplasm, ‘such a controversy is irresolvable
because sound arguments can be advanced in favour of and
against both hypotheses. The arguments are based on personal
bias rather than on scientific evidence.’[12-14] In 2009, Garg
et al stated certain unusual findings based on the clinical,
radiographic and histological features that supported its
neoplastic nature.[12] The relatively small size of tumor and
lack of recurrences in most cases support the fact that it is
a hamartoma.[3] On the contrary, few authors suggest that
early detection could be the reason for small size of lesion.
[3,12]
Gadewar et al in 2010 proposed a cystic variant of AOT
justifying the controversy of it being a cyst of a tumor.[15]
Adenomatoid odontogenic cyst (AOC) is a cyst arising
from the root sheath epithelium. It will characteristically
have a lumen lined by epithelium from which exuberant
proliferations fill much and sometimes all of the lumen space,
thus mimicking a solid tumor.[9] This cyst has sometimes
been referred to as ‘two-thirds tumor’ because about two-
thirds occur in the maxilla, two-thirds occur in young women
(preteen and teenage years), two-thirds are associated with
an unerupted tooth and two-thirds of those teeth are canine
teeth. The two-thirds statistics vary slightly, but the rough
distribution is accurate.[7,9,12]
27
 Figure 1: Orthopantomograph showing a well defined radiolucency with a radio-opaque border.

Figure 2: a) Cells arranged in whorled nodules, sheets, interlacing strands, nest-like patterns and tubular arrangements (H & E,
4x magnification). b) Proliferation of epithelial cystic lining into the lumen (H & E, 4x magnification). c) Duct-like structures
of varying size along with lattice-work pattern (H & E, 10x magnification). d) Rosette of tumor cells seen along with cellular
areas of cribriform and plexiform patterns (H & E, 10x magnification). e) Columnar cells lining tubular or duct-like structure
with an eosinophilic rim at the periphery of the lumen (H & E, 40x magnification). f) Duct-like structure with an eosinophilic
rim of increased thickness (H & E, 40x magnification). g) Spindle shaped cells arranged in a rosette-like arrangement (H &
E, 40x magnification). h) Pale, eosinophilic, irregular calcifications seen within the cystic lumen (H & E, 40x magnification).

28 ASIA PACIFIC DENTAL JOURNAL, Vol. 1, issue 2, Sept to Dec 2014

AOT is generally intraosseous, but can occur rarely in
peripheral locations. It is mostly encountered in young
patients, especially in the second decade of life, and is
uncommon in patients older than 30 years.[12] It is frequently
asymptomatic and is discovered during routine radiographic
examination. Larger lesions may however cause painless
expansion of the bone.[10]
Philipsen et al reported three clinico-topographic variants
of adenomatoid odontogenic tumor (AOT). Follicular AOT
associated with the crown of an embedded tooth most
frequently a permanent canine; an extrafollicular variant,
having no pericoronal or other relationship to an embedded
tooth; and a peripheral (epulis-like) variant, located in
gingival mucosa clinically appearing as a gingival fibroma or
fibrous epulis. The fact that all AOT variants show identical
histological features strongly pointed towards a common
origin.[16,17]
The present case is of interest because of the diverse
histological features that were appreciated in a single case.
Histologically, as many as 20 different patterns of AOT have
been described in the literature.[3]
Presence of an intact capsule in most of the cases reinforces
benign nature of AOT.[3] Garg et al have reported a case of
unencapsulated AOT that caused root resorption and was fast
growing in nature.[12]
Mechanism of formation of tubular structures is not entirely
clear but it is likely because of the secretory activity of tumor
cells, which appear to be preameloblasts. These structures are
not true ducts and no glandular elements are present in the
tumor.[10] Few authors believe it to be due to a cystic change
in the follicles of tumor islands or probably an attempt to form
glandular tissue since the origin is from basal cells of oral
epithelium that has multiple differentiation capacity.[3,18]
Small foci of calcifications, scattered throughout have been
interpreted as abortive enamel formation. Some AOTs contain
larger areas of matrix material or calcification interpreted as
dentinoid or cementum.[10] Almost all will have histologically
identifiable dentinoid calcifications, but in only 50% cases
will they be sufficiently large or coalesced to appear on a
panoramic or periapical radiograph.[9] Calcifications can
be seen in the form of irregular masses, leisegang rings,
spheroidal and globular forms.[3]
Other than the above described features, histological features
like ribbon-like pattern, sieve-like pattern, trabaecular pattern,
necrosis, hyalinization, melanin pigmentation, dysplastic
dentinoid, osteodentin, presence of mitotic features, nuclear
pleomorphism and nuclear hyperchromatism have also been
reported in the literature.[3]
Conclusion
The search for an ideal terminology and accurate place
in the classification system still continues instigating the
ASIA PACIFIC DENTAL JOURNAL, Vol. 1, issue 2, Sept to Dec 2014
pathologists to further study this fascinating lesion and
know more about its histomorphological diversity. Certain
immunohistochemical studies conducted reinforce the theory
of hamartomatous character of this lesion indicating AOT is
not a true neoplastic lesion.[19] The term AOC, as stated by
Marx and Stern[9], seems to be a better suitable term based
on histological features studied for this lesion and is gaining
widespread awareness among people of the fraternity.
References
1. Ochsenius G, Escobar E, Godoy L, Penafiel C. Odontogenic
cysts: analysis of 2,944 cases in Chile. Med Oral Patol Oral Cir
Bucal 2007;2:E85-91.
2. Tjioe KC, Oliveira DT, Poleti ML, Ferreira Jr O, Sant’Anna
E, Gonçales ES. Adenomatoid odontogenic tumour displacing
multiple teeth in an adolescent. Open Journal of Stomatology
2012;2:146-8.
3. Mutalik VS, Shreshtha A, Mutalik SS, Radhakrishnan R.
Adenomatoid odontogenic tumor: A unique report with
histological diversity. Journal of Oral and Maxillofacial
Pathology 2012;16:118-21.
4. Mohamed A, Singh AS, Raubenheimer EJ, Bouckaert MMR.
Adenomatoid odontogenic tumour: review of the literature and
an analysis of 33 cases from South Africa. International Journal
of Oral and Maxillofacial Surgery 2010;39:843-6.
5. Handschel J, Depprich RA, Zimmermann AC, Braunstein S,
Kübler NR. Adenomatoid odontogenic tumor of the mandible:
review of the literature and report of a rare case. Head Face
Med 2005;1.
6. Wood NK, Goaz PW. Differential Diagnosis of Oral &
Maxillofacial Lesions. 5th ed. St. Louis, Missouri: Mosby;
1997.
7. Vasudevan K, Kumar S, Vijayasamundeeswari SV.
Adenomatoid odontogenic tumor, an uncommon tumor.
Contemporary clinical dentistry 2012;3:245.
8. Bernier JL, Tiecke RW. Adenoameloblastoma: Report of
nine cases. Oral Surgery, Oral Medicine, Oral Pathology
1956;9:1304-17.
9. Marx RE, Stern D. Oral & Maxillofacial Pathology: A Rationale
for Diagnosis & Treatment. 2nd ed. Chicago: Quintessence
Publishing Co; 2012.
10. Neville BW, Damm DD, Allen CM, Bouquot JE. Oral &
Maxillofacial Pathology. 3rd ed. New Delhi: Elsevier; 2005.
11. Rajendran R, Sivapathasundharam B, eds. Shafer’s Textbook of
Oral Pathology. 7th ed. New Delhi: Elsevier; 2012.
12. Garg D, Palaskar S, Shetty V, Bhushan A. Adenomatoid
odontogenic tumor–hamartoma or true neoplasm: a case report.
Journal of Oral Science 2009;51:155-9.
13. Philipsen H, Reichart P, Zhang K, Nikai H, Yu Q. Adenomatoid
odontogenic tumor: biologic profile based on 499 cases. Journal
of Oral Pathology & Medicine 1991;20:149-58.
14. Kurra S, Gunupati S, Prasad P R, Raju S, Reddy BVR. An
Adenomatoid Odontogenic Cyst (AOC) with an Assorted
Histoarchitecture: A Unique Entity. Journal of Clinical and
Diagnostic Research 2013;7:1232-5.
29
 15. Gadewar DR, Srikant N. Adenomatoid odontogenic tumor:
Tumor or a cyst, a histopathological support for the controversy.
Int J Pediatr Otorhinolaryngol 2010;74:333-7.
16. Philipsen H, Samman N, Ormiston I, Wu P, Reichart P. Variants
of the adenomatoid odontogenic tumor with a note on tumor
origin. Journal of Oral Pathology & Medicine 1992;21:348-52.
17. Philipsen H, Reichart P. Adenomatoid odontogenic tumour:
facts and figures. Oral oncology 1999;35:125-31.
18. Oehlers FA. The so called adenoameloblastoma. Oral Surg Oral
Med Oral Pathol 1961;14:712-25.
19. Sempere FJV, Martinez MJA, Sirera BV, Marco JB. Follicular
adenomatoid odontogenic tumor: immunohistochemical study.
Med Oral Patol Oral Cir Bucal 2006;11:E305-8.
30
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Dr. Prabhpreet Kaur, MDS
Senior Lecturer, Department of Oral and Maxillofacial
Pathology, B.R.S. Dental College & General Hospital,
Panchkula, Haryana (India)
Dr. Madhusudan Astekar, MDS, Ph.D
Professor and Head, Department of Oral and Maxillofacial
Pathology, Institute of Dental Sciences, Bareilly, Uttar
Pradesh (India)
Dr. Karandeep Singh Arora, MDS
Senior Lecturer, Department of Oral Medicine, Diagnosis
& Radiology, Daswani Dental College & Research Center,
Kota, Rajasthan (India)
ASIA PACIFIC DENTAL JOURNAL, Vol. 1, issue 2, Sept to Dec 2014