UNIVERSIDADE ESTADUAL PAULISTA
“JÚLIO DE MESQUITA FILHO”
Instituto de Ciência e Tecnologia
Campus de São José dos Campos
CASE REPORT DOI: https://doi.org/10.4322/bds.2023.e3823
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Braz Dent Sci 2023 Oct/Dec;26 (4): e3823
Calcifying odontogenic cyst with AOT-like features: a case report
and literature review
Cisto odontogênico calcificante com características adenomatóides: relato de caso e revisão de literatura
Lorena Vieira SACRAMENTO1 , Ianna Josefa Valeska de Aniz CASTRO2 , Leonardo M. G. FIGUEIREDO3 ,
Braúlio CARNEIRO JUNIOR4 , Jean Nunes dos SANTOS2 , Águida Cristina Gomes HENRIQUES2
1 - Universidade Federal da Bahia, Laboratório de Patologia Cirúrgica. Salvador, BA, Brazil.
2 - Universidade Federal da Bahia, Laboratório de Patologia Cirúrgica, Programa de Pós-graduação em Odontologia e Saúde. Salvador,
BA, Brazil.
3 - Universidade Federal da Bahia, Departamento de Cirurgia e Traumatologia Bucomaxilofacial. Salvador, BA, Brazil.
4 - Universidade Estadual do Sudoeste da Bahia. Jequié, BA, Brazil.
How to cite: Sacramento LV, Castro IJVA, Figueiredo LMG, Carneiro Junior B, Santos JN, Henriques ACG. Calcifying odontogenic cyst
with AOT-like features: a case report and literature review. Braz Dent Sci. 2023;26(4):e3823. https://doi.org/10.4322/bds.2023.e3823
ABSTRACT
Odontogenic lesions are a heterogeneous group of diseases that presents differences in their biological behavior
and the occurrence of variable inductive interactions. Calcifying odontogenic cyst (COC), or Gorlin’s cyst, is
a well-recognized example of these lesions. We describe a case of COC with AOT-like areas and highlights its
morphological diversity. A 60-year-old pheoderma man presented with a large swelling in the anterior buccal
region of the mandible. Panoramic radiography revealed a well-dened, unilocular, radiolucent lesion associated
with important root resorption. Complete enucleation of the lesion was performed and the histopathological
ndings met the criteria for the diagnosis of COC, although the cyst exhibited unusual AOT-like features. The
patient has been recurrence free for 6 months after surgery. COCs with AOT-like features are rare, and reect the
multipotentiality and complexity of the inductive effects of the odontogenic epithelium with the ectomesenchyme.
Enucleation seems to be the most indicated treatment, similar to classical COC.
KEYWORDS
Adenomatoid odontogenic tumor; Calcifying odontogenic cyst; Odontogenic cysts; Oral pathology; Oral surgery.
RESUMO
As lesões odontogênicas são um grupo heterogêneo de patologias que apresentam diferenças no seu comportamento
biológico, e ocorrência de interações indutivas variáveis. O cisto odontogênico calcicante (COC), ou cisto de Gorlin, é
um exemplo bem conhecido destas lesões. Descrevemos um caso de COC com áreas adenomatóides e destacamos a sua
diversidade morfológica. Paciente do sexo masculino, 60 anos de idade, apresentou um aumento de volume na região
anterior da mandíbula. A radiograa panorâmica revelou uma lesão bem denida, unilocular e radiolúcida associada
a uma reabsorção radicular importante. A enucleação completa da lesão foi realizada e os achados histopatológicos
preencheram os critérios para o diagnóstico de COC, embora o cisto exibisse características adenomatóides pouco usuais.
O paciente permanece livre de recidivas durante 6 meses após a cirurgia. Os COCs com características adenomatóides
são raros, e reetem a multipotencialidade e complexidade dos efeitos indutivos do epitélio odontogênico com o
ectomesênquima. A enucleação parece ser o tratamento mais indicado, semelhante ao COC clássico.
PALAVRAS-CHAVE
Tumor odontogênico adenomatóide; Cisto odontogênico calcicante; Cistos odontogênicos; Patologia oral;
Cirurgia oral.
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Braz Dent Sci 2023 Oct/Dec;26 (4): e3823
Sacramento LV et al.
Calcifying odontogenic cyst with AO T-like features: a case r eport and literatur e re view
Sacramento LV et al. Calcifying odontogenic cyst with AOT-like features: a case
report and literature review
INTRODUCTION
Calcifying odontogenic cyst (COC), or
Gorlin’s cyst, is a rare developmental odontogenic
cyst characterized histologically by ghost cells,
which often calcify [1]. This lesion was first
reported in 1962 [2]. However, the World
Health Organization (WHO) has changed its
classication over the years, and it was called
calcifying cystic odontogenic tumor (CCOT) for
12 years, because it was believed in its neoplastic
potential [3]. COC accounts for less than 1% of
all odontogenic cysts and is part of the group of
ghost cell lesions of the jaws [1,4,5].
Clinically, COC usually presents as a
slow-growing painless swelling, with a slight
predilection for the anterior maxilla [4,6].
Radiographically, COC appears as a well-
dened unilocular or multilocular radiolucency
that can contain variable amounts and shapes
of radiopaque material [4]. In some of cases,
the cyst is associated with an impacted tooth,
frequently a canine [4].
Histopathologically, COC exhibits a brous
cystic wall lined with epithelium whose basal cells
are columnar or cuboidal. The suprabasal layers
resemble the stellate reticulum [5]. Numerous
ghost cells and calcications are identied within
the epithelial lining. Masses of ghost cells often
pass into the connective tissue of the cyst wall,
eliciting a foreign body reaction and inducing
dentinoid [1]. Cysts may show intraluminal
and/or mural epithelial proliferation producing
ameloblastoma-like areas [1,5].
Odontogenic lesions mainly develop due
to the capacity of the odontogenic epithelium
to undergo differentiation and mesenchymal
induction, which gives these lesions a wide
variety of morphological features [7,8].
Interestingly, COC can occur in association with
odontogenic tumors, frequently odontoma [9],
ameloblastoma [10], ameloblastic broma [11],
adenomatoid odontogenic tumor (AOT) [7] or
with odontogenic keratocyst (OK) [12].
This study describes a case of COC with AOT-
like areas and highlights the great differentiation
potential and morphological diversity of this cyst.
CASE REPORT
A 60-year-old pheoderma man, with a one-
year history of a large swelling on the left side
of the anterior mandible, attended at the Oral
and Maxillofacial Surgery and Traumatology
Service of Santo Antônio Hospital - Irmã Dulce
Social Works (Figure 1A and 1B). The patient
had no history of extraoral trauma to the region
and reported a history of tobacco and alcohol
use. Intraoral examination revealed a large
lesion of hardened consistency exhibiting mild
pain upon palpation. The mass was covered
with intact mucosa and there were no signs
of infection (Figure 1C and 1D). Pulp vitality
tests showed sensitivity loss in the anterior
teeth (3.1 to 3.4). Panoramic radiography
revealed a well-dened, unilocular, radiolucent
lesion extending from the sagittal midline
(Figure 2). The lesion caused important
resorption at the roots of the anterior teeth
(3.3 and 3.4). Aspiration yielded a serous
yellowish fluid and the first hypothesis was
unicystic ameloblastoma (UA).
An incisional biopsy was performed and
sent to the Surgical Pathology Laboratory of the
Federal University of Bahia, where the study was
conducted. The report was compatible with a
ghost cell odontogenic lesion suggestive of COC.
Then, an excisional biopsy was obtained and
complete enucleation of the lesion and peripheral
ostectomy were performed. During surgery, the
surgeon observed the extent of the lesion from the
region of the 3.4 to the 4.3. All teeth associated
with the lesion were removed.
The specimen was sent for histopathological
analysis, which revealed the presence of a cystic
fibrous wall lined with ameloblastomatous
epithelium (Figure 3A) whose basal columnar
cells resembled ameloblasts and exhibited
a palisade arrangement and inverted
nuclear polarity. The upper epithelial layers
resembled the stellate reticulum of the enamel
organ (Figure 3B). Ghost cells were found
interspersed, with the fusion of these cells
forming amorphous acellular eosinophilic
material (Figure 3B). The epithelial component
together with the ghost cells proliferated
into the cystic lumen and toward the brous
capsule (Figure 3C and 3D). Dentinoid material
was also observed amidst the epithelium and
capsule (Figure 3E).
These histopathological features were
consistent with COC. Interestingly, the epithelium
proliferating into the cystic lumen exhibited
morphological features not commonly observed
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Braz Dent Sci 2023 Oct/Dec;26 (4): e3823
Sacramento LV et al.
Calcifying odontogenic cyst with AO T-like features: a case r eport and literatur e re view
Sacramento LV et al. Calcifying odontogenic cyst with AOT-like features: a case
report and literature review
in COC. They were represented by numerous
spindle-shaped and oval epithelial cells. Narrow
strands of epithelial cells were also found
(Figure 4A). Duct-like structures of variable size
were observed amidst this proliferation, and were
lined with a single layer of cells which exhibited
AOT-like features, as it showed terminal bar
(Figure 4B and C).
These histopathological characteristics were
consistent with COC with AOT-like features.
Figure 1 - Extraoral and intraoral views. (A and B) Note large swelling on the left side of the anterior mandible; (C and D) Large lesion covered
with intact mucosa.
Figure 2 - Panoramic radiograph. Note large circumscribed radiolucent image extending from the sagittal midline and resorption in the roots
of the teeth 3.3 and 3.4.
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Braz Dent Sci 2023 Oct/Dec;26 (4): e3823
Sacramento LV et al.
Calcifying odontogenic cyst with AO T-like features: a case r eport and literatur e re view
Sacramento LV et al. Calcifying odontogenic cyst with AOT-like features: a case
report and literature review
The patient continues under clinical and
radiographic follow-up and has shown no signs of
recurrence after 6 months of follow-up (Figure 5).
To support the discussion of the case
presented, a literature review was carried out in
the Medline databases. The descriptors “calcifying
odontogenic cyst”, “adenomatoid odontogenic
tumor”, “AOT-like”, “calcifying cystic odontogenic
tumor” and “hybrid” were used to search for case
reports published in English in all years.
Figure 3 - Histological sections of COC stained with hematoxylin-eosin. (A) Note cystic lumen (asterisk) and fibrous wall lined with
ameloblastomatous epithelium (arrow) (
40x
); (B) Detail of the basal columnar cells with a palisade arrangement and inverted nuclear polarity
(arrow). The upper epithelial layers resembled the stellate reticulum of the enamel organ. Note clusters of ghost cells forming amorphous
acellular eosinophilic material (asterisk) (
200x
); (C) Proliferation of epithelial component and ghost cells (arrows) into the cystic lumen
(asterisk) (
40x
); (D) Epithelial and ghost cells in the fibrous capsule (arrows) (
100x
); (E) Detail of the dentinoid material in the fibrous capsule
(asterisk) (
200x
).
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Braz Dent Sci 2023 Oct/Dec;26 (4): e3823
Sacramento LV et al.
Calcifying odontogenic cyst with AO T-like features: a case r eport and literatur e re view
Sacramento LV et al. Calcifying odontogenic cyst with AOT-like features: a case
report and literature review
Figure 4 - Histological sections of COC stained with hematoxylin-eosin. (A) Note narrow strands of epithelial cells (arrows) and duct-like
structures of variable size (arrowhead) (
100x
); (B) AOT-like area demonstrating the presence of duct-like structures lined with a single layer of
cells (arrowhead) (
200x
); (C) Detail of the terminal bar in duct-like structures (arrowhead) (
400x
).
Figure 5 - Panoramic radiograph. Osseo repair after two months (A) and six months (B) of lesion removal.
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Braz Dent Sci 2023 Oct/Dec;26 (4): e3823
Sacramento LV et al.
Calcifying odontogenic cyst with AO T-like features: a case r eport and literatur e re view
Sacramento LV et al. Calcifying odontogenic cyst with AOT-like features: a case
report and literature review
DISCUSSION
This study reported the case of a 60-year-
old man with COC involving the anterior
mandible that exhibited unusual features.
The histopathological diagnosis of COC at this
site is not surprising since other odontogenic cysts
have been described in this region, including COC
with AOT-like areas [13].
COC was rst described by Gorlin et al. [2] in
1962. In 2005, in view of the complexity of clinical
and histopathological characteristics presented
by this group of lesions, the WHO classified
COC as a benign cystic neoplastic variant of
odontogenic origin, and renamed it CCOT [3].
Additionally, the lesions with invasive biological
behavior and tumor growth have been described
as dentinogenic ghost cell tumor (DGCT) and,
together with ghost cell odontogenic carcinoma
(GCOC), the three entities form the group of ghost
cell lesions of the jaws [3]. Over the years, some
classications were proposed, and a very useful
and comprehensive classication that includes
all cystic and solid subtypes of COC has been
suggested by Ledesma-Montes et al. [6]. Recently,
based on its behavior and clinicopathological
features, the WHO renamed the CCOT as COC,
a developmental cyst that originates from dental
lamina [5].
Finally in 2022, the classification of the
COC as a development odontogenic cyst was
consolidated. The scientic evidence conrms
the cystic nature and indolent behavior of the
lesion and therefore no change occurred in its
nomenclature and classication [14].
COC have been found in association with
odontogenic tumors such as odontoma [9],
ameloblastoma [10], ameloblastic broma [11],
and AOT [7], or with OK [12]. COC associated
exclusively with AOT is rare [6-8,13,15].
The mechanism that causes the occurrence
of two odontogenic lesions together is not
well known. However, several theories have
been proposed to explain the phenomenon,
including a transformation of one lesion into
another, a collision of two separate lesions,
and an inductive effect of one lesion into
another [11]. Thus, although extremely rare,
it is not unexpected that COC gives rise to
AOT because of the multipotentiality of the
odontogenic epithelium [7,8].
The histopathological ndings of the present
case meet the criteria for the diagnosis of
COC [4,5]. However, part of the cystic epithelium
that proliferated into the lumen exhibited some
features resembling those seen in AOT, such as
spindle-shaped and oval cells forming duct-like
structures with an evident terminal bar. It is
important to state that terminal bar is not present
in microcysts and this aspect is important to the
differential diagnosis. Additionally, cells clustered
in a cord-like arrangement were observed in focal
areas.
It should be noted that there was no
collision of two odontogenic lesions, COC and
AOT, in the present case. Furthermore, other
features necessary for the characterization
of AOT, such as the presence of rosette-like
structures, were absent. Thus, there are not
sufficient criteria to classify this case as a
hybrid odontogenic lesion. The morphological
features observed suggest an unusual pattern of
differentiation of part of the epithelial component
of COC, since the odontogenic epithelium has
a diverse differentiation potential under the
inuence of ectomesenchyme and also due to
its multipotentiality. The multipotentiality of
odontogenic tissues favors the biological event
of transdifferentiation in some odontogenic cysts
and tumors, without inuencing the biological
behavior of the lesions. The present case therefore
meets the histopathological criteria for COC with
AOT-like features.
Although previous studies [6-8,13,15] have
reported an association between COC and AOT,
we highlight some differences and similarities
compared to the present case. The microscopic
findings described by Zeitoun et al. [13] are
very similar to our case in which the AOT-like
component appeared to arise from the epithelial
component that proliferated into the cystic
lumen, but differed by the presence of epithelial
rosettes. Also, in contrast to the present case,
Soares et al. [7] detected an AOT with evident
features such as the presence of epithelial cells
with secretory activity forming solid nodules,
which contained some rosettes and several duct-
like structures adjacent to the COC.
Balaji and Rooban [8] published a case of
COC with unusual features, including AOT-like
areas. The authors attributed this morphological
diversity to the pluripotent cells present in the
epithelial lining of COC, which could give origin to
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Braz Dent Sci 2023 Oct/Dec;26 (4): e3823
Sacramento LV et al.
Calcifying odontogenic cyst with AO T-like features: a case r eport and literatur e re view
Sacramento LV et al. Calcifying odontogenic cyst with AOT-like features: a case
report and literature review
odontogenic tumors. Narrow anastomosed cords
alongside duct-like structures were observed, in
agreement with the present case. However, the
rosette-like pattern was also described. As in
our case, the authors of the latter study did not
consider the lesion to be a hybrid and called it
COC with AOT-like areas. According to Balaji and
Rooban [8], if the case had been diagnosed at a
later stage, it is likely that a COC and an AOT
would have been clearly found.
The present case concurs in some aspects
with previous studies on COC. According to
Ledesma-Montes et al. [6] and literature review of
Arruda et al. [4], there is a male predilection and
COC occurs over a broad age range. Concerning
localization, there is no consensus, but the
anterior maxilla seems to be the site of greatest
occurrence, and the cyst commonly manifests
as a painless swelling with a mean size ranging
from 3 to 4 cm [4,6]. In the present case, the
cyst occurred in a 60-year-old male patient, was
located in the anterior mandible, and was of
large size.
A search of the English-language literature
for cases of COC with AOT-like areas identied
ve case reports [6-8,13,15]. These cases are
summarized in Table I.
Most authors classied the cyst as a hybrid
odontogenic lesion and only one case was called
COC with unusual features [8], similar to the
present case. These aspects were a limitation of
the literature review carried out. Analysis of the
clinical data (Table I) showed male-to-female
ratio: 1.5:1, the mean age was 22.8 years (range,
2-43 years), with 3 cases in the maxilla and
2 in the mandible, and there appeared to be a
preference for the anterior region.
Radiographically, COC appears as a well-
dened unilocular or multilocular radiolucency
that can contain variable amounts and shapes of
radiopaque material. The cyst may be associated
with an impacted tooth and root resorption and
divergence of adjacent roots are frequent [6].
The present case manifested as an extensive
radiolucent, unilocular lesion associated with
important root resorption and root divergence.
Foci of radiopaque material and impacted teeth
were absent.
The clinical and radiographic features of the
present case can mimic other odontogenic lesions,
including glandular odontogenic cyst, UA, OK
and residual radicular cyst. Although UA is more
common in young patients and is generally
associated with an impacted tooth, it was the
rst hypothesis because it is the most frequent
and because the unilocular radiographic image
favored this clinical hypothesis [1]. Analyzing
the radiographic features of COC with AOT-like
areas (Table I), similar to the present case, all
cysts had a unilocular appearance and only one
case was associated with root resorption [13].
Few cases exhibited radiopaque foci [7,13] or
were associated with an impacted tooth [7].
COC is considered a cyst of non-invasive
biological behavior and enucleation has been
the treatment of choice [5], including cases with
AOT-like areas (Table I). In this case report,
the patient showed significant resolution of
the mandibular radiolucency and no signs of
recurrence 6 months after surgery. According to
Table I - Epidemiological data of COC with AOT-like areas published in English language
Age,
years Sex Location Clinical
Diagnosis Size, cm Radiopaque
clusters Treatment Reference
15 Female Posterior
Mandible X X No Enucleation with curettage Freedmanetal. [15]
35 Male Anterior
Mandible CCOT 5 Yes Enucleation with apicetomy
of adjacente teeth Zeitounetal. [13]
19 Male Posterior
Maxilla X 4 X X Ledesma-Montesetal. [6]
2Female Anterior
Maxilla AFO 3 Yes Enucleation without
associated tooth Soaresetal. [7]
43 Male Anterior
Maxilla PG 1.7 No Enucleation Balaji and Rooban [8]
60 Male Anterior
Mandible UA 5 No Enucleation with curettage Present
X - Not avaliable; CCOT - Calcifying Cystic Odontogenic Tumor; AFO - Ameloblastic Fibro-Odontoma; PG - Periapical Granuloma; UA -
Unicystic Ameloblastoma.
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Sacramento LV et al.
Calcifying odontogenic cyst with AO T-like features: a case r eport and literatur e re view
Sacramento LV et al. Calcifying odontogenic cyst with AOT-like features: a case
report and literature review
Ledesma-Montes et al. [6], cases of isolated or
combined COC are indeed associated with low
recurrence.
This study reported a case of COC with
AOT-like features, highlighting the morphological
diversity of odontogenic lesions. These cysts
are uncommon and we believe that each case is
indeed unique and has its particularities, since the
inductive effects between odontogenic epithelium
and ectomesenchyme are complex. In addition, the
multipotentiality of the odontogenic epithelium
favors a diversied potential for differentiation.
Finally, COC with AOT-like features does not
seem to differ about the biological behavior from
that of classical COC when the period of follow-up
of the present case is considered.
Acknowledgements
We thank the Laboratório de Anatomia
Patológica, Faculdade de Odontologia da UFBA,
Salvador, BA, Brazil, for help with the study.
Author’s Contributions
LVS: Investigation, Data Curation, Writing
– Original Draft Preparation, Writing – Review
& Editing, Visualization. IJVAC: Writing –
Original Draft Preparation, Writing – Review &
Editing, Visualization. LMGF: Conceptualization,
Investigation. BCJ: Conceptualization,
Investigation. JNS: Investigation, Writing –
Review & Editing. ACGH: Conceptualization,
Investigation, Writing – Original Draft Preparation,
Writing – Review & Editing.
Conict of Interest
The authors declare no conict of interest.
Funding
None.
Regulatory Statement
A signed consent form was obtained from
the patient to disclose the case.
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Braz Dent Sci 2023 Oct/Dec;26 (4): e3823
Sacramento LV et al.
Calcifying odontogenic cyst with AO T-like features: a case r eport and literatur e re view
Sacramento LV et al. Calcifying odontogenic cyst with AOT-like features: a case
report and literature review
Águida Cristina Gomes Henriques
(Corresponding address)
Universidade Federal da Bahia, Faculdade de Odontologia, Laboratório de Patologia
Cirúrgica, Salvador, BA, Brasil.
Email: aguidacgh@gmail.com Date submitted: 2023 Mar 08
Accept submission: 2023 Sep 06
