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
).