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.2024.e4337
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Braz Dent Sci 2024 July/Sept;27 (3): e4337
This is an Open Access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in
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Recurrent odontogenic keratocyst and the use of Carnoy’s solution:
a case report
Ceratocisto Odontogênico recidivante e o emprego da solução de Carnoy: relato de caso
João César Guimarães HENRIQUES1 , Keren Quaiatti da SILVA2 , Cristiane Angélica de Paiva PAULA3 , Lívia Bonjardim Lima4 ,
Luiz Fernando Barbosa de PAULO4 , Paulo Rogério de FARIA5
1 - Universidade Federal de Uberlândia, Área de Diagnóstico Estomatológico. Uberlândia, MG, Brazil.
2 - Universidade Federal de Uberlândia, Residente Cirurgia e Traumatologia Bucomaxilofacial. Uberlândia, MG, Brazil.
3 - Centro Universitário Instituto Master de Ensino Presidente Antônio Carlos-Medicina. Araguari, MG, Brazil.;
4 - Universidade Federal de Uberlândia, Departamento de Cirurgia e Traumatologia Bucomaxilofacial. Uberlândia, MG, Brazil
5 - Universidade Federal de Uberlândia, Departamento de Morfologia. Uberlândia, MG, Brazil.
How to cite: Henriques JCG, Silva KQ, Paula CAP, Lima LB, Paulo LFB, Faria PG. Recurrent odontogenic keratocyst and the use of
Carnoy’s solution: a case report. Braz Dent Sci. 2024;27(3):e4337. https://doi.org/10.4322/bds.2024.e4337
ABSTRACT
Background: Odontogenic keratocyst is a challenging odontogenic lesion, originating from the epithelial remnants
of the dental lamina, ranging from small cysts to extensive lesions with a high recurrence rate and morbidity
for patients. The sometimes aggressive nature of these pathological entities explains the controversy among
researchers regarding the best classication for these lesions, justifying the fact that these odontogenic cysts were
once dened as benign odontogenic tumors for some years. However, there is consensus among scholars that
it is a disease that requires careful follow-up of affected patients due to the high recurrence rates, especially in
the rst two decades after treatment completion. Objectives: The present study aims to elucidate the case of a
patient with a mandibular odontogenic keratocyst who prematurely discontinued follow-ups and had a signicant
recurrence of the lesion after 7 years. Description: the second treatment was performed using Carnoy’s solution.
Conclusion: Carnoy’s solution is an absolutely essential therapeutic resource in the treatment of odontogenic
keratocysts, as its use signicantly reduces the potential recurrence of the disease.
KEYWORDS
Decompression; Odontogenic cysts; Odontogenic keratocyst and mandible; Odontogenic tumor; Recurrences.
RESUMO
Contexto: Ceratocisto odontogênico é uma lesão odontogênica desaadora, originada a partir dos restos epiteliais
da lâmina dentária, que permeia desde pequenos cistos até extensas lesões com alta taxa de recorrência e
morbidade para os pacientes. O caráter por vezes agressivo destas entidades patológicas, explica as controvérsias
entre os pesquisadores com relação à qual seria a melhor classicação destas lesões, justicando o fato de que
estes cistos odontogênicos já foram por alguns anos denidos como tumores odontogênicos benignos. Entretanto,
é consensual entre os estudiosos que se trata de uma enfermidade que carece de criteriosa proservação dos
pacientes acometidos dado as elevadas taxas de recidiva, especialmente nas duas primeiras décadas após
o tratamento nalizado. Objetivos: O presente estudo objetiva elucidar o caso de um paciente portador de
ceratocisto odontogênico mandibular que abandonou os acompanhamentos precocemente e teve importante
recidiva da lesão após 7 anos. Descrição: Descrição: o segundo tratamento foi realizado empregando-se a solução
de Carnoy. Conclusão: A solução de Carnoy mostrou ser um recurso essencial no tratamento do queratocisto
odontogênico por ter reduzido signicativamente o potencial de recorrência da doença.
PALAVRAS-CHAVE
Descompressão; Cístos odontogênicos; Ceratocisto odontogênico e mandíbula; Tumor odontogênico; Recidivas.
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Henriques JCG et al.
Recurrent odontogenic k er atocyst and the use of Carnoy’s solution: a case r eport
Henriques JCG et al. Recurrent odontogenic keratocyst and the use of Carnoy’s
solution: a case report
INTRODUCTION
The Odontogenic Keratocyst (OKC) is
a relatively common benign cystic lesion of
odontogenic origin with high recurrence rates,
first described by Philipsen in 1956 [1,2].
OKC accounts for about 8 to 11% of all these cysts
of the jaw bones, thus presenting a considerable
incidence and being the third most prevalent in
terms of these cystic lesions [2]. OKC has been the
subject of much controversy and debate among
experts regarding the best classication of this
nosological entity. Proof of this is the change
in its classication from 2005 to 2017, where
the lesion was reclassied by the World Health
Organization as a benign odontogenic neoplasm
instead of an odontogenic cyst, as had been the
case until then. However, in 2017, the lesion was
reclassied again as an odontogenic cyst, and
the orthokeratinized odontogenic cyst became
an independent pathological entity, no longer
considered a variant of the OKC, as its clinical
and radiological characteristics are less aggressive
compared to the OKC. The reclassication of the
OKC occurred because specialists understand
that its behavior, although occasionally locally
aggressive, tends to respond favorably to the
marsupialization process and is only slightly
associated with genetic mutations, except when
associated with Gorlin-Goltz syndrome [3-5].
Clinically, OKC affects the mandible in
about 73% of cases, notably the posterior body
and ascending ramus of the mandible, with
the peculiar characteristic of developing in the
anteroposterior direction and marrow space
of the bone, as opposed to the buccolingual
development. In general, OKCs have a wide
variation in their epidemiological presentations,
encompassing both sexes and a varied range of
age groups. Moreover, the imaging of OKCs is
quite diverse, including uni- and multilocular
lesions, from small dimensions to very extensive
lesions, usually with well-dened edges. Painful
symptoms are rarely reported; however, they may
be present in cases of more extensive lesions or
associated infections, as well as paresthesia in the
area affected by the OKC, since the expansion of
the lesion can cause compression of the nerve
associated with the injury [3,4,6].
The treatment applied to OKCs is also diverse
and depends on each particular situation. Small
lesions are directly enucleated and subjected to
curettage or complementary peripheral adjuvant
ostectomy. Lesions more than two centimeters in
their largest diameter are usually marsupialized
first, which favors metaplastic change in the
cystic epithelium, with loss of keratin and
epithelial thickening. Such tissue changes
resulting from the marsupialization process
signicantly facilitate the subsequent removal
of the cyst, which originally has a very friable
cystic capsule that is prone to fragmentation.
The fact is that these lesions are eminently
recurrent, and because of this, the indication
of Carnoy’s solution use after enucleation
and curettage or cystic osteotomy is currently
a consensus issue among researchers [7-9].
However, the occasional difficulty of health
services in having all the components of Carnoy’s
solution available may have contributed to its use
being sometimes underestimated, which would
imply an increase of approximately 17% in the
disease’s recurrence rates [10]. In light of the
issues addressed previously, the present study
aims to report the case of a patient with OKC and
signicant recurrence after 7 years from the initial
treatment, discussing the importance of periodic
follow-up and the use of Carnoy’s solution as a
tool of solid scientic evidence in favor of the
therapeutic success of this cystic lesion and the
substantial minimization of potential recurrences.
CASE REPORT DESCRIPTION
A 16-year-old male patient attended the Oral
pathology clinic of a of the Dentistry course at the
Federal University of Uberlândia,complaining of a
slowly growing swelling in the left side of his face
at mandibular region with occasional symptoms
painful.The anamnesis revealed that the patient
had no comorbidities, and the extraoral physical
examination showed an increase in volume on the
left side of the mandible, with a hardened consistency
and slightly sensitive to palpation, causing mild
facial asymmetry. The intraoral evaluation identied
an oral mucosa of normal coloration in the swollen
area (Figures 1, 2, 3, 4 and 5).
For a better diagnostic evaluation, a pan-
oramic radiograph of the patient was performed,
revealing an extensive well-defined osteolytic,
radiolucent, and unilocular image, approximately
5 cm in its largest diameter, causing thinning of
the mandibular base. The lesion involved the
mandibular body region, extending to the man-
dibular ramus and involving tooth 38. For better
imaging detail and therapeutic planning, a cone-
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Henriques JCG et al.
Recurrent odontogenic k er atocyst and the use of Carnoy’s solution: a case r eport
Henriques JCG et al. Recurrent odontogenic keratocyst and the use of Carnoy’s
solution: a case report
beam computed tomography was also performed,
allowing the visualization of the lesion’s expansion
in both the buccal and lingual directions through
multiplanar reconstructions in axial, coronal, and
sagittal sections, as well as panoramic and 3D
views (Figures 4, 5, 6 and 7).
Based on the clinical examination and
complementary exams, such as the various
sections of the cone-beam and helical CT
scans, the diagnostic hypotheses considered in
decreasing order of probability were Odontogenic
Keratocyst, Dentigerous Cyst, Ameloblastic
Fibroma, or Unicystic Ameloblastoma. Therefore,
given the apparent absence of dental resorptions
Figure 1 - Patient at first consultation, showing volumetric change
in the left mandibular region.
Figure 2 - There is a volumetric increase in the left mandible.
Figure 3 - Oral mucosa in the posterior region of the mandible, on
the left side, showing normal coloration and the presence of mild
edema.
Figure 4 - View of the panoramic image from a cone-beam computed
tomography, showing the extent of the osteolytic lesion in the body
region of the left mandible, unilocular, extending from tooth 36 to
the area of tooth 38, which is presented as displaced superiorly in
the mandibular ramus region.
Figure 5 - Image obtained from a cone-beam CT scan, in a sagittal
section, illustrating the lesion in it anteroposterior direction.
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Henriques JCG et al.
Recurrent odontogenic k er atocyst and the use of Carnoy’s solution: a case r eport
Henriques JCG et al. Recurrent odontogenic keratocyst and the use of Carnoy’s
solution: a case report
of the involved elements and the considerable
antero-posterior growth, the possibility of an
Odontogenic Keratocyst was primarily suggested.
Consequently, aspiration was performed in the
area of the lesion, revealing a predominantly
yellowish fluid content with whitish areas
suggestive of keratin. At Therefore, an incisional
biopsy was performed through curettage of
the cystic capsule, and the histopathological
examination result was consistent with an
Odontogenic Keratocyst (Figures 8 and 9).
Given the extent of the lesion, at the same
surgical time as the biopsy was performed, it
was decided to proceed with the progressive
decompression of the lesion through cystic
marsupialization (Figures 10 and 11). The patient
Figure 6 - Axial section of a helical CT scan showing a unilocular
lesion, without septa or locules, with well-defined peripheral
borders, affecting the left mandibular region, with ex- pansion of
the lingual and buccal cortices. The lesion is observed in close
contact with teeth 37 and 36.
Figure 7 - Coronal view, bone window, region of the mandibular
first molars, showing a unilocular, radiolucent, homogeneous
lesion, causing expansion of the buccal cortex of the mandible and
displacement of the inferior alveolar neurovascular plexus to the
basal region of the mandible, which is reduced in size.
Figure 8 - Histopathological aspect of the lesion, demonstrating the
parakeratinized stratified squamous lining epithelium characteristic
of the OKC.
Figure 9 - Histopathological image illustrating areas suggestive of
the presence of satellite cysts in the cystic capsule. (HE, 400X).
Figure 10 - Incisional biopsy obtained by inci- sion in the vestibular
floor region near teeth 36 and 37, followed by mucoperiosteal flap
elevation exposing the lateral surface of the body of the mandible.
Figure 11 - Image demonstrating the area of marsupialization, with
continuous suture to maintain the edges in position and ensure
hemostasis.
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Henriques JCG et al. Recurrent odontogenic keratocyst and the use of Carnoy’s
solution: a case report
was then instructed on the importance of proper
daily hygiene of the cavity and the periodic
monthly returns to the clinic for clinical and
imaging follow-ups. During the follow-up visits,
the patient showed some negligence with local
care in the region of the lesion, and only after
1 year and 9 months from the decompressing
procedure was it possible to perform the denitive
enucleation of the lesion with additional curettage
and extraction of tooth 38 (Figures 12 and 13).
Finally, the patient was reminded of the
importance of periodic follow-up visits, through
clinical examination and imaging of the lesion,
which should be quarterly for the rst two years
due to its high potential for recurrence. Despite
this, in the second year of follow-up, the patient
and his family stopped attending the clinic
and did not respond to attempts to reschedule
appointments. Then, after 7 years from the
cystic enucleation, the patient, now 23 years old,
contacted the specialized service complaining of
a new increase in volume in the left mandibular
area, this time associated with fever and pain with
the presence of pus. An antibiotic combination
of Amoxicillin 500mg and Metronidazole 250mg
for 7 days was then prescribed, and a new
computed tomography exam was performed,
which identied the existence of a large recurrent
lesion, this time with a multiloculated appearance,
approximately 8.5 cm in size, extending from the
vicinity of the left mandibular notch to the apex
of tooth 35 (Figures 15 to 17).
The patient underwent another aspiration
and incisional biopsy, conrming the recurrence
of a Keratocyst, and then a new marsupialization
followed by monthly follow-up were scheduled
(Figure 18). Eight months after marsupialization,
Figure 12 - After marsupialization, the surgery for enucleation of
the lesion was performed, followed by curettage and extraction of
tooth 38.
Figure 13 - Intraoperative view of the surgical wound suture during
enucleation surgery.
Figure 14 - Immediate postoperative panoramic radiograph.
Figure 15 - Panoramic reconstruction obtained from cone-beam
tomography illustrating the characteristics of the lesion’s recurrence,
noting radiographic changes such as: a shift from unilocular to
multilocular lesion with lobulated peripheral borders, measuring up
to 8.5 cm in its greatest extent.
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Recurrent odontogenic k er atocyst and the use of Carnoy’s solution: a case r eport
Henriques JCG et al. Recurrent odontogenic keratocyst and the use of Carnoy’s
solution: a case report
and now with the patient’s collaboration with the
proposed treatment plan, as they were already in
adulthood and attending appointments without
family members, the lesion signicantly reduced
in size, allowing for the planning of a new
surgery. This time, the procedure involved cystic
enucleation, curettage, and vigorous ostectomy
in the surrounding lesion areas, complemented
with the use of Carnoy’s solution.
The patient has been under follow-up for a
year now, aware of the need to attend periodic
check-ups and without signs of new recurrence.
DISCUSSION
Odontogenic Keratocysts are developmental
odontogenic cysts formed from the epithelial
remnants of the dental lamina or from cells,
which are usually found in the gingival tissue and
alveolar bone. Among odontogenic cysts, they are
relatively common lesions, and mutations in tumor
suppressor genes, especially the PTCH1 gene, are
present in a considerable percentage of cases. This
is one of the factors that justify its controversial
nature, sometimes presenting with typical cystic
characteristics and sometimes with characteristics
of a benign neoplasm. Its high recurrence rate,
association with syndromic conditions (Gorlin-
Goltz syndrome), and potential for signicant
antero-posterior growth also support the view of
some scholars that the disease is closer to a benign
neoplasm than to a cystic lesion [11]. However, in
the 2017 World Health Organization classication
of head and neck tumors, later ratied in the latest
2022 publication, the Odontogenic Keratocyst
was again categorized among the developmental
odontogenic cysts. In any case, the need for
periodic and long-term follow-up, usually for
more than 10 years, highlights the greater
severity of this lesion compared to most other
jaw cysts. Perhaps the only cyst with similar
aggressiveness to this odontogenic cyst is the
glandular odontogenic cyst [12,13].
Figure 16 - Axial view of the helical tomography, bone window
illustrating the expansion of bone cortices, anteroposterior extent,
poorly defined borders, and primarily the peripheral lobu- lation and
loculation within the lesion.
Figure 17 - Helical tomography in a coronal sec- tion, bone tissue
window, shows lateromedial and superoinferior expansion as well as
peripheral lobulation and loculation.
Figure 18 - Marsupialization.
Figure 19 - Postoperative image of approximately 1 year.
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Recurrent odontogenic k er atocyst and the use of Carnoy’s solution: a case r eport
Henriques JCG et al. Recurrent odontogenic keratocyst and the use of Carnoy’s
solution: a case report
A recurrence rate of 25-30% is reported in the
literature for odontogenic keratocysts, indicating a
high recurrence rate for a cystic lesion. Incomplete
enucleations, usually hindered by a friable cystic
capsule and the potential persistence of small satellite
cysts, would explain these percentages. For this
reason, the treatment of odontogenic keratocysts
involves more aggressive therapies ranging from
enucleations followed by curettage or osteotomies
to surgical resections for larger and more aggressive
lesions [13,14]. It’s worth highlighting the literature
foundation regarding the choice of performing the
decompressive procedure of marsupialization for
larger lesions. Therefore, these lesions not only
respond positively to this decompressive procedure
but also undergo metaplastic transformation of
the epithelium, loss of keratin, and thickening
of the cystic capsule, making the subsequent
enucleation easier due to the dimensional regression
of the lesion [15,16]. In the present case report,
both at the initial diagnosis of the keratocyst
and at the recurrence of the lesion, the patient
underwent marsupialization which responded
favorably, leading to a progressive decrease in the
lesion’s size, despite the patient’s poor cooperation.
The decient hygiene of the lesion cavity resulting
from marsupialization and the patient’s failure to
attend from the second year of follow-up post-
enucleation, underscore how necessary it is for the
team to have frank, enlightening, and sometimes
repetitive dialogue with the adolescent patient and
their family members about the seriousness of the
lesion in question, its high potential for recurrence,
and the undesirable consequences of a potential
disappearance for the future prognosis of the disease.
Especially considering individuals with lower
educational levels and understanding, it falls upon
the involved professionals to have the necessary
sensitivity to perceive whether the communication
was indeed effective and the information about
the continuation of care was properly assimilated.
The case in question seemingly reects some failure
in this professional-patient communication.
Despite the support for treatment through
surgical resections of large keratocysts, the
signicant morbidity for patients undergoing this
therapeutic modality has led many professionals
to opt for enucleation followed by curettage or
osteotomies [12]. And in order to make this more
conservative treatment modality more efcient,
the addition of Carnoy’s solution has become an
important resource in favor of a considerable
decrease in the recurrence rates of the disease and
exponential improvements in prognoses [17,18].
A mixture of 3ml of chloroform, 6ml of absolute
ethanol, 1ml of acetic acid, and 1g of ferric
chloride composes the so-called Carnoy’s solution,
which acts by promoting a chemical cauterization
of the cystic lesion bed after its enucleation
and curettage, by applying the medication to
the bony defect of the lesion for about three
minutes and subsequent irrigation with saline
solution [18,19]. However, studies have shown
that chloroform, a component of Carnoy’s
solution, is a potential carcinogen. In light of
this, Carnoy’s solution was modied by removing
chloroform from its composition. Nevertheless,
studies have indicated the low efcacy of the
modied Carnoy’s solution. A study conducted
by Dashow et al. [20] compared the effects
of Carnoy’s solution versus modied Carnoy’s
solution and the recurrence of OKC when used
as an adjunct treatment in the enucleation and
simple curettage of OKC. The results showed
that the recurrence rates were significantly
higher in the group of patients who used the
modied Carnoy’s solution (35%) compared to
the Carnoy’s solution (10%). The literature has
shown a new and effective adjunct treatment for
the enucleation and curettage of OKC: the topical
application of 5-uorouracil. This agent is an
antimetabolite frequently used to treat basal cell
carcinoma (BCC). Since the PTCH gene is present
in most cases of OKC, and neoplastic growth
results from PTCH1 mutations that activate the
Smoothened (SMO) and Sonic Hedgehog (SHH)
pathways, it is believed that blocking SMO and
SHH may be effective against the recurrence
of OKC. 5-FU works by inhibiting the enzyme
thymidylate synthase (TS), which inuences DNA
synthesis, leading to cell death. Therefore, 5-FU
could be an alternative to the original Carnoy’s
solution and modied Carnoy’s solution in the
treatment of OKC [21].
Although surgical resection results in a
lower recurrence rate of keratocysts (2%),
therapeutic supplementation with the use of
Carnoy’s solution also allows for a relatively
low recurrence (8%) combined with lower
morbidity and higher quality of life for treated
patients, provided that the solution’s application
time does not exceed ve minutes, which could
cause damage to adjacent nerve and vascular
structures [19,22]. In the present case report, the
initial treatment of the patient did not involve the
use of Carnoy’s solution due to a lack of supplies at
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Henriques JCG et al.
Recurrent odontogenic k er atocyst and the use of Carnoy’s solution: a case r eport
Henriques JCG et al. Recurrent odontogenic keratocyst and the use of Carnoy’s
solution: a case report
that time. Despite this, the patient’s proper annual
attendance at the reference service could have
prevented the recurrent lesion from presenting
with such exacerbated dimensions, which would
facilitate new treatment. Fortunately, in treating
the extensive recurrent lesion, the use of Carnoy’s
solution for three minutes was possible, and the
patient continues without recurrences after the
rst-year post-enucleation and curettage with
osteotomy, which necessitates that there be no
absences from annual follow-ups for at least the
next ten years.
CONCLUSION
The Odontogenic Keratocyst is a cyst that
sometimes behaves in a challenging manner and
may even resemble benign neoplasms. Given
the high recurrence rate of the lesion and the
long follow-up time required (over 10 years), it
becomes mandatory to have clear and effective
communication from professionals to patients
and their families to ensure proper adherence to
the follow-up over the years. Carnoy’s solution
is an absolutely essential therapeutic resource in
the treatment of odontogenic keratocysts, as its
use signicantly reduces the potential recurrence
of the disease.
Author’s Contributions
JCGH: Writing – Original Draft Preparation,
Writing – Review & Editing. KQS: Writing –
Original Draft Preparation, Writing – Review &
Editing. CAPP: Writing – Review & Editing. LBL:
Review. LFBR: Writing – Review & Editing PRF:
Writing – Review & Editing.
Conict of Interest
The authors have no conicts of interest to
declare.
Funding
This research did not receive any specic
grant from funding agencies in the public,
commercial, or not-for-prot sectors.
Regulatory Statement
This study was conducted in accordance with
all the provisions of the local human subjects
oversight committee guidelines and policies: Free
consent was signed by the patients.
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Keren Quaiatti da Silva
(Corresponding address)
Universidade Federal de Uberlândia, Uberlândia, MG, Brazil.
Email: kerenquaiatti.cd@gmail.com Date submitted: 2024 Apr 14
Accept submission: 2024 Sept 22
