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diagnostics
Case Report
Dentigerous Cysts with Diverse Radiological Presentation
Highlighting Diagnostic Challenges
Alexandre Perez 1, * , Vincent Lenoir 2
1
2
3
*
and Tommaso Lombardi 3
Unit of Oral Surgery and Implantology, Division of Oral and Maxillofacial Surgery, Department of Surgery,
University of Geneva & University Hospitals of Geneva, 1205 Geneva, Switzerland
Division of Radiology, Diagnostic Department, Geneva University Hospitals, University of Geneva,
1205 Geneva, Switzerland
Unit of Oral Medicine and Oral Maxillofacial Pathology, Division of Oral and Maxillofacial Surgery,
Department of Surgery, University of Geneva & University Hospitals of Geneva, 1205 Geneva, Switzerland
Correspondence: alexandre.perez@hcuge.ch
Abstract: Dentigerous cyst is an odontogenic developmental cyst arising from the pericoronal tissue
of an impacted tooth, and that may exhibit various radiological aspects. The aim of this article is
to present four cases of histologically confirmed mandibular dentigerous cysts to highlight diverse
radiological presentations: one of classical appearance (well-limited unilocular radiolucent lesion
surrounding the crown) and three which have shown radiological peculiarities (one cyst displacing
the adjacent tooth, with bone but no root resorption, one cyst presenting hallmarks of infection and
one multilocular cyst with thin septa). Such radiologic diversity may, on occasion, suggest a clinical
aggressive lesion such as an odontogenic keratocyst or ameloblastoma. The diagnosis of dentigerous
cyst requires a thorough evaluation of the clinical presentation and accurate radiological studies.
Keywords: odontogenic cysts; dentigerous cyst; jaws; oral surgery; oral pathology; imaging; OPG; CBCT
Citation: Perez, A.; Lenoir, V.;
Lombardi, T. Dentigerous Cysts with
Diverse Radiological Presentation
Highlighting Diagnostic Challenges.
1. Introduction
Diagnostics 2022, 12, 2006. https://
A dentigerous cyst (DC), also known as a follicular cyst, is a cyst of non-inflammatory
odontogenic origin that develops from the pericoronal tissue (dental sac or dental follicle) of
an impacted tooth, either permanent or deciduous or supernumerary [1–6]. Several factors
have been investigated and are known to play an important role in tooth eruption [7]. DCs
represent more than 24% of the cysts of the maxilla [8–10]. Their incidence peaks in the
third decade of life (21–30 years), followed by a gradual decrease with age. It is also slightly
higher in men (sex ratio 1.5 man/L female) [2,4] and is not influenced by ethnicity [8,10].
Furthermore, 2.5 to 4% of patients with an impacted tooth develop a DC [4,8], of which
95% are associated with a permanent tooth and 5% with a supernumerary tooth [2,11,12].
Regarding localization, 74% of DCs localize in the mandible and 26% in the maxilla with a
symmetrical left/right distribution [13–16].
Bilateral lesions are extremely rare, and when occurring, association with development
anomalies and syndromes, such as mucopolysaccharidosis or cleidocranial dysplasia,
should be suspected [1,2,5].
Clinically, DCs are often asymptomatic but may occasionally cause swelling and
dental displacement [8,9,14,15]. More rarely, a DC may be accompanied by pain caused by
superposed infection or paresthesia when mechanical compression on a nerve occurs [11].
DCs are most often diagnosed incidentally during an oral check-up, and the panoramic
dental X-ray (OPG) is generally the most frequent diagnostic imaging technique carried out.
Radiologically, DCs usually appear as well-defined unilocular radiotransparent homogeneous lesions with a round or ovoid shape attached to the cementoenamel junction of an
impacted tooth, usually third molars, and largely in the mandible [9,11,12].
doi.org/10.3390/diagnostics
12082006
Academic Editors: Luca Testarelli
and Shankargouda Patil
Received: 25 July 2022
Accepted: 16 August 2022
Published: 19 August 2022
Publisher’s Note: MDPI stays neutral
with regard to jurisdictional claims in
published maps and institutional affiliations.
Copyright: © 2022 by the authors.
Licensee MDPI, Basel, Switzerland.
This article is an open access article
distributed under the terms and
conditions of the Creative Commons
Attribution (CC BY) license (https://
creativecommons.org/licenses/by/
4.0/).
Diagnostics 2022, 12, 2006. https://doi.org/10.3390/diagnostics12082006
https://www.mdpi.com/journal/diagnostics
Diagnostics 2022, 12, 2006
ried out.
Radiologically, DCs usually appear as well-defined unilocular radiotransparent homogeneous lesions with a round or ovoid shape attached to the cementoenamel junction
of an impacted tooth, usually third molars, and largely in the mandible [9,11,12].
The borders of the lesion may appear sclerotic and are less well-defined when2 of
the
14
cyst becomes infected. When the cyst is large, it may displace or even resorb adjacent dental roots or may induce bone remodelling. However, the bone cortex usually remains intact.
Thethis
borders
of the
may
appear
are less well-defined
the cyst
In
article,
we lesion
present
four
casessclerotic
of DC and
to highlight
the diversewhen
radiological
becomes
infected.
When
the
cyst
is
large,
it
may
displace
or
even
resorb
adjacent
dental
presentation: one of classical appearance (case #2) and three which have shown radiologroots or may induce bone remodelling. However, the bone cortex usually remains intact.
ical peculiarities (case #1, cyst displacing the adjacent tooth, with bone but no root resorpIn this article, we present four cases of DC to highlight the diverse radiological
tion; case #3, cyst presenting hallmarks of infection; case #4, multilocular cyst with thin
presentation: one of classical appearance (case #2) and three which have shown radiological
septa).
peculiarities (case #1, cyst displacing the adjacent tooth, with bone but no root resorption;
case #3, cyst presenting hallmarks of infection; case #4, multilocular cyst with thin septa).
2. Case Series
All Series
the cases presented refer to lower third molars.
2. Case
Case
#1:cases presented refer to lower third molars.
All the
A
30-year-old
male in good general health consulted the Oral Surgery and ImplanCase #1:
tologyAUnit
of
the
Geneva
Hospitals
for pain experienced
for two
local30-year-old male in University
good general
health consulted
the Oral Surgery
anddays
Implantolized
in the
quadrant
of Hospitals
the mandible,
withexperienced
no fever nor
and throat
ogy Unit
ofleft
the lower
Geneva
University
for pain
forear,
twonose,
days localized
in
symptoms.
the left lower quadrant of the mandible, with no fever nor ear, nose, and throat symptoms.
On
On extraoral
extraoral examination,
examination, palpation
palpation at
at the
the angle
angle and
and the
the horizontal
horizontal branch
branch of
of the
the
mandible
was
painful,
and
so
was
intraoral
palpation
at
the
vestibular
level
from
tooth
mandible was painful, and so was intraoral palpation at the vestibular level from tooth
#37
#37
to the
ascending
branch
of the
mandible.
to the
left left
ascending
branch
of the
mandible.
On
On dental
dental examination,
examination, tooth
tooth #37
#37 showed
showed sensibility
sensibility positive
positive test,
test, slightly
slightly painful
painful
percussion,
of of
more
than
9 mm
localized
distally
andand
aspercussion, and
andaapocket
pocketdepth
depthononprobing
probing
more
than
9 mm
localized
distally
sociated
with
suppuration.
associated
with
suppuration.
Panoramic
Panoramic radiography
radiography revealed
revealed the
the presence
presence of
of aa pericoronal
pericoronal radiolucent
radiolucent lesion
lesion
around
the
crown
of
an
impacted
tooth
#38
(horizontally
positioned
with
mesial
orientaaround
crown of an impacted tooth #38 (horizontally positioned with mesial
orientation)
tion)
(Figure
1).lesion
The lesion
measuring
27mm
× 22was
mmwell
wasdefined
well defined
and unilocular,
sur(Figure
1). The
measuring
27 × 22
and unilocular,
surrounded
rounded
a thin bone.
sclerotic
bone. It overlapped
distal
root #37,
of tooth
#37, adjacent
causing
by a thinby
sclerotic
It overlapped
with the with
distalthe
root
of tooth
causing
adjacent
bone resorption
root resorption.
Theseemed
cyst seemed
to interfere
bone resorption
but withbut
nowith
signsnoofsigns
root of
resorption.
The cyst
to interfere
with
the inferior
alveolar
canalcanal
(IAC),
which
appeared
displaced
caudally
by by
thethe
lesion.
A
with
the inferior
alveolar
(IAC),
which
appeared
displaced
caudally
lesion.
presumptive
diagnosis
of of
CD
was
made.
A
presumptive
diagnosis
CD
was
made.
Figure 1. Panoramic X-ray of case #1.
An additional cone-beam computed tomography (CBCT) X-ray examination confirmed
the presence of the large pericoronal cystic lesion attached to the cementoenamel junction
of tooth #38 (Figure 2). We observed a bone expansion accompanied by slight thinning
of the mandibular cortex on the vestibular and lingual sides. The cyst was in contact
with the upper wall of the IAC, which was thinned and caudally displaced, but of normal
width. This lesion had an approximately 8 mm opening on the mucosal alveolar crest and
extended to the distal root of tooth #37 without signs of associated root resorption.
Diagnostics 2022, 12, 2006
firmed the presence of the large pericoronal cystic lesion attached to the cementoenamel
junction of tooth #38 (Figure 2). We observed a bone expansion accompanied by slight
thinning of the mandibular cortex on the vestibular and lingual sides. The cyst was in
contact with the upper wall of the IAC, which was thinned and caudally displaced, but of
normal width. This lesion had an approximately 8 mm opening on the mucosal alveolar
3 of 14
crest and extended to the distal root of tooth #37 without signs of associated root resorption.
Figure
Figure2.2.CBCT
CBCTX-ray
X-rayofofcase
case#1.
#1.
There
periosteal
reaction,
pathological
fracture,
evident
signssigns
of sequesTherewas
wasalso
alsonono
periosteal
reaction,
pathological
fracture,
evident
of sequestration
or sclerosis
the surrounding
nor infiltration
the perimandibular
tration
or sclerosis
of theofsurrounding
bone,bone,
nor infiltration
of theofperimandibular
soft
soft tissues.
#38two
had
two and
roots,
the mesial
rootinto
came
into contact
the
tissues.
Tooth Tooth
#38 had
roots,
theand
mesial
root came
contact
with thewith
lingual
lingual
wall
of the
The radiological
appearance
a DC. Differential
wall
of the
IAC.
TheIAC.
radiological
appearance
evoked aevoked
DC. Differential
diagnosesdiaginnoses included
odontogenic
andameloblastoma.
unicystic ameloblastoma.
Theenucleated
lesion was
cluded
odontogenic
keratocystkeratocyst
and unicystic
The lesion was
in toto
tooth
under local
of DC
inenucleated
toto together
withtogether
tooth #38with
under
local#38
anesthesia.
Theanesthesia.
diagnosis ofThe
DCdiagnosis
was confirmed
was
confirmed by histopathological
examination
(Figure
3).followed
The patient
wasyears.
followed
by
histopathological
examination (Figure
3). The patient
was
for four
At
for
four
years.
At
12
months
follow-up,
OPG
showed
satisfactory
healing
of
the
enucle12 months follow-up, OPG showed satisfactory healing of the enucleation cavity and tooth 4 of 16
Diagnostics 2022, 12, x FOR PEER REVIEW
ation
cavity
and tooth
#37
vitalwas
(Figure
4). The patient
waswith
followed-up
#37
was
still vital
(Figure
4).was
Thestill
patient
followed-up
for 4 years
no signs for
of
4
years
with
no
signs
of
recurrence.
recurrence.
Figure
3. 3.Histopathological
of the
theenucleated
enucleated
specimen
from
#1 showing
an
Figure
Histopathologicalexamination
examination of
specimen
from
case case
#1 showing
an
uninflamed
fibrous
cyst
wall
lined
by
a
thin
cuboidal
epithelial
lining
(H&E,
×40).
uninflamed fibrous cyst wall lined by a thin cuboidal epithelial lining (H&E, ×40).
Diagnostics 2022, 12, 2006
4 of 14
Figure 3. Histopathological examination of the enucleated specimen from case #1 showing an uninflamed fibrous cyst wall lined by a thin cuboidal epithelial lining (H&E, ×40).
Figure 4. Panoramic X-ray of case #1 at one-year follow-up.
Figure 4. Panoramic X-ray of case #1 at one-year follow-up.
Case #2:
#2:
Case
A
38-year-old
female
referred
byby
herher
orthodontist
for for
diA
female in
ingood
goodgeneral
generalhealth
healthwas
was
referred
orthodontist
agnosis
and
treatment
of
a
lesion
in
the
right
lower
quadrant
of
the
mandible
discovered
diagnosis and treatment of a lesion in the right lower quadrant of the mandible discovincidentally
on anon
OPG
X-rayX-ray
performed
prior to
orthodontic
treatment.
The oral
examiered
incidentally
an OPG
performed
prior
to orthodontic
treatment.
The
oral
nation was unremarkable,
and the
patient
was asymptomatic.
examination
was unremarkable,
and
the patient
was asymptomatic.
Diagnostics 2022, 12, x FOR PEER REVIEW
5 of 15
A new
new OPG
OPG X-ray
X-ray revealed
revealed the
the presence
presence of
of aa pericoronal
pericoronal radiolucent
radiolucent lesion
lesion on
on
an
A
an
impactedtooth
tooth#48,
#48,which
whichwas
wasinverted
inverted
mesio-caudal
direction
(Figure
5). The
leimpacted
inin
thethe
mesio-caudal
direction
(Figure
5). The
lesion
sionwell
was defined,
well defined,
measured
13mm,
× 15and
mm,was
and
was surrounded
bysclerotic
a thin sclerotic
was
measured
13 × 15
surrounded
by a thin
border
border overlapping
the
distal
root of
tooth
#47,
which appeared
slightly resorbed.
overlapping
with thewith
distal
root
of tooth
#47,
which
appeared
slightly resorbed.
The cyst
also
with the IAC,
walls
couldwalls
not have
during this
The overlapped
cyst also overlapped
withwhose
the IAC,
whose
couldbeen
not well-identified
have been well-identified
exam.
diagnosis was
a DC. was a DC.
duringThe
thispresumptive
exam. The presumptive
diagnosis
Figure 5. Panoramic X-ray of case #2.
Figure 5. Panoramic X-ray of case #2.
On the
the complementary
complementaryCBCT
CBCTX-ray,
X-ray,it it
was
observed
impacted
tooth
in horiOn
was
observed
anan
impacted
tooth
#48#48
in horizonzontal/inverted
orientation
in mesio-caudal
the mesio-caudal
direction
(Figure
6).two
Theroots
two were
rootsnot
were
tal/inverted
orientation
in the
direction
(Figure
6). The
in
not
in
direct
contact
with
the
right
IAC,
and
the
tooth
crown
did
not
come
into
direct
direct contact with the right IAC, and the tooth crown did not come into direct contact with
contact
the #47.
root The
of tooth
#47. Thespace
pericoronal
space was
markedly
enlarged,
13 mm
the
root with
of tooth
pericoronal
was markedly
enlarged,
13 mm
wide, 17
wide, 17 mm high, and 15 mm long, compatible with a DC. The visible resorption of the
distal root of tooth 47 made a differential diagnosis of odontogenic keratocyst less likely.
The lesion was associated with an alveolar crest dehiscence of over 4 mm in length and
thinned lingual cortex. The lesion displaced the IAC caudally, causing loss of visibility of
Figure 5. Panoramic X-ray of case #2.
Diagnostics 2022, 12, 2006
On the complementary CBCT X-ray, it was observed an impacted tooth #48 in horizontal/inverted orientation in the mesio-caudal direction (Figure 6). The two roots were
5 of 14
not in direct contact with the right IAC, and the tooth crown did not come into direct
contact with the root of tooth #47. The pericoronal space was markedly enlarged, 13 mm
wide, 17 mm high, and 15 mm long, compatible with a DC. The visible resorption of the
distal
47 made
a differential
of odontogenic
keratocyst
less likely.
high, root
and of
15 tooth
mm long,
compatible
with adiagnosis
DC. The visible
resorption
of the distal
root of
The
lesion
was aassociated
an alveolar
crest dehiscence
of over
4 mm The
in length
tooth
47 made
differentialwith
diagnosis
of odontogenic
keratocyst
less likely.
lesionand
was
associated
withcortex.
an alveolar
crest dehiscence
of IAC
overcaudally,
4 mm in length
lingual
thinned
lingual
The lesion
displaced the
causingand
lossthinned
of visibility
of
cortex.
its
wall. The lesion displaced the IAC caudally, causing loss of visibility of its wall.
Figure
Figure6.6.CBCT
CBCTX-ray
X-rayof
ofcase
case#2.
#2.
Teeth
4848
were
extracted,
andand
the the
pericoronal
lesion
in tooth
#38 was
Teeth #18,
#18,28,
28,3838and
and
were
extracted,
pericoronal
lesion
in tooth
#38
enucleated
in totoinunder
local anesthesia.
The diagnosis
of DC of
was
confirmed
by histowas enucleated
toto under
local anesthesia.
The diagnosis
DC
was confirmed
by
pathological
examination.
The patient
was followed-up
for two
years.
At 12
of
histopathological
examination.
The patient
was followed-up
for two
years.
Atmonths
12 months
Diagnostics 2022, 12, x FOR PEER REVIEW
6 of 15
control,
an an
intraoral
X-ray
showed
good
healing
ofofthe
of control,
intraoral
X-ray
showed
good
healing
theenucleation
enucleationcavity
cavityofofthe
thefollicular
follicular
cyst(Figure
(Figure7).
7).Clinically,
Clinically, tooth
tooth #47
#47 remained
remained vital
vital and
and asymptomatic.
asymptomatic.
cyst
Figure7.7.Intraoral
IntraoralX-ray
X-rayofofcase
case#2
#2atatone-year
one-yearfollow-up
follow-upexam.
exam.
Figure
Case#3:
#3:
Case
A
46-year-old
femalein
ingood
goodgeneral
generalhealth
healthconsulted
consultedthe
theOral
OralSurgery
Surgeryand
andImplanImplanA 46-year-old female
tology
Unit
of
the
Geneva
University
Hospitals
for
the
management
of
a
lesion
in
theleft
left
tology Unit of the Geneva University Hospitals for the management of a lesion in the
lowerquadrant
quadrantofofthe
the
mandible
discovered
fortuitously
on OPG
an OPG
X-ray
performed
by
lower
mandible
discovered
fortuitously
on an
X-ray
performed
by her
her dentist.
The patient
a history
of episodic
this
area
over several
dentist.
The patient
had ahad
history
of episodic
pain inpain
this in
area
over
several
months,months,
which
which
she intermittently
treated intermittently
with self-medicated
painkillers
and anti-inflammatory
she
treated
with self-medicated
painkillers
and anti-inflammatory
drugs.
drugs.
The oral clinical examination was unremarkable, and the patient was asymptomatic.
The
oral aclinical
examination
was unremarkable,
the patient
wastooth
asymptomatic.
On the
OPG,
pericoronary
radio-transparent
lesionand
around
impacted
#38 was
discovered
(Figure
8). The involved
tooth was lesion
oriented
mesially.
The lesion
poorly
On the OPG,
a pericoronary
radio-transparent
around
impacted
tooth was
#38 was
disdelimited,
measured
24 ×involved
10 mm, and
stretched
to the apparently
resorbed
root apex
covered (Figure
8). The
tooth
was oriented
mesially. The
lesiondistal
was poorly
deoflimited,
tooth #37.
The roots
overlapped
with the IAC,
raising
suspicion
of
measured
24 ×of10tooth
mm,#38
andclearly
stretched
to the apparently
resorbed
distal
root apex
interference.
of a DC with
infectionof
of tooth #37.The
TheOPG
rootsimages
of toothwere
#38 indicative
clearly overlapped
withpossible
the IAC,secondary
raising suspicion
and
perilesional
sclerosing
osteitis.
interference.
The
OPG images
were indicative of a DC with possible secondary infection
and perilesional sclerosing osteitis.
Diagnostics 2022, 12, 2006
The oral clinical examination was unremarkable, and the patient was asymptomatic.
On the OPG, a pericoronary radio-transparent lesion around impacted tooth #38 was discovered (Figure 8). The involved tooth was oriented mesially. The lesion was poorly delimited, measured 24 × 10 mm, and stretched to the apparently resorbed distal root apex
of tooth #37. The roots of tooth #38 clearly overlapped with the IAC, raising suspicion
6 of of
14
interference. The OPG images were indicative of a DC with possible secondary infection
and perilesional sclerosing osteitis.
Figure 8. Panoramic X-ray of case #3.
Figure 8. Panoramic X-ray of case #3.
The complementary CBCT examination found that tooth #38 was directed mesially
The complementary CBCT examination found that tooth #38 was directed mesially
and slightly lingually (Figure 9) and that it had three roots, with the deformed IAC tortuand slightly lingually (Figure 9) and that it had three roots, with the deformed IAC torously passaging between them. The crown of tooth #38 was close to the distal root of7tooth
Diagnostics 2022, 12, x FOR PEER REVIEW
of 15
tuously passaging between them. The crown of tooth #38 was close to the distal root
of
#37,
whose
apex
was
strongly
resorbed.
The
pericoronal
osteolytic
lesion
measuring
24 ×
tooth #37, whose apex was strongly resorbed. The pericoronal osteolytic lesion measuring
10
× 10
from the
crown-root
junction
of tooth
to #38
the to
partially
resorbed
24 ×
10mm
× 10extended
mm extended
from
the crown-root
junction
of #38
tooth
the partially
resorbed
distal
root of
tooth
#37.was
There
was
bone dehiscence
of on
12 the
mmalveolar
on the alveolar
crest
distal
root
of tooth
#37.
There
bone
dehiscence
of 12 mm
crest and
of
and
of
5
mm
on
the
vestibular
cortex.
The
borders
of
the
lesion
were
in
places
irregular
5 mm on the vestibular cortex. The borders of the lesion were in places irregular and
and strongly
sclerotic,
indicating
a superimposed
infection.
no signs
of bone
strongly
sclerotic,
indicating
a superimposed
infection.
ThereThere
werewere
no signs
of bone
sesequestration,
periosteal
reaction
or associated
fracture.
findings
supported
the
questration,
periosteal
reaction
or associated
fracture.
TheseThese
findings
supported
the initial
initial
diagnosis
of
DC.
diagnosis of DC.
Figure
Figure9.9.CBCT
CBCTX-ray
X-rayof
ofthe
thecase
case#3
#3patient.
patient.
Teeth
Teeth #37,
#37, 38
38 and
and 48
48 were
were extracted
extracted under
under local
local anesthesia.
anesthesia. The
Thelesion
lesionon
on tooth
tooth #38
#38
was
wassubmitted
submitted for
for histopathological
histopathological examination,
examination, which
which confirmed
confirmed the diagnosis of DC.
Case#4:
#4:
Case
A 59-year-old
59-year-oldmale
malewas
wasreferred
referredto
to the
the Oral
Oral Surgery
Surgery and
and Implantology
ImplantologyUnit
Unitof
ofthe
the
A
GenevaUniversity
UniversityHospitals
Hospitalsfor
foraacomprehensive
comprehensiveoral
oralexamination
examinationand
anddental
dentalcare
careprior
prior
Geneva
to the
the start
start of
of radioradio- and
and chemotherapy
chemotherapyfor
forsquamous
squamouscell
cellcarcinoma
carcinomaof
ofthe
thefloor
floorof
of the
the
to
® for epilepsy, Beloc ZOC®® and Atacand®® for high
®
mouth.
The
patient
was
taking
Rivotril
mouth. The patient was taking Rivotril for epilepsy, Beloc
and Atacand for high
bloodpressure
pressureand
andCrestor
Crestor®®for
forhyperlipidemia.
hyperlipidemia.He
Hewas
wasaaheavy
heavysmoker
smoker(70
(70pack
packyear)
year)
blood
andconsumed
consumed60
60ggof
ofalcohol
alcoholaa day.
day.
and
The extraoral clinical examination was unremarkable. Intraorally, the mucosa of the
hard and soft palate as well as the lingual mucosa showed keratotic dots; on the floor of
the mouth, there was a 3 cm long and 1 cm wide ulceration; the oral vestibule presented
a smoker keratosis. Oral hygiene was poor with plaque and tartar deposits; the patient
Diagnostics 2022, 12, 2006
7 of 14
The extraoral clinical examination was unremarkable. Intraorally, the mucosa of the
hard and soft palate as well as the lingual mucosa showed keratotic dots; on the floor of
the mouth, there was a 3 cm long and 1 cm wide ulceration; the oral vestibule presented a
smoker keratosis. Oral hygiene was poor with plaque and tartar deposits; the patient had
severe periodontal disease with grade III furcation involvement of teeth #46, 26 and 16,
polycaries of teeth #16, 14, 13, 12, 23, 25, 46, 45, 43, 42, 41, 32, 33, 34 and 35; teeth #15, 14, 11,
22, 27, 47, 36 and 37 were absent.
On an OPG X-ray, teeth #38 and 48 were impacted and distally orientated (Figure 10).
Tooth #38 showed a radiotransparent lesion measuring 20 × 15 mm, with a well-defined
Diagnostics 2022, 12, x FOR PEER REVIEW
8 of 15
border, multilocular appearance, and a thin septum, most compatible with a DC versus
ameloblastoma versus odontogenic keratocyst.
Figure 10. OPG X-ray of case #4.
Figure 10. OPG X-ray of case #4.
On
CBCT X-ray,
X-ray,the
thedistal
distal
root
of the
impacted
tooth
#38 in
was
in contact
with
the
On CBCT
root
of the
impacted
tooth
#38 was
contact
with the
upper
upper
wall
of
the
left
IAC,
generating
a
discreet
deformation
of
the
canal
(Figure
11).
The
wall of the left IAC, generating a discreet deformation of the canal (Figure 11). The cyst
cyst
showing
a multilocular
was located
adjacent
to the of
crown
tooth
and
showing
a multilocular
aspectaspect
was located
adjacent
to the crown
toothof#38
and#38
seemed
seemed
to be attached
to the
tooth
neck.
Theextending
lesion extending
to the ascending
to be attached
to the tooth
neck.
The
lesion
slightly slightly
to the ascending
branch
branch
of the mandible
measured
in height,
in length
in width.
of the mandible
measured
17 mm17
in mm
height,
21 mm21
inmm
length
and 10and
mm10inmm
width.
In its
In
its caudal
part,
the was
lesion
in contact
thewall
upper
wall
of over
the IAC
over
aboutThe
13
caudal
part, the
lesion
in was
contact
with thewith
upper
of the
IAC
about
13 mm.
mm.
The
wall
was
thinned
but
with
little
impact
on
its
internal
diameter.
The
vestibular
wall was thinned but with little impact on its internal diameter. The vestibular and lingual
and
lingual
were also
strongly
thinned
focally.
There
was noreaction
periosteal
cortex
were cortex
also strongly
thinned
focally.
There
was no
periosteal
or reaction
fracture or
at
fracture
at the mandibular
angle.
Onofthe
bases
of these radiological
thediagnosis
differenthe mandibular
angle. On the
bases
these
radiological
findings, thefindings,
differential
tial
diagnosis
DC, ameloblastoma,
andkeratocyst.
odontogenic keratocyst.
included
DC, included
ameloblastoma,
and odontogenic
The lesion was enucleated in toto, and the teeth extracted (Figure 12) under general
anesthesia, during which the ENT and maxillofacial surgeons proceeded with the placement
of a mandibular osteosynthesis plate, followed by non-interruptive mandibulectomy, and
finally, pelvi-glossectomy with tracheostomy and reconstruction with an anterolateral
thigh flap and neck dissection. Histopathological examination of the cystic lesion allowed
to diagnose a DC. The patient was followed regularly for two years, and no recurrence
was observed.
Diagnostics 2022, 12, 2006
seemed to be attached to the tooth neck. The lesion extending slightly to the ascending
branch of the mandible measured 17 mm in height, 21 mm in length and 10 mm in width.
In its caudal part, the lesion was in contact with the upper wall of the IAC over about 13
mm. The wall was thinned but with little impact on its internal diameter. The vestibular
and lingual cortex were also strongly thinned focally. There was no periosteal reaction or
8 of 14
fracture at the mandibular angle. On the bases of these radiological findings, the differential diagnosis included DC, ameloblastoma, and odontogenic keratocyst.
Diagnostics 2022, 12, x FOR PEER REVIEW
9 of 15
Diagnostics 2022, 12, x FOR PEER REVIEW
9 of 15
Figure 11. CBCT X-ray of case #4.
The lesion was enucleated in toto, and the teeth extracted (Figure 12) under general
anesthesia, during which the ENT and maxillofacial surgeons proceeded with the placement of a mandibular osteosynthesis plate, followed by non-interruptive mandibulectomy, and finally, pelvi-glossectomy with tracheostomy and reconstruction with an anterolateral thigh flap and neck dissection. Histopathological examination of the cystic lesion allowed to diagnose a DC. The patient was followed regularly for two years, and no
Figure
X-ray
recurrence
was observed.
Figure11.
11.CBCT
CBCT
X-rayofofcase
case#4.
#4.
The lesion was enucleated in toto, and the teeth extracted (Figure 12) under general
anesthesia, during which the ENT and maxillofacial surgeons proceeded with the placement of a mandibular osteosynthesis plate, followed by non-interruptive mandibulectomy, and finally, pelvi-glossectomy with tracheostomy and reconstruction with an anterolateral thigh flap and neck dissection. Histopathological examination of the cystic lesion allowed to diagnose a DC. The patient was followed regularly for two years, and no
recurrence was observed.
Figure
tooth
3838
and
inin
toto
enucleation
of of
thethe
cyst
in in
case
#4.#4.
Figure12.
12.Avulsion
Avulsionofof
tooth
and
toto
enucleation
cyst
case
Healing
uneventful
and
thethe
patient
hadhad
no complaints.
Radiologically,
at theat6-the
Healingwas
was
uneventful
and
patient
no complaints.
Radiologically,
and
12-month
follow-up
(Figure
13), 13),
there
waswas
no no
reossification
at the
cystectomy
andand
6- and
12-month
follow-up
(Figure
there
reossification
at the
cystectomy
tooth
which
could
bebe
explained
byby
a side
effect
of postoperative
radiotherapy
andand
tooth3838sites,
sites,
which
could
explained
a side
effect
of postoperative
radiotherapy
chemotherapy.
there
were
nono
signs
of of
enlargement
of the
residual
bone
cavity,
chemotherapy.However,
However,
there
were
signs
enlargement
of the
residual
bone
cavity,
suggesting
a
recurrence
of
DC.
suggesting a recurrence of DC.
Figure 12. Avulsion of tooth 38 and in toto enucleation of the cyst in case #4.
Healing was uneventful and the patient had no complaints. Radiologically, at the 6and 12-month follow-up (Figure 13), there was no reossification at the cystectomy and
Diagnostics 2022, 12, x
2006
FOR PEER REVIEW
Diagnostics 2022, 12, x FOR PEER REVIEW
of 15
14
109 of
11 of 16
Figure13.
13.CBCT
CBCT X-ray of
Figure
of case
case44atatone
oneyear
yearfollow-up.
follow-up.
Figure 13. CBCT X-ray of case 4 at one year follow-up.
Discussion
3.
3.3.Discussion
Discussion
DC is
is the
the second
second most
cyst
occurring
in association
withwith
an an
DC
common
odontogenic
cyst
occurring
in
DC
is
the
second most
mostcommon
commonodontogenic
odontogenic
cyst
occurring
in association
association
with
an
unerupted tooth.
tooth. It
It develops
from
the
accumulation
of of
fluid
between
the the
reduced
enamel
unerupted
develops
from
the
accumulation
fluid
between
reduced
enamel
unerupted tooth. It develops from the accumulation of fluid between the reduced enamel epepithelium of
of the
the dental
follicle
and
the
ofof
anan
unerupted
tooth
(Figure
14). 14).
The The
epithelium
dental
follicle
and
thecrown
crown
unerupted
tooth
ithelium of the
dental
follicle
and the
crown
of
an unerupted
tooth (Figure
14).(Figure
The microscopic
microscopic features of DC are dependent whether it is not inflamed or inflamed
microscopic
of DCwhether
are dependent
whetheroritinflamed
is not [8,9,17,18].
inflamed or
inflamed
features of DCfeatures
are dependent
it is not inflamed
In the
case of
[8,9,17,18]. In the case of non-inflamed DC, the epithelial lining is formed by two to four
[8,9,17,18].
In DC,
the case
of non-inflamed
DC, theby
epithelial
lining
is formed
by two to four
non-inflamed
the epithelial
lining is formed
two to four
layers
of cuboidal/squamous
layers of cuboidal/squamous non-keratinizing cells without rete ridges and a flat
layers
of cuboidal/squamous
without rete ridges and
a flatinterface
epithenon-keratinizing
cells withoutnon-keratinizing
rete ridges and acells
flat epithelium-connective
tissue
epithelium-connective tissue interface (Figure 3). The cyst wall consists of fibrous or fibrolium-connective
tissue
cyst wall tissue
consists
of fibrous
or fibro-myx(Figure
The cyst
wallinterface
consists
of(Figure
fibrous3).
orThe
fibro-myxoid
containing
considerable
myxoid3).tissue
containing
considerable
glycosaminoglycan
ground
substance.
Small glyoid
tissue
containing
considerable
glycosaminoglycan
ground
substance.
Small
islands
cosaminoglycan
ground
substance.
Small
islands
or
cords
of
inactive-appearing
odontogenic
islands or cords of inactive-appearing odontogenic epithelial rests are usually present or
cords
inactive-appearing
odontogenic
epithelial
rests
are usually present within the
epithelial
rests
are usually
present
within the
connective
tissue.
withinofthe
connective
tissue.
connective tissue.
Figure 14. Dental follicle partly lined by reduced enamel epithelium (H&E, ×20).
Diagnostics 2022, 12, x FOR PEER REVIEW
12 of 16
Diagnostics 2022, 12, 2006
10 of 14
Figure 14. Dental follicle partly lined by reduced enamel epithelium (H&E, ×20).
of inflamed
inflamed DC,
DC, histopathologic
histopathologic examination
examination reveals
reveals aa fibrous-connective
fibrous-connective
In the case of
infiltration of chronic inflammatory
inflammatory cells
cells and,
and, on
on occasion,
occasion,
tissue wall with a variable infiltration
lined
in part
or entirely
by non-keratinizing
squamous
epithecholesterol clefts.
clefts.The
Thecyst
cystis is
lined
in part
or entirely
by non-keratinizing
squamous
lium, which which
shows shows
varyingvarying
amountsamounts
of hyperplasia
with the development
of elongated
epithelium,
of hyperplasia
with the development
of
and
interconnected
rete
ridges.
These
features
may
lead
to
a
misdiagnosis
of
radicular
elongated and interconnected rete ridges. These features may lead to a misdiagnosis of
cyst (Figure
Mucus
or, cells
rarely,
columnar
cells may
bemay
observed
in the
radicular
cyst15).
(Figure
15).cells
Mucus
or,ciliated
rarely, ciliated
columnar
cells
be observed
epithelial
lining.lining.
in
the epithelial
Figure
15. Non-keratinized
Non-keratinizedspongiotic
spongioticsquamous
squamous
epithelium
presenting
hyperplastic
ridges.
Figure 15.
epithelium
presenting
hyperplastic
reterete
ridges.
The
The fibrous wall contains a lymphoplasmocytic infiltrate (H&E, ×10).
fibrous wall contains a lymphoplasmocytic infiltrate (H&E, ×10).
Although
thedefinitive
definitive
diagnosis
of cystic
histopathological
Although the
diagnosis
of cystic
lesionslesions
requiresrequires
histopathological
analysis,
analysis,
clinical
and
radiological
examinations
are
of
paramount
importance the
in
clinical and radiological examinations are of paramount importance in establishing
establishing
the differential
diagnosis
[8,9].theInpresumptive
the case ofpreoperative
DC, the presumptive
differential diagnosis
[8,9]. In the
case of DC,
diagnosis is
preoperative
is usually
based
on [1,9,10].
an analysis
of OPG
images signs
[1,9,10].
usually baseddiagnosis
on an analysis
of OPG
images
Certain
radiological
are Certain
specific,
radiological
signs
are
specific,
while
others
are
non-specific
or
variable.
The
radiological
while others are non-specific or variable. The radiological peculiarity of DCs, which
peculiarity
of DCs,
which facilitates
theirthe
recognition,
the facttothat
the lesions are
facilitates their
recognition,
is the fact that
lesions are is
attached
the cementoenamel
attached
to
the
cementoenamel
junction
and
surround
the
tooth
crown
of
an impacted
junction and surround the tooth crown of an impacted tooth [8–10]. The center
of the
tooth
[8,9,10].
The center
cystthe
may
be located
or below
except
cyst may
be located
aboveof
orthe
below
crown,
except above
when the
tooth isthe
notcrown,
in an upright
when
theor
tooth
is not
anorients
uprightlaterally
positionduring
or when
cyst orients laterally
during its
position
when
the in
cyst
itsthe
development.
Other radiological
development.
Other
signs, less specific
to DC,generally,
are the boundaries
of the
lesion:
signs, less specific
to radiological
DC, are the boundaries
of the lesion:
well-defined,
rounded,
generally,
well-defined,
rounded,
a thin
sclerotic margin
[4,8,11], and
the radiolucent
a thin sclerotic
margin [4,8,11],
and
the radiolucent
homogeneous,
unilocular
internal
homogeneous,
unilocular
internal
appearance
of the
On the other
hand,
appearance of the
lesion [4–9].
On the
other hand,
the lesion
effects[4,5,6,7,8,9].
on the surrounding
anatomical
the
effects (adjacent
on the surrounding
anatomical
(adjacent
teeth, sinus
bone cortex,
IAC,
structures
teeth, bone cortex,
IAC, structures
nasal cavity,
and maxillary
floor) can
be
nasal
cavity,
and maxillary
floor) can
be variable
and include
discharge,
variable
and include
discharge, sinus
displacement,
and expansion
or resorption
[8,9,12].
CBCT
exam is very important
in theordiagnosis
of [8,9,12].
DC. Their
appearance
CBCT
is similar
to
displacement,
and expansion
resorption
CBCT
exam is on
very
important
in the
panoramic of
radiography,
this exam
provides
more precise
information
on the size,
diagnosis
DC. Theirhowever,
appearance
on CBCT
is similar
to panoramic
radiography,
position, and relationship of the lesion to the surrounding structures. In classical helical
computed tomography, the content of a DC typically appears as low density on CBCT
Diagnostics 2022, 12, 2006
11 of 14
(liquid-like). The 3-dimensional analysis of the lesion enabled by CBCT provides precise
and important information for clinicians developing a treatment strategy and surgical
approach. A superior projection of the lesion on the nasal sinus cavities and displacement
of the IAC can be accurately highlighted. Similarly, the expansion of the vestibular or
lingual cortex is easily evaluated. Magnetic resonance imaging (MRI) has a very limited
role in the diagnostics of this pathology, helping to distinguish these lesions from other
cystic bone lesions only when the presentation is atypical. The contents of DC typically
appear liquid-like on MRI (in hypointensity T1 and hyperintensity T2) and lack solids’
high-contrast partitions, except for occasional fine peripheral contrast enhancement when
the cyst is infected.
In the cases presented here, the presumptive radiological diagnosis of DC was confirmed by histopathological examination. However, in the OPG and CBCT images, the
lesions varied in size, contour appearance, relationship, and effects on the surrounding
anatomical structures. Differential radiological diagnoses differed from case to case and are
summarized in Table 1.
Table 1. Summary of the features of the presented cases.
Case No
Impacted
Tooth
DC Size
Contour
Relationship with
Dental Anatomical
Structures
Adjacent teeth:
1
38, horizontal
position with
mesial
orientation
27 × 22 mm
(OPG and CBCT)
Well defined,
thin sclerotcs
border of
-
Tooth 37, distal
bone resorption
No root
resorption
Discharge effect
(distalized
dental axis)
Tooth vital
Adjacent teeth:
2
48, inverted
position with
mesio-caudal
orientation
13 × 15 mm
(OPG)
13 × 17 × 15 mm
(CBCT)
Well defined,
thin sclerotic
border
-
-
Severely
resorbed apex
of the distal
root of tooth 47
Tooth vital
Adjacent teeth:
-
3
38, mesial
24 × 10 mm
(OPT)
24 × 10 × 10 mm
(CBCT)
In places
irregular,
strongly sclerotic
(sign of
superinfection)
-
Suspicion of
resorption of
root apex 37
(OPT)
Strongly
resorbed root
apex 37 (CBCT)
Tooth
37 necrotic
Relationship with
Anatomical Bone
Structures
IAC:
Signs of
interference
Strongly thinned
wall
Caudally
displaced, no
narrowing of the
canal
Cortex:
Bubble-like
vestibular and
lingual cortex
(infra-millimeter
thinning)
IAC:
Signs of
interference
Caudally
displaced
Cranial cortex
discontinuity
Cortex:
Bubble-like
lingual cortex
4 mm alveolar
crest dehiscence
IAC:
Apeces 38
interfering with
IAC
Internal canal
deformation
Cortex:
12-mm alveolar
crest dehiscence
lingually and
5-mm vestibulary
Differential
Diagnosis
-
Dentigerous
cyst
Keratocyst
Unicystic
ameloblastoma
-
Dentigerous
cyst
(root resorption
means keratocyst
was less likely)
-
Infected
dentigerous
cyst
(root resorption
means keratocyst
was less likely)
Diagnostics 2022, 12, 2006
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Table 1. Cont.
Case No
4
Impacted
Tooth
38, distal
DC Size
20 × 15 mm
(OPG)
17 × 21 × 10 mm
(CBCT)
Contour
Well defined,
multilocular,
thin septa,
absence of
periosteal
reaction
Relationship with
Dental Anatomical
Structures
Relationship with
Anatomical Bone
Structures
-
IAC:
Contact of
superior edge
(13 mm)
IAC thinned wall
No canal
deformation
Cortex:
Bubble-like
lingual and
vestibular cortex
Differential
Diagnosis
-
Dentigerous
cyst
-
Ameloblastoma
Keratocyst
Radiologically differential diagnosis of DC is mainly made with hyperplastic dental
follicle, odontogenic keratocyst, and unicystic ameloblastoma. The following characteristics
point toward a DC: neighboring tooth infiltration, bubble-like cortex, and the pericoronal
space thicker than 5 mm [1,4,8,9,16]. An odontogenic keratocyst in pericoronal localization
(about 21% of cases) can be considered in the differential diagnosis, especially in large
lesions, but it causes bubble-like cortex and root resorption less frequently, has a slightly
denser content in radiographic images [19–23], and never really attaches to the cementoenamel junction [8]. A unicystic ameloblastoma or an ameloblastic fibroma cannot be
differentiated radiologically due to the absence of an internal structure [8,21]. A radicular
cyst located at the apex of a deciduous tooth and surrounding the crown of the underlying
permanent tooth can also resemble a cyst [8,24]. An odontogenic adenomatoid tumor or a
calcified odontogenic cyst can also resemble a DC [8,24].
In this case series, the most likely differential diagnoses were keratocyst and unicystic
ameloblastoma. The radiological findings that influenced the differential diagnosis were
mainly the effects of the lesion on the adjacent anatomical structures (root resorption vs.
infiltration) and the multilocular contour (case #4). On the other hand, although the 3D
CBCT X-ray allowed us to appreciate the lesion volume and the relationship between the
lesion and the surrounding structures, the differential diagnosis remained the same as the
one deduced based on the OPT.
For cases #2 and 3, the presumptive diagnosis of DC was quite high because of the
classical appearance and the resorption of the adjacent roots made it possible to exclude
keratocyst. This was in contrast to case #1, where the teeth/roots were displaced. Case
#4, particularity, was the presence of a well-defined multilocular contour and the presence of septa, both of which, however, did not exclude the possibility of a keratocyst
and ameloblastoma.
When the DC is of large size, it can predispose the patient to pathological infections or
fractures [8,9]. Rare cases of ameloblastic transformation in DC have been described in the
literature [21].
The treatment of DC is represented by enucleation followed by curettage. It has been
proposed that natural polymers may favorably help bone regeneration [25].
The use of local antibiotics after the enucleation of the cyst has been advocated [26], as
well as the use of analgesics [27].
4. Conclusions
We have presented four examples of radiological presentation of DCs. Diagnosing
DC lesions involved multiple aspects. Although it is most often based on OPT images
where it appears as a radiolucent lesion surrounding an impacted tooth crown, other
characteristics of the lesion should be taken into account since they may lead to a different
differential diagnosis.
Limitations of this study is the observational nature based on a small number of cases.
Diagnostics 2022, 12, 2006
13 of 14
Author Contributions: Draft of the manuscript, preparation of images, patients treatment, A.P.; draft
of the manuscript, radiological interpretation preparation of images, V.L.; conception, drafting and
critical review of the manuscript, T.L. All authors have read and agreed to the published version of
the manuscript.
Funding: This research received no external funding.
Institutional Review Board Statement: The presented study adhered to the Helsinki Declaration of
ethical principles by the World Medical Association. The study did not require approval of Ethic
Commission on Human Research of Geneva (CCER-Geneva), according to the Federal Human
Research Act (Art.3al.a). Since the study involved less than five patients.
Informed Consent Statement: Written informed consent has been obtained from the patients to
publish this paper.
Data Availability Statement: Not applicable.
Conflicts of Interest: The authors declare no conflict of interest.
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