dentistry journal

Case Report
Unusual Imaging Features of Dentigerous Cyst:
A Case Report
Carla Patrícia Martinelli-Kläy 1,2, *, Celso Ricardo Martinelli 2 , Celso Martinelli 2 ,
Henrique Roberto Macedo 2 and Tommaso Lombardi 1
1 Laboratory of Oral & Maxillofacial Pathology, Oral Medicine and Oral and Maxillofacial Pathology Unit,
Division of Oral Maxillofacial Surgery, Department of Surgery, Geneva University Hospitals, University of
Geneva, 1211 Geneva, Switzerland
2 Centre for Diagnosis and Treatment of Oral Diseases, Ribeirão Preto 14025-250, Brazil
* Correspondence: CarlaPatricia.Martinelli-Klay@hcuge.ch; Tel.: +41-22-379-4034




Received: 26 March 2019; Accepted: 5 July 2019; Published: 1 August 2019


Abstract: Dentigerous cysts (DC) are cystic lesions radiographically represented by a well-defined
unilocular radiolucent area involving an impacted tooth crown. We present an unusual radiographic
feature of dentigerous cyst related to the impacted mandibular right second molar, in a 16-year-old
patient, which suggested an ameloblastoma or odontogenic keratocyst (OKC) because of its
multilocular appearance seen on the panoramic radiography. A multi-slice computed tomography
(MSCT), however, revealed a unilocular lesion without septations, with an attenuation coefficient
from 3.9 to 22.9 HU suggesting a cystic lesion. Due to its extension, a marsupialization was performed
together with the histopathological analysis of the fragment removed which suggested a dentigerous
cyst. Nine months later, the lesion was reduced in size and then totally excised. The impacted
mandibular right second molar was also extracted. Histopathological examination confirmed the
diagnosis of a dentigerous cyst. One year later, the panoramic radiography showed a complete
mandible bone healing. Large dentigerous cysts can sometimes suggest other more aggressive
pathologies. Precise diagnosis is important to avoid mistakes since DC, OKC and ameloblastoma
require different treatments. Histological examination is, therefore, essential to establish a definitive
diagnosis. In our case, MSCT and the tissue attenuation coefficient analysis contributed to guide the
diagnosis and management of the dentigerous cyst.

Keywords: dentigerous cyst; radiographic differential diagnosis; multislice computed tomography;

hounsfield unit analysis

1. Introduction
A dentigerous cysts (DC) are defined as cystic lesions involving the crown of impacted teeth
caused by fluid accumulation between the follicular epithelium and the crown of the tooth. It is
considered the most common type of noninflammatory odontogenic cyst, and it occurs mainly in
the lower third molar tooth of male patients with peak incidences in adolescents or young adults.
Dentigerous cysts present a slow painless swelling and can cause the teeth displacement or teeth and
bone resorption. Large cysts, however, may be associated with pain [1–4]. Histologically, they are
represented by a cavity lined by the non-keratinizing thin epithelium without rete pegs. Their wall is
usually fibrous and devoid of inflammatory cells [1].
Radiographically, DCs often present a unilocular radiolucent area around the crown of the
impacted tooth surrounded by a well-defined sclerotic area [1,2,4,5]. Large cysts may cause cortical
bone expansion. Moreover, DCs can rarely show a multilocular feature in the panoramic radiography.
This is probably due to the cyst growth in areas of different bone densities [2]. In this case, the

Dent. J. 2019, 7, 76; doi:10.3390/dj7030076 www.mdpi.com/journal/dentistry

Dent. J. 2019, 7, 76 2 of 7

differential diagnosis
Dent. J. 2019, 7, x FOR PEERshould
REVIEWbe made with other more aggressive lesions, such as ameloblastomas, 2 of 7
odontogenic keratocysts (OKCs), other odontogenic tumours [1–3].
case, the differential diagnosis should be made with other more aggressive lesions, such as
Computed tomography (CT), such as Cone Beam CT (CBCT) or multi-slice computed tomography
ameloblastomas, odontogenic keratocysts (OKCs), other odontogenic tumours [1–3].
(MSCT), has an important application in the evaluation of head and neck lesions. It is a non-invasive
Computed tomography (CT), such as Cone Beam CT (CBCT) or multi-slice computed
technique that permits the lesion size and margins, the bone destruction and expansion patterns to be
tomography (MSCT), has an important application in the evaluation of head and neck lesions. It is a
precisely analysed [6,7]. In addition, MSCT provides an accurate measurement of the tissue attenuation
non-invasive technique that permits the lesion size and margins, the bone destruction and expansion
coefficient
patterns [7–10]. Each tissue
to be precisely has[6,7].
analysed the ability to absorb
In addition, MSCTa provides
certain proportion
an accurate of X-rays as the
measurement bone,
of the
fortissue
example, which absorbs
attenuation a lot
coefficient of X-rays
[7–10]. whilehas
Each tissue the the
air ability
almosttonone.
absorbAna arbitrary scale named
certain proportion of X-the
Hounsfield scale shows these attenuation coefficients: −1000 Hounsfield unit (HU)
rays as the bone, for example, which absorbs a lot of X-rays while the air almost none. An arbitrary represents the air
attenuation, 0 is the water attenuation and +1000 HU is the bone attenuation [7,8].
scale named the Hounsfield scale shows these attenuation coefficients: −1000 Hounsfield unit (HU)
This paper
represents theillustrates a case0of
air attenuation, is an
theunusual dentigerous
water attenuation andcyst initially
+1000 HU is diagnosed as ameloblastoma
the bone attenuation [7,8].
due to its
Thismultiloculated
paper illustrates feature
a caseobserved in panoramic
of an unusual dentigerous radiography.
cyst initially diagnosed as ameloblastoma
due to its multiloculated feature observed in panoramic radiography.
2. Case Presentation
2. Case Presentation
A 16-year-old male was referred to the Centre for Diagnosis and Treatment of Oral Diseases with
a clinicalA diagnosis
16-year-oldofmale was referred having
ameloblastoma to the Centre for Diagnosis and as
hemi-mandibulectomy Treatment of Oraltherapy.
the suggested Diseases At
with a clinical diagnosis of ameloblastoma having hemi-mandibulectomy as the
admission, no relevant aspects within his medical history were observed. He brought a panoramicsuggested therapy.
At admission, no relevant aspects within his medical history were observed. He brought
radiography (Figure 1) which revealed the presence of a multilocular lesion involving the impacted a panoramic
radiography
mandibular (Figure
right second1) molar
which suggesting
revealed themainly
presence
an of a multilocular or
ameloblastoma lesion involving
an OKC. the impacted
It extended from the
mandibular right second molar suggesting mainly an ameloblastoma or an OKC.
right mandibular notch to the lower right first premolar. A displacement of the mandibular canal It extended fromand
the right mandibular notch to the lower right first premolar. A displacement of the mandibular canal
the lower right third molar towards the mandibular notch was also observed. We had access to another
and the lower right third molar towards the mandibular notch was also observed. We had access to
radiography taken at the age of thirteen, which presented a well-defined sclerotic area around the
another radiography taken at the age of thirteen, which presented a well-defined sclerotic area
crown of the mandibular right second molar suggesting a hyperplastic dental follicle or a dentigerous
around the crown of the mandibular right second molar suggesting a hyperplastic dental follicle or
cyst (Figure 2). During those three years, the patient did not consult. Intraoral examination revealed a
a dentigerous cyst (Figure 2). During those three years, the patient did not consult. Intraoral
slight painless swelling
examination revealed aofslight
the buccal and
painless lingual
swelling ofcortical extending
the buccal from
and lingual the lower
cortical rightfrom
extending first molar
the
region to the mandibular branch (not shown).
lower right first molar region to the mandibular branch (not shown).

Figure
Figure 1. Panoramic
1. Panoramic radiography:
radiography: A well-defined
A well-defined multiloculated
multiloculated radiolucent
radiolucent lesion involving lesion
impacted
involvingright
mandibular impacted
secondmandibular
molar. right second molar.
Dent. J. 2019, 7, 76 3 of 7
Dent. J. 2019, 7, x FOR PEER REVIEW 3 of 7

Dent. J. 2019, 7, x FOR PEER REVIEW 3 of 7

Figure
Figure 2. Panoramic
2. Panoramic radiography
radiography mademade at theat agetheof age of thirteen:
thirteen: A unilocular
A unilocular well-defined
well-defined radiolucent
radiolucent lesion (measuring around 4 mm)
lesion (measuring around 4 mm) surrounding impacted mandibular right second molar.surrounding impacted mandibular right
Figure 2. Panoramic radiography made at the age of thirteen: A unilocular well-defined
second molar.
radiolucent lesion (measuring around 4 mm) surrounding impacted mandibular right
With the aim of further analysis of the lesion, an MSCT (Figure 3) was carried out, and unlike the
second molar.
panoramic Withradiography,
the aim of further it showed analysis an of areatheoflesion, an MSCT (Figure
hypoattenuation without3) was carried out,
septations. The and unlike
Hounsfield
unitthe
(HU) panoramic
value radiography, it showed 3.9an to area of hypoattenuation without septations. The
With theinaimtheoflesion
further varied
analysis fromof the 22.9anHU,
lesion, MSCT which suggested
(Figure a lesion
3) was carried out,containing
and unlikeliquid
Hounsfield
that could unit
be compatible
the panoramic (HU) value in the
with a dentigerous
radiography, lesion
it showed an varied
cystarea from
or even 3.9 to 22.9 HU, which
a unicystic ameloblastoma.
of hypoattenuation suggested a
From this
without septations. lesion
Thetissue
containing
Hounsfield
attenuation liquid
unitthat
coefficient (HU) could
value
analysis, be acompatible
inlesion
the lesion
such withas aan
varied dentigerous
from 3.9 tocyst
ameloblastoma 22.9or(solid/multicystic
evenwhich
HU, a unicystic ameloblastoma.
suggestedtype),a lesion
or a cystic
From this tissue attenuation
that couldcoefficient analysis, aadentigerous
lesion suchcyst as an
or ameloblastoma (solid/multicystic
lesioncontaining
containing liquid
a cheese-like besubstance
compatible with
as found in odontogenic even a unicystic
keratocyst ameloblastoma.
could be disregarded.
type),
From or a
thiscystic
tissue lesion containing
attenuation a
coefficient cheese-like
analysis, substance
a lesion suchas found
as an in odontogenic
ameloblastoma keratocyst could
(solid/multicystic
Because of the lesion extension, marsupialization was performed together with the histopathological
betype),
disregarded.
or a cystic Because of the lesion
lesion containing extension,substance
a cheese-like marsupialization
as found was performedkeratocyst
in odontogenic together with couldthe
analysis of the fragment removed, which suggested a dentigerous cyst. During this procedure, a clear
histopathological
be disregarded. analysis Because of of the
the lesion
fragment removed,
extension, which suggested
marsupialization was aperformed
dentigerous cyst. During
together with thethis
andprocedure,
pale fluid aflowed
histopathological
out pale
clear analysis
and of the
offluid
cavity. Theremoved,
flowed
the fragment
lesion
out of was
the irrigated
cavity.
which Theweekly
suggested lesion with chlorhexidine
was irrigated
a dentigerous weekly
cyst. During
gluconate
with
this
0.12% oral solution.
procedure, agluconate
chlorhexidine During
clear and 0.12% treatment,
pale fluid a candida
flowed out
oral solution. (Candida
of thetreatment,
During sp.)
cavity. The infection
lesion was
a candida of the contents
irrigated
(Candida coming
weekly with
sp.) infection from
of the
thecontents
cavity (not
chlorhexidine shown)
cominggluconate was
from the0.12% detected
cavity oral(not through
solution.
shown) cytological
During
was treatment, examination and
a candida (Candida
detected through promptly
cytological treated
sp.) examination
infection of the andwith
itraconazole
promptly capsules
contentstreated
comingwith (1×itraconazole
from daily for 10
the cavity days).
(not shown)
capsules After
(1× was nine
daily formonths,
detected a After
through
10 days). considerable
cytological
nine months,decrease
examination of the
andlesion
a considerable
promptly
wasdecrease
observed treated
of onthepanoramic
lesionwithwasitraconazole
observedcapsules
radiography on(not (1× dailyradiography
shown).
panoramic for 10 days).
Excision After
of (not
the nine lesion,
cystic
shown). months, asa considerable
Excision wellof as
theextraction
cystic
of the decrease
impacted of the lesion
mandibular was observed
right second on panoramic
molar, was radiography
then (not
performed.
lesion, as well as extraction of the impacted mandibular right second molar, was then performed. shown). Excision
Histopathological of the cystic
examination
lesion,the
confirmed as well
Histopathological asexamination
diagnosis extraction of confirmed
of a dentigerous the impactedcystthe mandibular
(Figure
diagnosis4). Oneright
of second
ayear later,molar,
dentigerous a new was
cyst then performed.
panoramic
(Figure 4). radiography
One year
Histopathological examination confirmed the diagnosis of a dentigerous cyst (Figure 4).bone
One healing
year
waslater, a new
carried outpanoramic
showing aradiography
complete mandible was carried bone outhealing
showing a complete
(Figure 5). The mandible
mandibular right third
later, a new panoramic radiography was carried out showing a complete mandible bone healing
(Figure
molar was 5).onlyThe mandibular
removed afterright third molar
its migration to was only removed
the retromolar afterinits
region migration
order to cause to the
the retromolar
least surgical
(Figure 5). The mandibular right third molar was only removed after its migration to the retromolar
region
trauma in
(Figureorder 5).to cause the least surgical trauma (Figure 5).
region in order to cause the least surgical trauma (Figure 5).

Figure 3. Sagital multi-slice computed tomography (MSCT) image: A large uniloculated lesion
involving impacted mandibular right second molar. The lesion attenuation coefficient varies from 3.9
to 22.9 HU.
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Dent. J.J. 2019,
2019, 7,
7, xx FOR
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REVIEW 44 of
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Figure 3. Sagital multi-slice computed tomography (MSCT) image: A large uniloculated lesion
involving
Dent. J. 2019, 7, 76 impacted mandibular right second molar. The lesion attenuation coefficient 4 of 7
varies from 3.9 to 22.9 HU.

Figure
Figure 4. Photomicrograph:
4. Photomicrograph: A non-keratinizing
A non-keratinizing epithelial
epithelial lining without
lining without rete pegsrete
andpegs and wall
a fibrous a
fibrous
with wall with rare
rare inflammatory inflammatory
cells. HES, ×20. cells. HES, ×20.

Figure
Figure5.5.Panoramic
Panoramicradiography
radiographyone
one year
year later: A complete
later: A completemandible
mandiblebone
bonehealing.
healing.

3. Discussion and
3. Discussion Conclusions
and Conclusions
Although
Although dentigerous
dentigerouscysts cysts often appearasasa asimple
often appear simple radiolucent
radiolucent areaarea surrounding
surrounding the crown
the crown of
an impacted
of an impacted tooth,
tooth, large
large cysts
cystsmaymayshowshowaamultilocular
multilocularfeature
feature andandsuggest
suggestother
otherlesions such
lesions as as
such
ameloblastoma
ameloblastoma and and
OKCOKC [2,3].
[2,3]. Therefore,a histological
Therefore, a histologicalexamination
examinationisisessential
essentialininorder
ordertotoprecise
precisethe
the diagnosis [1,11]. Dentigerous cysts present a cavity lined by a non-keratinized
diagnosis [1,11]. Dentigerous cysts present a cavity lined by a non-keratinized stratified epithelium stratified
epithelium
containing containing
between two between
and three two and three
layers layers of
of cuboidal cuboidal
and/or and/orcells.
flattened flattened
Thecells. The connective
connective tissue wall
is usually fibrous and often devoid of inflammatory cells. Odontogenic keratocysts show show
tissue wall is usually fibrous and often devoid of inflammatory cells. Odontogenic keratocysts a cavity
a cavity lined by a thin and regular parakeratinized stratified squamous epithelium without rate
lined by a thin and regular parakeratinized stratified squamous epithelium without rate pegs. The
pegs. The basal layer cells are cuboidal or columnar and are often hyperchromatic. The lumen of these
basal layer cells are cuboidal or columnar and are often hyperchromatic. The lumen of these cysts
cysts contains cheese-like material representing aggregated keratin scales. Similar to the dentigerous
contains cheese-like material representing aggregated keratin scales. Similar to the dentigerous cyst,
cyst, the interface between OKC’s epithelium and connective tissue is flat. Unicystic ameloblastomas
the interface between OKC’s epithelium and connective tissue is flat. Unicystic ameloblastomas are a
are a variant of the ameloblastoma which appear as cysts. The epithelium lining these cysts, however,
variant of the ameloblastoma
is composed of ameloblastic cellswhich appearpalisading
showing as cysts. and
Thereverse
epithelium lining
nuclear theseThe
polarity. cysts, however, is
suprabasilar
composed of ameloblastic
areas often display a loosen cells showing
stellate palisading
reticulum and reverse
appearance. The same nuclear polarity.
histological The suprabasilar
features are found
areas often display a loosen stellate reticulum
in the solid variant of ameloblastomas [1,2]. appearance. The same histological features are found in
the solid variant of ameloblastomas [1,2].
Unlike dentigerous cysts, ameloblastomas and odontogenic keratocysts have an aggressive
behaviour and require different forms of treatment. Ameloblastomas are benign odontogenic tumours
that usually require a wide surgical resection [2,12] whereas dentigerous cysts are usually treated with
enucleation and curettage. Odontogenic keratocysts are benign cysts which have a relatively high rate
Dent. J. 2019, 7, 76 5 of 7

of recurrence. They are preferably treated with surgical enucleation, marsupialization, decompression
or marginal resection [13,14].
We presented a case of dentigerous cyst initially diagnosed as ameloblastoma having
hemi-mandibulectomy as a suggested therapy. The ameloblastoma diagnosis was given by taking only
the panoramic radiography and clinical aspect into account. The multilocular feature observed in the
panoramic radiography is probably due to an uneven expansion of a large dentigerous cyst in areas of
different bone densities [2]. It is known that panoramic radiography is a useful aid for diagnoses, but
it only shows two-dimensional images of three-dimensional structures. In addition, it has a limited
value to determine the lesion size and margins, tissue composition, as well as bone destruction and
expansion patterns [15]. In our study, MSCT was carried out and, unlike the panoramic radiography, it
showed an area of hypoattenuation without septations (Figure 3). We also found a variation of the
tissue attenuation coefficient from 3.9 to 22.9 HU, which suggested a lesion containing serous fluid
compatible with the content of a dentigerous cyst [16]. According to the literature, ameloblastomas
(solid/multicystic type) have 35.9 plus/minus 12.6 HU and the OKCs show 28.4 plus/minus 10.5 [17] or
up to 40 HU [14]. Unicystic ameloblastomas present a Hounsfield Unit of 31.0 plus/minus 6.0 HU [17].
It is known that MSCT provides accurate measurement of the tissue attenuation coefficient [7–10]. In
addition, the incisional biopsy done during the marsupialization confirmed the diagnosis of dentigerous
cyst [1,11].
Because of the lesion extension [11], the marsupialization was the initial treatment of choice.
Decompression and marsupialization are both conservative procedures aimed at reducing the cyst size
without damaging anatomical structures such as alveolar nerve, or even causing mandibular fracture
during the surgery. In addition, these techniques allow a gradual apposition of bone tissue before the
complete cyst enucleation [18–21].
Through cytological examination, Candida sp. infection coming from the cavity was observed and
promptly treated with itraconazole (1× daily for 10 days). The cytological smear is used in clinical
practice [22] and may be useful during the marsupialization treatment.
After a considerable decrease nine months later, the lesion was excised, and the impacted
mandibular right second molar was extracted. According to Anavi et al. [19], the average of
dentigerous cyst decompression is 7.6 months in patients under 18 years of age. In children, only the
marsupialization, decompression and/or enucleation of the dentigerous cyst have been suggested in an
attempt to preserve the tooth and promote its eruption [23–25]. In our case, preserving the tooth was
not considered possible, mainly due to the size of the cystic lesion. Histological analysis confirmed the
diagnosis of a dentigerous cyst which presented a wall of uninflamed fibrous connective tissue lined
by a thin non-keratinized stratified epithelium.
In case of large or radiographically unusual cystic lesions, it is mandatory to perform a biopsy before
planning surgical treatment. Depending on lesion size and diagnosis, an enucleation, marsupialization
or decompression could be the forms of treatment chosen. In our case, the initial incisional biopsy
made during the marsupialization process provided the diagnosis of a dentigerous cyst allowing a
conservative approach. In addition, the tissue attenuation coefficient analysis done by MSCT was also
useful in the diagnosis process to eliminate the possibility of other lesions such as a solid ameloblastoma
or an odontogenic keratocyst.

Author Contributions: C.P.M.-K. and C.M.: cytological and histopathological diagnoses; T.L., C.R.M. and
C.P.M.-K.: conception and drafting of the manuscript; C.R.M. and H.R.M.: patient care and interpretation of the
MSCT-scan; C.M., T.L., C.P.M.-K., C.R.M. and H.R.M.: critical revision of the manuscript.
Funding: Not applicable.
Conflicts of Interest: The authors declare that there is no conflict of interest regarding the publication of this paper.
Ethics Statement: This case report is for academic communication only. It was carried out in accordance with The
Code of Ethics of the World Medical Association (Declaration of Helsinki). Written consent was obtained from a
family member and every precaution was taken to protect the privacy of the patient and the confidentiality of
their personal information.
Dent. J. 2019, 7, 76 6 of 7

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