8. Orthokeratinized Odontogenic Cyst: A Case Report S

Archives of Oral Sciences and Research
Orthokeratinized Odontogenic Cyst: A Case Report
S Hemavathy*, Sahana NS*, D Vinay Kumar*, Suresh T*, Chandrakala J*, Mohammed
Asif*.
ABSTRACT
Orthokeratinized odontogenic cyst (OOC) is a relatively uncommon developmental cyst
comprising about 10% of cases that had been previously coded as odontogenic keratocysts.
Odontogenic keratocyst was designated as keratocystic odontogenic tumor (KCOT) in the
new World Health Organization classification and OOC should be distinguished from KCOT
for differences in histologic features and biologic behavior. OOC occurs more commonly in
mandibular posterior region and in majority of cases it is observed in third and fourth decade
with male predilection. Here we are presenting a case report of OOC in 48 year old patient
in posterior mandibular region.
AOSR 2013;3(2):144-150
Key Words: Orthokeratinised odontogenic cyst; keratocystic odontogenic tumor.
*Department of Oral Pathology, Government Dental College and Research Institute,
Bangalore.
INTRODUCTION
Orthokeratinized odontogenic cyst (OOC)
of odontogenic Keratocyst. Wright (1981)
is a developmental cyst that occurs in the
specified its clinico pathological aspects
maxilla and the mandible, was initially
and
defined by the World Health Organization
odontogenic keratocyst - Orthokeratinized
(1992) as the uncommon orthokeratinized
variant.2
type of odontogenic keratocyst (OKC).
suggested
that
it
to
be
called
Li et al (1998) suggested a term
OOC was first described by Schultz (1927).
‘‘Orthokeratinized Odontogenic cyst”. The
Philipsen (1946) considered it to be a type
World
144
Health
Organization
new
Keratocystic odontogenic tumor
classification (2005) for head and neck
tumors
has
designated
OKC
as
Keratocystic Odontogenic Tumor (KCOT)
and reclassified it as a neoplasm in view of
its intrinsic growth potential and propensity
to
recur.
According
to
this
new
Fig 1:Extraoral view showing mild
swelling in left posterior region of
mandible
classification, OOC should not be part of
the spectrum of KCOT and should be
distinguished from the latter.2,3,4,7
defined radiolucency with sclerotic border
CASE REPORT
which extends from lower right first molar
A 48 year old male patient presented to our
to
college with a chief complaint of pain and
mandible.(Fig2)
the
coronoid
process
of
the
swelling in left posterior region of the jaw
since one month. His past medical history
was of no medical relevance and general
physical status was good. Extra oral
examination revealed swelling in left lower
one third of the face which extends from
Fig 2: OPG showing well defined
radiolucency in lower left posterior tooth
region extending to the ramus of mandible
angle of the mouth to the tragus of ear
which
is
localised,
firm
and
no
tender.(Fig1) On intra oral examination
there was slight obliteration of buccal
After clinical and radiographic evaluation a
vestibule with minimal cortical expansion
provisional
in the region of 36 to 38. OPG reveals well
keratocyst was made. Incisional biopsy of
145
diagnosis
of
Odontogenic
Vinay Kumar et al
the
lesion
was
for
The cyst was surgically enucleated along
On
with the removal of 36, 37 and 38 under
microscopic examination it showed a cystic
local anaesthesia and chemically cauterised
wall lined by odontogenic epithelium
with carnoys solution. Gross examination
which was around 8 to 10 layers. Surface of
of the excised specimen revealed a thin
the epithelium exhibits orthokeratinization
cystic sac with a smooth luminal surface.
with numerous keratin flakes in the lumen,
The lumen also contained white cheesy
prominent granular layer and the basal
material.(Fig4)
layer shows cuboidal cells which lacks
examination it showed the similar features
palisading arrangement and underlying
of incisional biopsy and final diagnosis of
connective tissue consists of radially
OOC was given.
histopathological
done
and
sent
examination.
On
microscopic
arranged collagen fibers, endothelial lined
capillaries with mild infiltration of chronic
inflammatory cells. Microscopic features
suggestive of orthokeratinised odontogenic
cyst. (Fig 3)
Fig 4: Photomicrograph of OOC showing
keratin flakes filling the lumen with
prominent granular layer.
DISCUSSION
The orthokeratinized odontogenic cyst
identified as an orthokeratinzed variant of
Fig 3: Excisional biopsy measuring around
4.5x3x1.5 cm
the odontogenic keratocyst for the first time
146
Keratocystic odontogenic tumor
by Wright in 1981 because of it’s different
as
biological behaviour and histopathology.6
parakeratinized OKC. Recurrence rate in
Crowley separated odontogenic keratocyst
parakeratinised OKCs is at least 42.6% of
(OKC) into three histologic categories:
the cases, compared with only 2.2% for
parakeratinized,
a
orthokeratinized OKCs.(Table 1) But due
combination of the two types and founded
to the less aggressive clinical behavior and
that 86.2% of the 449 cases were
recurrence pattern of the orthokeratinized
parakeratinized,
variant
orthokeratinized,
12.2%
or
were
compared
with
47.8%
ultimately
for
warranted
the
the
orthokeratinized, and 1.6% had features of
designation of the orthokeratinized variant
both orthokeratin and parakeratin. The
as a separate entity, "Orthokeratinized
orthokeratinized OKC was more often
Odontogenic Cyst".1
associated with an impacted tooth (75.7%),
Table 1: Comparison between Odontogenic keratocyst and Orthokeratinized
odontogenic cyst
PARAKERATINISED OKC
OOC
(OR) KCOT
RECURRENCE RATE
42.6%
2.2%
ASSOCIATION WITH IMPACTED 47.8%
75.7%
TOOTH
SYNDROME RELATION
Associated With Nevoid Basal Cell No Association
Syndrome.
HISTOPATHOLOGY
1. Basal cells are columnar showing 1. Basal cells are cuboidal and shows
palisading arrangement.
little
tendency
to
polarize
and
2. Parakeratin layer shows surface palisading.
corrugation.
Granular
prominent.
layer
not 2. Shows orthokeratinisation with
numerous
keratin flakes in the
lumen. Prominent granular layer.
147
Vinay Kumar et al
Both the entities show similar findings
and is composed of either columnar or
clinically regarding age, sex and site of
cuboidal cells that are arranged in a
occurrence but the OOCs are generally
palisaded pattern and the luminal surface,
solitary asymptomatic lesions whereas
often corrugated, is typically covered with
KCOT associated with Nevoid Basal Cell
parakeratin.7
Carcinoma Syndrome (NBCCS) exhibits
Enucleation with curettage is the usual
multiple
more
treatment for orthokeratinized odontogenic
commonly in the mandible with an affinity
cysts. Recurrence has rarely been noted,
for the posterior portion features coinciding
and the reported frequency is around 2%,
with the case presented in our study.2,6
which is in marked contrast with the 30%
lesions.
Radiographically
OOCs
be
or higher recurrence rate associated with
unilocular lesions and are more often
odontogenic keratocysts. KCOTs are treated
associated with impacted teeth as compared
similarly to other odontogenic cysts, that is,
to KCOTs and in our case the radiolucency
by enucleation and curettage. Due to higher
was unilocular in posterior region without
recurrence rate simple cyst enucleation
any impacted teeth.3
without curettage was no longer advocated.
Histologically the OOC is characterized by
Peripheral ostectomy was recommended as
a thin, uniform epithelial lining, 4-8 cell
an adjunct approach to enucleation when
layers
of
resections could be avoided but there still
squamous
remains some uncertainty over the success
epithelium with a prominent granular cell
rate. Enbloc osseous resection may have to
layer.2 The basal cells are usually cuboidal
be considered for some cases. This may be
or flattened and show little tendency to
a
polarize or palisade. Whereas in KCOT the
continuity, or segmental resection that
thick
orthokeratinized
OOCs
occur
and
tend
to
composed
stratified
basal layer of the epithelium is well defined
148
marginal
resection
that
preserves
Keratocystic odontogenic tumor
violates
continuity
and
will
require
lesions look similar but with different
additional reconstruction.8
biological behavior and there is more
tendency of clinical misdiagnosis which in
CONCLUSION
turn could affect the prognosis of the
Thus this review shows the importance in
patient.
diagnosis of OOC and KCOT as both the
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odontogenic tumour: systematic review.
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CORRESPONDENCE
Dr Vinay Kumar
Postgraduate student
Department of Oral Pathology
GDCRI, Bangalore.
E-mail- [email protected]
Ph No.-9035002480
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