BoTryoiD oDoNTogeNiC CyST: rePorT of A CASe

BoTryoiD oDoNTogeNiC CyST: rePorT of A CASe

CLINICAL DENTISTRY AND RESEARCH 2015; 39(1): 42-47
Case Report
Botryoid Odontogenic Cyst: Report Of A Case
Naile Cura DDS, PhD
Private Practice
ABSTRACT
İzmir, Turkey
Botyroid odontogenic cyst (BOC) is considered a rare multilocular
Serkan Dadakoğlu DDS, PhD
premolar-canine area, followed by the anterior region of the maxilla.
Private Practice
İzmir, Turkey
variant of the lateral periodontal cyst, usually involves the mandibular
Adults older than 50 years are the most affected group. A 57 yearold male patient referred to our clinic, Ankara University Faculty
Timur Songür, DDS, PhD
of Dentistry Department of Oral and Maxillofacial Surgery for
Department of Oral and Maxillofacial Surgery,
evaluation of a swelling in the right anterior mandible. The diagnosis
Faculty of Dentistry, Turgut Özal University,
of a BOC was made based on location and the histopathological
Ankara, Turkey
findings of multiple cystic spaces lined by nonkeratinized stratified
squamous epithelium. The 9-month follow-up revealed a normal
clinical appearance with evidence of radiographic bone fill at the site
of the lesion. BOC is known to be a recurrent odontogenic cyst. The
recurrence rate may range between 15% and 20%. The prevailing
opinion is that main reason for recurrence was failure to remove the
entire multilocular lesion during surgery. An extended post-surgical
follow-up is necessary for a patient who has been diagnosed with
BOC.
Keywords: Botryoid Odontogenic Cyst, Lateral Periodontal
Correspondence
Naile Cura, DDS, PhD
İzmir CTG Dental Care, Kahramanlar ,İzmir
Telephone: +90 5546325616
E-mail: nailecura@gmail.com
42
Cyst, Radiolucency.
Submitted for Publication: 12.10.2013
Accepted for Publication : 01.22.2015
CLINICAL DENTISTRY AND RESEARCH 2015; 39(1): 42-47
Olgu Bildirimi
BOTRYOİD ODONTOJENİK KİST: VAKA RAPORU
Naile Cura
ÖZ
Dr., Ağız, Diş ve Çene Cerrahisi Uzmanı
Botyroid odontojenik kist lateral periodontal kistin nadir görülen
Serbest Diş Hekimi
multilokuler bir varyantıdır, genellikle mandibular premolar-kanin
İzmir, Turkiye
dişler bölgesinde ve maksilla anterior bölgede görülür. Lezyon
Serkan Dadakoğlu
genellikle 50 yaş sonrası erişkinlerde görülür. Bu olgu sunumunda
Dr., Ağız,Diş ve Çene Cerrahisi Uzmanı
57 yaşında erkek hasta, sağ anterior mandibular bölgede şişlik
Serbest Diş Hekimi
Ankara, Türkiye
Timur Songür
Dr., Turgut Özal Üniversitesi Diş Hekimliği Fakültesi
Ağız, Diş ve Çene Cerrahisi Anabilim Dalı
Ankara, Türkiye
şikayeti ile Ankara Üniversitesi Diş Hekimliği Fakültesi Ağız,
Diş ve Çene Cerrahisi Anabilim Dalı’na başvurmuştur. Botryoid
odontojenik kist tanısı lokalizasyona ve nonkeratinize çok
katlı yassı epitelle çevrilmiş multiple kistik alanlar şeklindeki
histopatolojik bulgulara dayanarak konulmuştur. 9 aylık post
operatif takip sonrası kist çıkarılan bölgede radyolojik incelemede
gözlenen kemik oluşumu ile birlikte normal klinik görünüm
izlenmiştir. Botryoid odontojenik kist rekürrens oranı %15 ile
%20 arasında değişen nüks ihtimali yüksek bir odontojenik kisttir.
Rekürrensin ana sebebi multilokuler lezyonun geniş cerrahi
eksizyonunun yapılmaması olarak düşünülmektedir. Botryoid
odontojenik kist tanısı konulan hastalarda uzun sureli post
operatif takip önerilmektedir.
Sorumlu Yazar
Naile Cura,
Anahtar Kelimeler:
Botryoid Odontojenik Kist, Lateral
Periodontal Kist, Radyolusensi
Özel İzmir CTG Ağız ve Diş Sağlığı Merkezi ,1416.Sok.
No:34 35230 Kahramanlar, İzmir
Phone: +90 5546325616
E-mail: nailecura@gmail.com
Yayın Başvuru Tarihi : 10.12.2013
Yayına Kabul Tarihi : 22.01.2015
43
CLINICAL DENTISTRY AND RESEARCH
INTRODUCTION
A multilocular variant of the lateral periodontal cyst (LPC)
known as the botryoid odontogenic cyst (BOC) has been
identified in 1973 by Weathers and Waldron.1 It has been
suggested the term botryoid odontogenic cyst as an
appropriate descriptive name because of the grapelike
configuration of the gross appearance of these cysts and
their odontogenic origin.1
The most current theory of origin suggests that LPCs and
BOCs arise from the dental lamina, in part because of the
presence of glycogen-rich clear cells in the cyst wall that are
similar to the cells found in dental lamina rests.2
Histologically, both lesions appear the same. The cystic
Figure1. Bucco-lingual width of the lesion
lining is composed of one to five cell layers of squamous
or cuboidal epithelium with a fibrous connective tissue
according to inflammation.3,4 The epithelium shows areas
of focal thickening with glycogen containing clear cells
observed around the cyst lining.5-8 The BOC and LPC often
have a subepithelial zone of hyalinization.6,9
The odontogenic epithelium responsible for the cystic lining
in the BOC and LPC is hypothesized to arise from three
possible sources: (1) a reduced enamel epithelium (REE), (2)
epithelial cell rests of Malassez (ERMs), or (3) residuals of
dental lamina.4
Figure 2. CT shows unilocular cystic lesion about 15 mm in diameter
in the right mandibular region.
CASE REPORT
A 57 year-old male patient referred to our clinic, Ankara
University Faculty of Dentistry Department of Oral and
Maxillofacial Surgery for evaluation of a swelling seen in
the right anterior mandible. His systemic diseases include:
diabetes mellitus type 2 and hypertension. He had been
aware of the asymptomatic swelling during the past 8
months. At the time of dental examination, an expansion
behind the right mandibular canine was observed, panoramic
radiography and CT revealed a well-defined unilocular
radiolucent lesion distal to the right mandibular canine in
the edentulous region (Figures 1, 2 and 3). The canine tooth
was devital. There was no paresthesia, tenderness or other
changes in sensation. An excisional biopsy was performed
Figure 3. Operative view of the intra-oral approach.
that included the canine tooth under local anesthesia
(Figures 4, 5 and 6).
The pathology report was indicated as a diagnosis of BOC.
Multiple cystic spaces lined by nonkeratinized stratified
squamous epithelium were seen histopathologically (Figure
7). The cyst lining was thickest layer of the three cell layers
with areas of plaque-like thickening in the epithelium.
44
The cyst wall was composed of fibrous tissue exhibiting
extravasated blood cells without inflammation.
At the 9-month follow-up, the clinical appearance of the
surgical site was normal (Figures 8, 9 and 10). The patient is
using total prosthesis and there is no complaining about the
RADIOLUCENCY IN MANDIBULA
Figure 4. Enucleation and tooth extraction
Figure 6. Post-operative intra-oral photograph.
Figure 5. Macroscopic appearance of the surgical specimen.
right mandibular region.
DISCUSSION
BOC usually involves the mandibular premolar-canine area,
followed by the anterior region of the maxilla.10 The most
affected group is adults older than 50 years.11 Although
some of the reported cases of BOC have shown a multilocular
radiolucency, this image is not characteristic for BOC. There
are similarities between BOC and some odontogenic tumors
such as ameloblastoma, odontogenic myxoma in terms of
appearance. Preoperative differential diagnosis can be
carried out by means of incisional biopsy. Some authors
demonstrated that this lesion frequently presents as a
unilocular radiographic image, thus resembling a variety of
other odontogenic cysts or neoplastic conditions.12
Figure 7. In the right side of the picture stratified squamoid plaques
were shown.
The enucleation of a BOC or LPC results in an osseous cavity
or defect. Lehrhaupt et al.4 treated a residual LPC defect
that perforated the facial and lingual mandibular cortex in a
similar characterization. A demineralized freeze-dried bone
allograft had been used and a membrane had not been
placed over the bone graft material. Nart et al.5 reported
45
CLINICAL DENTISTRY AND RESEARCH
Figure 8. Post-operative 1-month panoramic radiography
Figure 9. Post-operative 9-month panoramic radiography
Figure 10.Post-operative 9-month intra-oral photograph
There is no comment about the state of vital and nonvital
adjacent teeth to the lesion in the literature. When a
radiolucent lesion is seen related with teeth that is identified
on a radiographic examination , histologic examination must
be performed if it has a diagnosis about normal, vital pulps.
BOC is known as a recurrent odontogenic cyst. The
recurrence rate may range between 15% and 20%.13 The
histopathological appearance of BOC is quite typical in
the presence of epithelial proliferations (plaques). The
proliferation rate of the epithelial lining can be determined
to estimate the possibility of recurrence. The recurrence risk
of BOC is similar to odontogenic keratocyst.
The characteristic that distinguishes BOC from LPC is the
larger size of the BOC because LPC has a limited growth
potential.14 The importance of the differentiation between
LPC and BOC is due to the histologic multilocular aspect of
the BOC that induces this lesion more expansive. Thereby,
increasing the possibility of recurrence because its complete
surgical removal is more difficult.9,14
BOC shows some histopathological similarities to the
glandular odontogenic cyst (GOC) or sialo-odontogenic
cyst (SOC) depending on the presence of mucous cells and
surface columnar cells.10,15,16 However, the best classification
for these lesions is BOC. These lesions can be seen at
opposite ends of a spectrum. The presence of mucous
cells does not suspend the BOC from an odontogenic
origin, this characteristic of BOC has been reported in a
variety of odontogenic cysts such as the dentigerous
cyst with a metaplastic phenomenon.17 Furthermore,
immunohistochemical studies18,19 have suggested that the
GOC is a histological variant of BOC.
According to a prevalent opinion, main reason for recurrence
can be a failure during the complete removal of the
multilocular lesion during surgery. A long term post-surgical
follow-up is necessary for a patient whit a diagnosis of
BOC.3,13 Alternative treatment techniques for BOC can be
considered such as odontogenic keratocyst.11
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a similar treatment of a residual LPC osseous defect using
guided tissue regeneration with a demineralized freezedried bone allograft and resorbable collagen membrane. At
the 7-month follow-up, they found significant radiographic
bone fill, which was also seen on surgical reentry. Because
of the recurrence risk of BOC, wide excision is required for
treatment. In the current case, we treated the patient by
enucleation.
46
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RADIOLUCENCY IN MANDIBULA
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