Case Report ISSN 2639-9490

Research Article Oral Health & Dental Science

Rare Case of Huge Bi-Locular Dentigerous Cyst Involving Mandibular

Canine
Maher Al-assaf, DDS, MSc1*, Ali Al-awad, BDS2, Ahmad Al Manadili, DDS, MSc, PhD1, Sami Ibrahim, BDS,
MSc, PhD2 and Moutaz Al-Khen, BDS, MSc, PhD2

*
Correspondence:
Department of Oral Histology and Pathology, Faculty of Dentistry,
1 Maher Al-assaf, DDS, MSc, Department of Oral Histology and
Damascus University, Damascus, Syria. Pathology, Faculty of Dentistry, Damascus University, Damascus,
Syria, almazzeh/601, Damascus, Syria, Tel: +963991880364.
2
Department of Oral and Maxillofacial Surgery, General Assembly ORCID ID: 0000-0001-6691-6992.
of Damascus Hospital, Damascus, Syria.
Received: 27 December 2020; Accepted: 15 January 2021

Citation: Maher Al-assaf, Ali Al-awad, Ahmad Al Manadili, et al. Rare Case of Huge Bi-Locular Dentigerous Cyst Involving Mandibular
Canine. Oral Health Dental Sci. 2021; 5(1); 1-4.

ABSTRACT
Dentigerous cyst is benign odontogenic cystic lesion that envelops the crown of an impacted tooth. Like odontogenic
tumors, dentigerous cysts may grow to large sizes before they are diagnosed, and the large radiolucent cystic
lesions associated with an impacted tooth are often diagnosed as odontogenic tumors like unicystic ameloblastoma
or others developmental cystic lesions like odontogenic keratocyst, but less likely to be dentigerous cysts. In this
case report we highlight the first case of huge bi-locular dentigerous cyst in a 35-year-old woman related with
impacted mandibular canine.

Keywords The radiographs show a unilocular radiolucent cystic lesion

Dentigerous cyst, Impacted tooth, Odontogenic cysts, Odontogenic covering the crown of an impacted tooth (central location), and
tumors. on the side of the tooth (lateral location), or completely around
the tooth (circumferential location). The radiolucency area has a
Introduction well-defined and often sclerotic border [1]. Large DCs can have
Dentigerous cyst (DC) or follicular cyst is benign odontogenic a multilocular appearance on radiograph, given the existence
cystic lesion that envelops the crown of an impacted tooth formed of bone trabeculae within the radiolucency. However, they are
by extension of its follicle because of aggregation of fluid between histologically a unilocular lesion [7].
Nasmyth's membrane and the surface of enamel, and the expansion
of this cyst is related to an increase in cyst fluid osmolality and the Herein, we reported a rare case of huge bi-locular DC involving
release of bone resorption factors [1-3]. This eventually leads to permanent mandibular left canine in a female patient. After verifying
the formation of the cyst in which the crown is located within the all the anterior mandibular cases in the literature, we found that this is
lumen and roots outside [4]. the first case of bi-locular DC in anterior mandible.

DCs are more than twice as common in males as females, and Case Report
two-thirds develop on lower third molars and most of these A 35-year-old woman consulted the department of oral and maxillofacial
cysts occur during the second and third decades [1]. Generally, surgery because of a mild swelling at the anterior mandible.
DCs are asymptomatic lesions and are observed during routine
radiographic investigations [5,6]. Huge DCs may cause expansion History of present illness
of the cortical bone and resorption of adjoining teeth, but such The patient complained of a 2-month history of progressive
cases still unfamiliar [7]. nonpainful swelling of her anterior mandible. The patient denied

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any history of pain, fever, purulence or trismus, and she did not On these clinical and radiographic characteristics, differential
report any neurosensory changes. diagnosis included dentigerous cyst, odontogenic Keratocyst and
unicystic ameloblastoma.
Clinical examination Surgical procedure
The patient had slight lower facial swelling; the mass was hard, After the nasal and endotracheal intubation, injection was done
non-fluctuant and non-tender to palpation. There were no facial or in the working area by lidocaine with vasoconstrictor above the
trigeminal nerve deficits. Neck: there were no palpable mass and periosteum in the lower anterior vestibular region. Then a flap was
no cervical or submandibular lymphadenopathy. designed on the teeth adjacent to the cystic lesion (33, 32, 31, 41,
42, and 44) with two release incisions, after that a full thickness
mucoperiosteal flap was elevated and the both mental foramina
Intraoral with mental nerves were isolated. After that, the whole cystic
Occlusion was stable and there was no movement in teeth, lesion was enucleated and the impacted canine was extracted.
the interincisal opening was within normal limits. There was Carnoy's solution was used until reaching a healthy bone. Finally,
buccal expansion of the anterior mandible extending from right Gel foam was applied then the flap was repositioned. VICRYL
mandibular first premolar toward the left mandibular first premolar. thread (4-0) was used for sutured (Figures 3, A-B-C-D).
All teeth were vital according to electrical test. Blood supply and Histologic examination: the microscopic revealed that hyperplastic
innervation have been maintained in remaining bone tissue. (about 8 to 20 cuboidal cells in thickness) non-keratinized stratified
squamous odontogenic epithelium surrounding the lumen of this
Radiographic examination (OPG and CBCT) cystic lesion. The connective tissue showed interweaving fibers of
There was large bi-locular radiolucent lesion with sclerotic well- collagen, vascularity and fibroblasts. Severe cholesterol clefts and
defined borders and associated with impacted mandibular left lymphocytes infiltration were also seen in the connective tissue,
revealing of inflamed DC (Figures 4, A-B-C).
canine. This large lesion extended from left mandibular premolars
to right ones, and there were clear signs of destruction of buccal Radiological signs of good bone formation after 6 months including
bone lamina (Figures 1 and 2). mixed radiolucent - radiopaque area in anterior mandible (Figure 5).

Figure 1: Orthopantomogram (OPG) showing the large bi-locular radiolucent lesion associated with impacted mandibular left canine.

Figure 2: CBCT finding: three sagittal sections of cone-beam computed tomography (CBCT) show: (A) the buccal cortical defect on the first loculus
(arrow). (B) The bony septa between two loculi which was lingually from impacted canine (solid arrowhead). (C) Shows the second loculus (open
arrowhead).

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 This study was legally authorized by the Ethics Committee at
General Assembly of Damascus Hospital (Approval ref: 200164),
and the signed informed consent for publication was obtained
from the patient.

Discussion
DC (sometimes called follicular cyst) is a developmental
odontogenic cyst that envelops the crown of impacted tooth, which
may be part of the regular dentition or a supernumerary tooth [5].
This cystic lesion arises from the epithelium of the dental follicle
and remains attached to the neck of the tooth, enclosing the crown
within the cavity of cyst [1,5].

DC may be observed as an incidental radiographic finding

or by investigating a clinical expansion, and the potential
neoplastic transformation include unicystic ameloblastoma, and
Figure 3: (A) Intra-oral photograph showing the lesion border with evident
carcinomatous transformation include intraosseous squamous
bone destruction. (B) Intra-oral photograph showing the healthy bone after
removal of whole lesion. (C) Post-operative photograph showing the complete cell carcinoma which may derived from changes of the epithelial
enucleation. (D) Post-operative photograph showing the wound suture. cells of DC [8-10]. Therefore, follow-up is essential to avoid
complications and to get better treatment according to the
diagnosis of each different case. Hence, early and correct diagnosis
will increase the percentage of success [11].

In addition, it has been suggested that the potential for development

of the rare intraosseous mucoepidermoid carcinoma (MEC) may
arise from the presence of mucous cells in the DC epithelial lining
[12-14].

Some DCs may appear as a multilocular appearance on

radiographs because of protruding bony septa from the walls, but
this appearance should raise suspicion for odontogenic neoplasms
or non-odontogenic lesions [15].

Conclusions
Huge DCs are unfamiliar, and such benign lesions related
with impacted tooth may be odontogenic tumors. Therefore,
Figure 4: Histological photomicrographs showing: (A) The fibrous it is important that the final diagnosis decision be based on
connective tissue lined with odontogenic epithelium (H&E, 40×). (B) The histopathological examination, rather than radiographical finding
hyperplastic odontogenic epithelium: 20 layers of cuboidal cells (H&E, only.
40×). (C) The severe cholesterol clefts (H&E, 10×).
Hence, early diagnosis and proper treatment planning for such
uncommon cases is necessary to avoid further complication, and a
long-term follow up is essential in these cases.

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© 2021 Al-assaf M, et al. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License

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