Dentigerous Cyst Over Maxillary Sinus : A Case Report and

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Taiwan J Oral Maxillofac Surg 台灣口外誌

Introduction

Dentigerous cyst (DC) is a common oral

lesion formed by fluid accumulation between the

fully formed tooth crown and the reduced enamel

epithelium. It is considered a developmental

abnormality arising from the reduced enamel

epithelium around the crown of an unerupted

tooth. The predilection site of DC is the

mandibular third molar. Other frequent sites

include maxillary canines, maxillary third molars,

and mandibular second premolar. It is always

associated with any unerupted teeth, usually

attached to the tooth at the cemento-enamel

junction. But rarely involves unerupted deciduous

teeth1.

Radiographically, the DC typically shows a

unilocular radiolucent shadow with a well-defined

sclerotic border associated with the crown of an

unerupted tooth, but an infected cyst will show

ill-defined borders2. Here, we will describe a case

of DC arising from right maxillary third molar and

involving the maxillary sinus with sinusitis.

Dentigerous Cyst Over Maxillary Sinus : A Case Report and Literature Review

Chih-Jen Wang*, Po-Hsien Huang, Yin-Lai Wang,

Yih-Chung Shyng, Wen-Bin Kao#

* Division of Oral and Maxillofacial Surgery, Department of Dentistry, Kaohsiung Armed

Forces General Hospital, Kaohsiung, Taiwan# School of Dentistry, National Defense Medical Center

Abstract

Dentigerous cyst (DC) is a common odontogenic cyst developed abnormally around unerupted maxillary or mandibular teeth. It is often asymptomatic and can be found incidentally on dental radiography with delayed eruption of teeth. However, it can be large and cause symptoms related to expansion and impingement on contiguous structures. Pain and swelling may be the major complains of patients. However, DC seldom caused head and neck inflammation or infection. Here, we described a 20-year-old male, who was found of a DC arising from right maxillary third molar involved the maxillary sinus and with sinusitis. We also reviewed articles to discuss the differential diagnosis of DC from other odontogenic cysts or odontogenic tumors.

Key words: dentigerous cyst, sinusitis, maxilla.

Taiwan J Oral Maxillofac Surg20: 116-124, June 2009 台灣口外誌

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台灣口外誌 Dentigerous Cyst over Maxillary Sinus : A Case Report and Literature Review

Case report

A 20-year-old young male soldier was

referred to the Division of Oral and Maxillofacial

Surgery, Kaohsiung Armed Force General

Hospital with a history of painful sensation and

swelling over right maxilla. He was also suffered

with yellow-green pus discharges from nasal

cavity. On physical examination, pus discharged

from right maxillary second molar distal gingival

crevice area was noted. The right maxillary third

molar cannot be seen. The patient had recurrent

sinusitis for six years with medical treatment but

in vain. He had no systemic disease or drug and

food allergy history. Family history did not show

any contribution.

The panoramic radiography showed a well-

defined radiolucent lesion with sclerotic margin.

The lesion pushed the impacted maxillary third

molar upward to the roof of the maxillary sinus.

The right maxillary sinus was not clear in the

radiography. Root resorption of upper right

second molar was also noted (Fig. 1). Computed

Tomography revealed an expansive mass in

the right maxillary sinus associated with the

radioactive intensity similar to soft tissue. There

is no bone destruction (Fig. 2).

The patient was arranged to receive opera-

tion under general anesthesia. Cyst enucleation

was done with Caldwell-Luc’s procedure. Full

thickness mucoperiosteal flap was reflected at

the right mucobuccal fold from right fist premolar

to second molar. A bony window was made to

approach the maxillary sinus (Fig. 3). The cyst

was totally removed and the surrounding bony

structures about 1 to 2 mm thickness were

also shaved by Stryker. The lesion was about

4 × 3 × 2 cm in size with firm in consistency.

The impacted tooth was also removed. Delayed

closure of the wound was done. The opening was

packed with sterilized petrolatum gauze. Oozy

discharges were found on the following 2 days.

On the third day, the mucoperiostum was sutured

with 4-0 silk. Postoperative antibiotics were

prescribed for 7 days.

Microscopically, the epithelium was composed

of non-keratinizing stratified squamous epithelium

with plenty chronic inflammatory cells infiltration

(Fig 4, 5). There is no evidence of malignant

change or other odontogenic cysts differentiation.

A dentigerous cyst was confirmed.

One month latter, the patient was free from

sinusitis after receiving cyst enucleation. No cyst

recurrence was noted after an 18-months follow

up.

Discussion

The DC is the second most common

odontogenic cyst, with periapical cyst being

found more commonly. It presents mostly in the

second or third decade of life in the maxillary

or mandibular third molar or maxillary canine

regions3. It can originate from any tooth,

including supernumerary tooth4. The DCs are

mostly asymptomatic and may be found on

routine dental radiographic check-up. They may

also cause symptoms like pain or swelling with the

enlargement of the cyst size1. Several researchers

reported the pathologic fracture of the mandible

caused by the huge of DC4,5. The outgrowth

of the cyst may also cause the resorption of

adjacent tooth. According to Eliasson’s report,

roughly 1% of impacted maxillary third molars will

subsequently become involved with a DC6.

Radiographically, the typical DC showed a

well-defined radiolucency with sclerotic border

associated with the crown of an unerupted

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Fig. 1. Unclear sinus image with distal root resorption of upper right 2nd molar were seen. The impacted

tooth was pushed to the roof of maxillary sinus by the cyst. The cyst extended from upper right

premolar to retromolar area and maxillary sinus roof.

Fig. 2. A cystic lesion associated with an impacted teeth was seen at right maxillary sinus.

There was no bony destruction at sinus wall.

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Fig. 3. The lesion was removed.

Fig. 4. H&E staining showed non-keratinizing stratified squamous epithelium lining

with plenty of chronic inflammatory cell infiltration (X100).

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Fig. 5. Same picture from Fig.4 H&E staining showed plenty of inflammatory cells

infiltration with hemorrhage (X200).

tooth1. Three varieties of the cyst-to-crown

relationships can be seen on radiographic

examination. They are central variety, lateral

variety and circumferential variety1. In the case

presented here, the cyst-to- crown relationship

was classified to a circumferential variety. The

expansion of the cyst caused the resorption

of the distal root over upper right second

molar. The cyst growth in this case was quite

extensive. The tumor extended from the mesial

aspect of upper first and second premolar, to

the retromolar area, and superiorly to the roof

of the maxillary sinus. This large cyst made

patient had symptoms such as pain, swelling and

sinusitis. In the mandible, the DC may grow up

in to the ramus and caused mandible expension7.

In our case, antral obliteration was seen from

computed tomography. Generally speaking, a

panoramic radiology was sufficient for evaluation.

The computed tomography may be useful for

evaluating the extent of bony involvement. Since

DCs may contain fluid, in the magnetic resonance

imaging (MRI), the cyst fluid may be seen as low

intensity on T1-weighted and high intensity on

T2-weighted images8.

Patients with DCs over maxillary sinus

might present nasal symptoms such as sinusitis.

In addition, ophthalmologic symptoms might

be present such as proptosis, diplopia, ptosis,

epiphora but rarely affected visual acuity.

Fractured of the orbital bone caused by DC have

been reported9. Spontaneous remission of the

lesion without surgical removal may happen, but

cases are few10.

The differential diagnosis of DC includes

odontogenic keratocyst (OKC), adenomatoid

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odontogenic tumor (AOT), calcifying epithelial

odontogenic cyst (COC), calcifying epithelial

odontogenic tumor (CEOT), and unicystic

ameloblastoma (UAs). In addition to the histo-

pathologic differences between the feature of

the epithelium of OKC and DC, the differential

diagnosis can also include the development

and the recurrence tendency of these cysts.

About 40% unilocular OKC contain impacted

tooth. The OKC is more aggressive with higher

recurrence risk than DC and may be associated

with nevoid basal cell carcinoma syndrome.

Recently, researches showed mutation of PTCH

gene and overactivated of Shh signaling may be

associated with the clinicopathological expression

of OKCs11. BMP-4 may be a useful biochemical

marker to differentiation of OKC and DC. BMP-4

is expressed more intensive in OKC compared

with DC, and is more intensively expressed

in the recurred cases12. The AOT and COC

generally are more frequently seen in maxillary

anterior area with some degree of calcification

within the cyst cavity, which may be observed

from radiography7,13. Histopathologically, the

COC may present keratinized epithelial cell

so-called ghost cells in the cavity. The AOT

different from the DC, its predilection in female

with epithelial cells syncytially arranged in

rosettes or duct-like structure. The CEOT may

be differentiated from DC by its honeycomb

pattern radiolucency with foci of radiopacity

in radiographs. Microscopically, it shows large

polygonal epithelial cells with variation in size and

shape with amyloid materials, which contained

concentric calcified deposits (Liesegan rings)13.

Although the unicystic ameloblastoma is rare,

its clinical expression and microscopic feature

can be deceptive and may be confused with DC.

The UAs is more locally aggressive than DC with

several immunohistochemical markers enhanced,

such as Bcl-2, Bcl-xL,FGF, MMPs, Ki-67 and

PCNA14,15. Recently, calretinin was suggested to

be a specific immunohistochemical biomarker for

neoplastic ameloblastic epithelium and may serve

as a diagnostic tool for differentiating cystic

odontogenic lesions from ameloblastoma16.

The best treatment of the UAs is radical

though enucleation is sufficient in subtype 1

and 217,18,21-23. Considering the similarity of UAs

and DC clinically, we enucleated the lesion and

also trimmed the surrounding bony structures

to about 1 to 2 mm in thickness. Hopefully, the

histopathological report confirmed the diagnosis

of DC.

The DC may cause head and neck infection

with a prevalence of 2.1%24. The cyst can undergo

carcinomatous transformation into ameloblastoma

or squamous cell carcinoma but is rare25-28. In

cases where mucous cells are present in the

epithelium, the intraosseous mucoepidermoid

carcinoma may be ruled out1. Enucleation of the

cyst contents with extraction of the associated

tooth is sufficient for DC. For extremely large

lesions, or in cases when the involved tooth is

desired to keep in the arch, marsupialization may

be done. With decompression, the involved tooth

may erupt spontaneously by orthodontically into

occlusion. When other odontogenic tumors are

highly suspected, radical removal of the lesions

or removal of the cyst with surrounding bony

structures is suggested.

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2. Wood NK, Goaz PW. Differential Diagnosis of

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4. Som PM, Shangold LM, Biller HF. A palatal

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5. Maroo SV. Clinico-radiological aspects of

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Received: March 28, 2009Accepted: May 31, 2009Reprint requests to: Dr. Wen-Bin Kao, Division of Oral and Maxillofacial Surgery, Department of

Dentistry, Kaohsiung Armed Forces General Hospital.

上顎竇之含牙囊腫：病例報告與文獻回顧

王峙仁*　黃寶賢　王銀來　邢憶春　高文斌#

* 國軍高雄總醫院牙科部口腔顎面外科

# 國防醫學院牙醫學系

摘　　要

含牙囊腫是一種常見於上下顎阻生齒周圍發育異常的齒源性囊腫。由於

通常並無症狀，故患者常因為牙齒延遲萌發，於接受放射檢查時而發現。然

而，此囊腫若持續擴大，侵犯擠壓到鄰近組織，便可能產生不適。腫大與疼

痛是大多數病人的主要抱怨的症狀，但含牙囊腫鮮少引起頭頸部發炎或感

染。本文提出一位20歲男性因右上顎第三大臼齒侵犯上顎竇導致鼻竇炎的個

案報告，同時藉由回顧文獻，討論對於含牙囊腫與其他齒源性囊腫的鑑別診

斷。

關鍵詞：含牙囊腫，鼻竇炎，上顎。

Documents

Dentigerous Cyst Over Maxillary Sinus : A Case Report and