1. Taiwan J Oral Maxillofac Surg
20: 116-124, Maxillofac
Taiwan J OralJune 2009 Surg 台灣口外誌
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: entigerous cyst, sinusitis, maxilla.
d
Introduction associated with any unerupted teeth, usually
attached to the tooth at the cemento-enamel
Dentigerous cyst (DC) is a common oral junction. But rarely involves unerupted deciduous
lesion formed by fluid accumulation between the teeth1.
fully formed tooth crown and the reduced enamel Radiographically, the DC typically shows a
epithelium. It is considered a developmental unilocular radiolucent shadow with a well-defined
abnormality arising from the reduced enamel sclerotic border associated with the crown of an
epithelium around the crown of an unerupted unerupted tooth, but an infected cyst will show
tooth. The predilection site of DC is the ill-defined borders2. Here, we will describe a case
mandibular third molar. Other frequent sites of DC arising from right maxillary third molar and
include maxillary canines, maxillary third molars, involving the maxillary sinus with sinusitis.
and mandibular second premolar. It is always
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2. 台灣口外誌 Dentigerous Cyst over Maxillary Sinus : A Case Report and Literature Review
Case report closure of the wound was done. The opening was
packed with sterilized petrolatum gauze. Oozy
A 20-year-old young male soldier was discharges were found on the following 2 days.
referred to the Division of Oral and Maxillofacial On the third day, the mucoperiostum was sutured
Surgery, Kaohsiung Armed Force General with 4-0 silk. Postoperative antibiotics were
Hospital with a history of painful sensation and prescribed for 7 days.
swelling over right maxilla. He was also suffered Microscopically, the epithelium was composed
with yellow-green pus discharges from nasal of non-keratinizing stratified squamous epithelium
cavity. On physical examination, pus discharged with plenty chronic inflammatory cells infiltration
from right maxillary second molar distal gingival (Fig 4, 5). There is no evidence of malignant
crevice area was noted. The right maxillary third change or other odontogenic cysts differentiation.
molar cannot be seen. The patient had recurrent A dentigerous cyst was confirmed.
sinusitis for six years with medical treatment but One month latter, the patient was free from
in vain. He had no systemic disease or drug and sinusitis after receiving cyst enucleation. No cyst
food allergy history. Family history did not show recurrence was noted after an 18-months follow
any contribution. up.
The panoramic radiography showed a well-
defined radiolucent lesion with sclerotic margin. Discussion
The lesion pushed the impacted maxillary third
molar upward to the roof of the maxillary sinus. The DC is the second most common
The right maxillary sinus was not clear in the odontogenic cyst, with periapical cyst being
radiography. Root resorption of upper right found more commonly. It presents mostly in the
second molar was also noted (Fig. 1). Computed second or third decade of life in the maxillary
Tomography revealed an expansive mass in or mandibular third molar or maxillary canine
the right maxillary sinus associated with the regions 3 . It can originate from any tooth,
radioactive intensity similar to soft tissue. There including supernumerary tooth4. The DCs are
is no bone destruction (Fig. 2). mostly asymptomatic and may be found on
The patient was arranged to receive opera- routine dental radiographic check-up. They may
tion under general anesthesia. Cyst enucleation also cause symptoms like pain or swelling with the
was done with Caldwell-Luc’s procedure. Full enlargement of the cyst size1. Several researchers
thickness mucoperiosteal flap was reflected at reported the pathologic fracture of the mandible
the right mucobuccal fold from right fist premolar caused by the huge of DC 4,5. The outgrowth
to second molar. A bony window was made to of the cyst may also cause the resorption of
approach the maxillary sinus (Fig. 3). The cyst adjacent tooth. According to Eliasson’s report,
was totally removed and the surrounding bony roughly 1% of impacted maxillary third molars will
structures about 1 to 2 mm thickness were subsequently become involved with a DC6.
also shaved by Stryker. The lesion was about Radiographically, the typical DC showed a
4 × 3 × 2 cm in size with firm in consistency. well-defined radiolucency with sclerotic border
The impacted tooth was also removed. Delayed associated with the crown of an unerupted
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3. Taiwan J Oral Maxillofac Surg 台灣口外誌
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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4. 台灣口外誌 Dentigerous Cyst over Maxillary Sinus : A Case Report and Literature Review
Fig. 3. The lesion was removed.
Fig. 4. HE staining showed non-keratinizing stratified squamous epithelium lining
with plenty of chronic inflammatory cell infiltration (X100).
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5. Taiwan J Oral Maxillofac Surg 台灣口外誌
Fig. 5. Same picture from Fig.4 HE staining showed plenty of inflammatory cells
infiltration with hemorrhage (X200).
tooth 1. Three varieties of the cyst-to-crown panoramic radiology was sufficient for evaluation.
relationships can be seen on radiographic The computed tomography may be useful for
examination. They are central variety, lateral evaluating the extent of bony involvement. Since
variety and circumferential variety1. In the case DCs may contain fluid, in the magnetic resonance
presented here, the cyst-to- crown relationship imaging (MRI), the cyst fluid may be seen as low
was classified to a circumferential variety. The intensity on T1-weighted and high intensity on
expansion of the cyst caused the resorption T2-weighted images8.
of the distal root over upper right second Patients with DCs over maxillary sinus
molar. The cyst growth in this case was quite might present nasal symptoms such as sinusitis.
extensive. The tumor extended from the mesial In addition, ophthalmologic symptoms might
aspect of upper first and second premolar, to be present such as proptosis, diplopia, ptosis,
the retromolar area, and superiorly to the roof epiphora but rarely affected visual acuity.
of the maxillary sinus. This large cyst made Fractured of the orbital bone caused by DC have
patient had symptoms such as pain, swelling and been reported 9. Spontaneous remission of the
sinusitis. In the mandible, the DC may grow up lesion without surgical removal may happen, but
in to the ramus and caused mandible expension7. cases are few10.
In our case, antral obliteration was seen from The differential diagnosis of DC includes
computed tomography. Generally speaking, a odontogenic keratocyst (OKC), adenomatoid
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6. 台灣口外誌 Dentigerous Cyst over Maxillary Sinus : A Case Report and Literature Review
odontogenic tumor (AOT), calcifying epithelial several immunohistochemical markers enhanced,
odontogenic cyst (COC), calcifying epithelial such as Bcl-2, Bcl-xL,FGF, MMPs, Ki-67 and
odontogenic tumor (CEOT), and unicystic PCNA14,15. Recently, calretinin was suggested to
ameloblastoma (UAs). In addition to the histo- be a specific immunohistochemical biomarker for
pathologic differences between the feature of neoplastic ameloblastic epithelium and may serve
the epithelium of OKC and DC, the differential as a diagnostic tool for differentiating cystic
diagnosis can also include the development odontogenic lesions from ameloblastoma16.
and the recurrence tendency of these cysts. The best treatment of the UAs is radical
About 40% unilocular OKC contain impacted though enucleation is sufficient in subtype 1
tooth. The OKC is more aggressive with higher and 217,18,21-23. Considering the similarity of UAs
recurrence risk than DC and may be associated and DC clinically, we enucleated the lesion and
with nevoid basal cell carcinoma syndrome. also trimmed the surrounding bony structures
Recently, researches showed mutation of PTCH to about 1 to 2 mm in thickness. Hopefully, the
gene and overactivated of Shh signaling may be histopathological report confirmed the diagnosis
associated with the clinicopathological expression of DC.
of OKCs11. BMP-4 may be a useful biochemical The DC may cause head and neck infection
marker to differentiation of OKC and DC. BMP-4 with a prevalence of 2.1%24. The cyst can undergo
is expressed more intensive in OKC compared carcinomatous transformation into ameloblastoma
with DC, and is more intensively expressed or squamous cell carcinoma but is rare25-28. In
in the recurred cases 12. The AOT and COC cases where mucous cells are present in the
generally are more frequently seen in maxillary epithelium, the intraosseous mucoepidermoid
anterior area with some degree of calcification carcinoma may be ruled out1. Enucleation of the
within the cyst cavity, which may be observed cyst contents with extraction of the associated
from radiography 7,13. Histopathologically, the tooth is sufficient for DC. For extremely large
COC may present keratinized epithelial cell lesions, or in cases when the involved tooth is
so-called ghost cells in the cavity. The AOT desired to keep in the arch, marsupialization may
different from the DC, its predilection in female be done. With decompression, the involved tooth
with epithelial cells syncytially arranged in may erupt spontaneously by orthodontically into
rosettes or duct-like structure. The CEOT may occlusion. When other odontogenic tumors are
be differentiated from DC by its honeycomb highly suspected, radical removal of the lesions
pattern radiolucency with foci of radiopacity or removal of the cyst with surrounding bony
in radiographs. Microscopically, it shows large structures is suggested.
polygonal epithelial cells with variation in size and
shape with amyloid materials, which contained References
concentric calcified deposits (Liesegan rings)13.
Although the unicystic ameloblastoma is rare, 1. Neville BW, Damm DD, Allen CM, Bouquot
its clinical expression and microscopic feature JE. Oral Maxillofacial Pathology. 2nd ed.
can be deceptive and may be confused with DC. Philadelphia: WB Saunders. 2002: 590-3.
The UAs is more locally aggressive than DC with 2. Wood NK, Goaz PW. Differential Diagnosis of
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7. Taiwan J Oral Maxillofac Surg 台灣口外誌
Oral Lesions. 5th ed. St. Louis Mosby 1997: the odontogenic keratocyst. J Oral Pathol
283-8. Med 2005; 34: 178-83.
3. Regezi JA, Sciubba JJ, Jodan R.C.K. Oral
13. R egezi JA, Sciubba JJ, Jodan RCK. Oral
pathology, clinical pathologic correlations. pathology, clinical pathologic correlations.
4th ed. St. Louis: WB Saunders. 2003: 246- 4th ed. St Louis: WB Saunders. 2003: 274-
88. 76.
4. Som PM, Shangold LM, Biller HF. A palatal
14. Kumamoto H, Ooya L. Immunohistochemical
dentigerous cyst arising from a mesiodente. analysis of Bcl-2 family proteins in benign and
Am H Neuroradiol 1992; 13: 212-4. malignant ameloblastomas. J Oral Pathol Med
5. M aroo SV. Clinico-radiological aspects of
1999; 28: 343-9.
dentigerous cysts. East Afr Med J 1991; 68: 15. Li TJ, Brown RM, Matthews JB. Expression
249-54. of proliferating cell nuclear antigen (PCNA)
6. E liasson S, Heimdahl A, Nordenram A.
and Ki-67 in unicystic ameloblastoma.
Pathological changes related to long- Histopathol 1955; 26: 219-28.
term impaction of third molars. Int J Oral 16. C oleman H, Altini M, Ali H, Doflioni C,
Maxillofac Surg 1989: 210-25. Favia G, Maiorano E. Use of calretinin
7. oaz PW, White SC. Oral radiology, principles
G in the differential diagnosis of unicystic
and interpretation. 2nd ed. St. Louis: Mosby. ameloblastomas. Histopathol 2001; 38: 312-
1987: 486-9, 426-692. 7.
8. SOM PM, Shugar JMA, Cohen BA, Biller HF.
17. Gardner D. Plexiform unicystic ameloblas-
The nonspecificity of the antral bowing sign toma: a diagnostic problem in dentigerous
in maxillary sinus pathology. J Comput Assist cyst. Cancer 1981; 47: 1358-63.
Tomogr 1981; 5: 350-2. 18. A ckermann GL, Altini M, Shear M. The
9. O mer K, Onder B. A misdiagnosed giant
unicystic ameloblastoma: a clinicopathological
dentigerous cyst involving the maxillary study of 57 cases. J Oral Pathol 1988; 17:
antrum and affecting the orbit. Case report. 541-6.
Aust Dent J 1994; 39: 165-7. 19. Philipsen HP, reichart PA. Unicystic amelo-
10. Freedman GL. A disappearing dentigerous
blastoma. A review of 193 cases from the
cyst:Report of a case. J Oral Maxillofac Surg literature. Oral Oncol 1998; 34: 317-25.
1988; 46: 885-6. 21. Shear M. Cysts of the Oral Regions, 3rd ed.
11. Koyama E, Yamaai T, Isseki S, Ohuchi H,
Oxfort: Wright. 1992; 75-98
Nohno T, Yoshiola H, Hayash Y, Leatherman 22. Robinson L, Martinez MG. Unicystic amelo-
JL, Golden EBm Noji S, Pacifici M. Polarizing blastoma: a prognostically distinct entity.
activity, Sonic hedgehog, and tooth develop- Cancer 1977; 40: 2278-85.
ment in embryonic and postnatal mouse. Dev 23. L i TJ, Wu YT, Yu SF, Yu GY. Unicystic
Dyn 1996; 206: 59-72. ameloblastoma: a clinicopathologic study of
12. Kim SG, Yang BE, Oh SH, Min SK, Hong SP,
33 Chinese patients. Am J Surg Pathol 2000;
Choi JY. The differential expression pattern 24: 1385-92.
of BMP-4 between the dentigerous cyst and 24. S mith JL, Kellman RM. Dentigerous cysts
- 122 -

8. 台灣口外誌 Dentigerous Cyst over Maxillary Sinus : A Case Report and Literature Review
presenting as head and neck infections. 1982; 27: 365-7.
Otolaryngol Head Neck Surg 2005; 133: 715- 27. Rafael M: Ameloblastoma developing from a
20. dentigerous cyst. Oral Surg Oral Med Oral
25. Kramer IRH, Pindborg JJ, Shear M. Histo-
Pathol 1960; 13: 781-6.
logical Typing of Odontogenic Tumors. 28. M cMillan MD, Millie AC. Ameloblastomas
Berlin: Springer 1992: 11-4. associated with dentigerous cysts. J Oral
26.
Yaacob HB, Ling KC. Ameloblastomatous Pathol Med 1981; 51: 489-96.
changes in dentigerous cyst. Aust Dent J
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9. Taiwan J Oral Maxillofac Surg 台灣口外誌
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。
Received: March 28, 2009
Accepted: May 31, 2009
Reprint requests to: r. Wen-Bin Kao, Division of Oral and Maxillofacial Surgery, Department of
D
Dentistry, Kaohsiung Armed Forces General Hospital.
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