|Year : 2015 | Volume
| Issue : 2 | Page : 70-75
Cone beam computed tomography findings of ectopic tooth in the maxillary sinus associated with dentigerous cyst: A report of two cases and review of the literature
Elif Sogur1, Meltem Özden2, Tayfun Günbay2, Zuhal Tugsel1
1 Department of Dentomaxillofacial Radiology, School of Dentistry, Ege University, Izmir, Turkey
2 Department of Dentomaxillofacial Surgery, School of Dentistry, Ege University, Izmir, Turkey
|Date of Web Publication||22-May-2015|
Dr. Meltem Özden
Ege University, Dishekimligi Fakultesi, Ağız, Diş ve Çene Cerrahisi AD, Bornova - 35100, Izmir
Source of Support: None, Conflict of Interest: None
Dentigerous cysts are the most common type of benign odontogenic lesions that associated with the crown of an unerupted or impacted tooth. They constitute the second most common cystic lesion of the jaws, after radicular cysts. Dentigerous cyst surrounding impacted teeth often displace these teeth into ectopic positions. Ectopic eruption of teeth into regions other than the oral cavity is common but dentigerous cysts associated with ectopic teeth within the maxillary sinus are fairly rare. The diagnosis is usually made by panoramic radiography. Cone beam computed tomography (CBCT) is indicated when an ectopic tooth is associated with an antral mass and prior to surgery. The standard treatment for a dentigerous cyst is enucleation and extraction of the cyst-associated impacted or unerupted tooth. We report CBCT findings of two cases of dentigerous cyst associated with an ectopic tooth in the maxillary sinus and review the literature reports of this condition over the past 34 years.
Keywords: Cone beam computed tomography, dentigerous cyst, ectopic tooth
|How to cite this article:|
Sogur E, Özden M, Günbay T, Tugsel Z. Cone beam computed tomography findings of ectopic tooth in the maxillary sinus associated with dentigerous cyst: A report of two cases and review of the literature. J Oral Maxillofac Radiol 2015;3:70-5
|How to cite this URL:|
Sogur E, Özden M, Günbay T, Tugsel Z. Cone beam computed tomography findings of ectopic tooth in the maxillary sinus associated with dentigerous cyst: A report of two cases and review of the literature. J Oral Maxillofac Radiol [serial online] 2015 [cited 2019 Dec 7];3:70-5. Available from: http://www.joomr.org/text.asp?2015/3/2/70/157529
| Introduction|| |
Tooth eruption is a process whereby the forming tooth migrates from its intraosseous location in the jaw to its functional position within the oral cavity. Ectopic eruption is a disturbance in which the tooth does not follow its usual course.  Pathologies such as cyst or tumor in relation to a developing tooth is one of the three main causes of ectopic eruption of the tooth, the others being-developmental disturbance, and iatrogenic activity. 
Ectopic eruption of the tooth into regions other than the oral cavity is rare, although there have been reports of tooth in the nasal septum,  mandibular condyle,  coronoid process.  One rather unusual location is the maxillary sinus.  The etiology of ectopic teeth in the maxillary antrum is not yet entirely clear. In literature, dentigerous cysts are mostly associated with the crowns of unerupted tooth and may displace the teeth into ectopic positions such as the maxillary sinus. ,
Dentigerous cyst is a type of odontogenic cyst formed by the separation of the follicle from the crown of an unerupted tooth, more common in males, occurring in the second or third decade of life. About 70% of dentigerous cysts occur in the mandible and 30% in the maxilla. , Although the mandibular third molar and maxillary canine are involved most frequently,  maxillary third molars are the most commonly encountered ectopic teeth associated with dentigerous cyst within the maxillary sinus. 
Clinical presentation of dentigerous cyst varies widely. Most patients are asymptomatic, whereas some present with chronic or recurrent sinusitis, nasolacrimal duct obstruction, and purulent rhinorrhea. , The diagnosis is usually made by plain-film radiography. However, specialized imaging modalities like computed tomography (CT) and cone beam CT (CBCT) are of great importance in the management of these cysts involving the maxillary sinus to determine the extension and features of the lesion prior to surgery. 
Enucleation via a Caldwell-Luc procedure under local or general anesthesia has been the standard treatment for dentigerous cysts, along with extraction of the associated tooth. In large cysts, an initial marsupialization to diminish the size of the osseous defect, followed by enucleation and tooth extraction, has been advocated. Endoscopic approach for management of dentigerous cyst of the maxilla is also described in the literature. 
A search of Medline from 1980 until now, using the key words "dentigerous cyst," "maxillary sinus," and "ectopic tooth," revealed 32 cases of dentigerous cysts associated with an ectopic tooth in the maxillary antrum. ,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,, In the present paper, we report additional 2 cases of dentigerous cysts associated with an ectopic tooth in the maxillary sinus and review the literature reports of this condition over the past 34 years. Furthermore, pathogenesis of ectopic tooth, differential diagnosis, role of advanced imaging, and alternative surgical management approaches are discussed.
| Case Reports|| |
A 24-year-old man reported to the Department of Oral and Maxillofacial Radiology with a chief complaint of continuous dull pain on the right side of the face. He was healthy with no history of any systemic disorders. There were no other abnormalities identified on clinical examination except the absence of the right upper third molar. Panoramic radiography revealed a relatively large and well-defined radiolucency and an impacted molar tooth located at the posterior wall of the right maxillary sinus [Figure 1]a. CBCT scan (Kodak 9000 3D CBCT system, Kodak Carestream Health, Trophy, France) showed a fully developed ectopic maxillary third molar in the roof of right maxillary sinus and a cystic lesion measuring 3.2 cm horizontally, 2.5 cm coronally, and 2.8 cm sagittally [Figure 1]b. After the patient signed an informed consent form, it was decided to enucleation of the cyst and removal of the ectopic third molar under local anesthesia. After intraoral incision, bone was excised by piezosurgery device to prevent damage to the vital anatomic structures [Figure 1]c. Once impacted third molar and cyst exposed, then the tooth was extracted carefully with enucleation of the lesion [Figure 1]d. The sinus was irrigated with saline solution and the wound was closed primarily [Figure 1]e. The specimen was sent for histopathologic examination which confirmed dentigerous cyst. The patient was kept on regular follow-up for 18 months when there were no signs of recurrence.
|Figure 1: (a) Panoramic view of the patient. (b) Cone beam computed tomography images at axial, coronal and sagittal planes showing a well-defined radiolucency and an impacted molar tooth located at the posterior wall of the right maxillary sinus. (c) Surgical view of the cyst using piezosurgery device (d) enucleated cystic material along with the ectopic tooth. (e) Postoperative view of the wound closed primarily|
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A 19-year-old male reported to the Department of Oral and Maxillofacial Radiology, with a complaint of pain and pus discharge from his left maxillary molar region occasionally since last 6 months. The patient was healthy with no medical disorders. In intraoral examination, no swelling, erythema or any sign of infection were detected but the absence of upper left canine and second premolar was revealed. Panoramic radiography represented a well-defined unilocular radiolucency surrounding two teeth in the left posterior region of the maxilla [Figure 2]a. On CBCT scan (Kodak 9000 3D CBCT system, Kodak Carestream Health, Trophy, France) examination, a cystic expansile lesion with an impacted canine tooth adjacent to the base of the nose and a second premolar tooth located in the roof of the left maxillary sinus [Figure 2]b and c. The patient was informed about the surgical protocol and a signed consent form was taken from him. Enucleation of the cystic lesion and removal of the impacted teeth were decided. Under local anesthesia, a vestibular incision was placed from canine to first molar. A bony window was created by piezosurgery device [Figure 2]d and the teeth were removed along with the enucleation of the cyst [Figure 2]e. The cavity was irrigated with saline solution and the wound was closed primarily [Figure 2]f. Histology confirmed the wall of the cyst to be dentigerous cyst. The patient is on a regular follow-up for more than a year with no evidence of recurrence.
|Figure 2: (a) Panoramic view of the patient. (b and c) Cone beam computed tomography images at axial, coronal and sagittal planes showing a cystic expansile lesion with an impacted canine tooth adjacent to the base of the nose and a second premolar tooth located in the roof of the left maxillary sinus. (d) Surgical view of the cyst using piezosurgery device (e) view of excised material along with the ectopic tooth. (f) Postoperative view of the wound closed primarily|
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| Discussion|| |
Location of ectopic tooth in a non-dentate area like the maxillary antrum is rare. , On a PubMed search, only about 66 cases of ectopic tooth in the maxillary sinus were published from 1980 to 2012 in the English literature.  Many reasons have been proposed to explain the pathogenesis of ectopic teeth. Some reports have highlighted the role of pressure caused by a benign odontogenic cysts such as dentigerous cysts in the displacement of the tooth.  Other etiologies of ectopic teeth in the maxillary sinus include trauma/iatrogenic activity, developmental anomalies and idiopathic etiology. , In the present two cases, the etiologic factor was dentigerous cyst.
Dentigerous cysts are the benign expansile epithelial-lined developmental cavities at the cementoenamel junction that arise from the enamel organ after amelogenesis is complete.  The most reasonable theory to explain its pathogenesis appears to be the accumulation of fluid between the unerupted tooth and the surrounding dental follicle results in the displacement of the tooth to other areas such as the maxillary sinus, as were seen in our cases. 
Dentigerous cysts are usually diagnosed in second and third decade of life. The age range for the reported cases varies widely, from 4 to 57 years of age. The mean age range for the 32 previously reported cases was 24.70 years, which was almost the same with the mean age of the present 2 cases. Besides, literature review showed that there were 22 male patients and 9 female patients, which may suggest that there is a higher incidence in men than in women [Table 1].
|Table 1: Literature review of the dentigerous cyst associated with an ectopic tooth in the maxillary sinus|
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Although dentigerous cysts are most frequently associated with the crowns of an unerupted permanent teeth, few rare cases are also reported in association with the crowns of deciduous tooth,  odontoma  and supernumerary teeth.  However, dentigerous cysts in the maxilla are usually associated with impacted third molar teeth. , Among the 32 case reports summarized in [Table 1], the molar tooth is the most commonly affected. There is only one report in the literature presenting a dentigerous cyst associated with an ectopic tooth and a supernumerary tooth.  One of the present cases was associated with the molar tooth, while the other one included a canine and a premolar tooth as in the case report of Ramakrishna et al.
The dentigerous cyst may exist for several years without being noticed due to its asymptomatic nature. Although symptoms usually occur late in the process when the maxillary sinus is invaded, the occurrence of ectopic tooth in the maxillary sinus associated with dentigerous cysts may cause some complications associated with the nasal cavity and maxillary sinus. This may include facial pain, swelling, headache, nasal stuffiness, and chronic discharge. ,,,, In literature, 19 of the 32 previously reported cases showed that the most frequent symptom was painless facial swelling. This may be due to the large growth potential of dentigerous cysts which can occupy the maxillary sinus partially or totally.  Consequently, ophthalmologic and nasal symptoms may also develop such as: Obstruction of the sinus,  epiphora due to nasolacrimal duct obstruction,  recurrent sinusitis  and purulent rhinorrhea  and blurred vision.  [Table 1] is a summary of the clinical features of ectopic tooth in the maxillary sinus.
An ectopic tooth in the maxillary sinus is easily diagnosed radiographically, because of their radiopaque image. Dentigerous cyst presents as a well-defined radiolucent entity surrounding the crown of an impacted tooth is pathognomonic for this pathology.  Water's view, panoramic radiography, and plain skull radiography are simple and inexpensive methods, which can be used in daily practice.  However, two-dimensional radiographic images are difficult to interpret because of the overlapping of complex osseous structure. 
The development of CT enabled three-dimensional assessment of craniofacial structures. CT has become a widely available means for head and neck diagnosis , and various oral surgical procedures.  However, excessive radiation exposure, increased cost, and limited availability impede the routine use of this technology for dental applications. At this point, CBCT offers a promising alternative approach since it provides sub-millimeter resolution images of high diagnostic quality, with short scanning time and reduced radiation dose up to 15 times lower than Multi-Slice CT scans.  The CBCT evaluation of the presented cases showed the entire extent of the enormous lesion and helped the surgeon to accurately assess the extent of the lesion as well as determine its proximity to the contiguous vital structures.
The management of this lesion depends on the age of the patient, site and extension of the cyst. Various treatment modalities have been proposed.  Enucleation has been the standard treatment for dentigerous cysts, along with extraction of the associated tooth. In large cysts, an initial marsupialization to diminish the size of the osseous defect, followed by enucleation and tooth extraction, has been advocated. The major disadvantage of the marsupialization is recurrence or persistence of the lesion along with the residual cystic lining. 
In the present case, enucleation was preferred using piezosurgery device. In line with the tendency toward minimally invasive surgery, the use of ultrasonic waves for bone cutting has been introduced in oral and maxillofacial surgery. , An important achievement of this approach, using a piezoelectric device, is the much lower risk of causing visible injury to the adjacent soft tissues.  Recent studies showed that this new and modern technique of bone surgery enables critical operations in simple and fully executable procedures; and effectively, areas that are difficult to access have less risk of soft tissue, and neurovascular tissue damage via piezosurgery. ,
In the present paper, we report 2 cases of dentigerous cysts associated with an ectopic tooth within the maxillary sinus. The differential diagnosis of dentigerous cyst includes ameloblastoma, odontogenic keratocyst, odontogenic fibroma, odontogenic myxoma, cementomas and Pindborg tumor.  Despite their benign nature, it must be kept in mind that some untreated dentigerous cysts may, in rare cases, grow large, and may have the potential to develop into an odontogenic tumor-like ameloblastoma, or become malignant as oral squamous cell carcinoma and mucoepidermoid carcinoma.  Thus, early recognition of the entity and removal of the cysts is important to reduce morbidity.
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[Figure 1], [Figure 2]