|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 Oct 17];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|
Click here to view
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|
Click here to view
| 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|
Click here to view
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.
| References|| |
Yaseen SM, Naik S, Uloopi KS. Ectopic eruption - A review and case report. Contemp Clin Dent 2011;2:3-7.
Thakur G, Nair PP, Thomas S, Ahuja R, Kothari R. Dentigerous cyst associated with ectopic maxillary third molar in maxillary antrum. BMJ Case Rep 2011;1-4. pii: bcr0220113873. doi: 10.1136/bcr.02.2011.3873.
Mohebbi S, Salehi O, Ebrahimpoor S. Ectopic supernumerary tooth in nasal septum: A case study. Iran J Otorhinolaryngol 2013;25:183-6.
Gadre KS, Waknis P. Intra-oral removal of ectopic third molar in the mandibular condyle. Int J Oral Maxillofac Surg 2010;39:294-6.
Toranzo Fernandez M, Terrones Meraz MA. Infected cyst in the coronoid process. Oral Surg Oral Med Oral Pathol 1992;73:768.
Mamatha NS, Krishnamoorthy B, Savitha JK, Bhai P. Diagnostic CBCT in dentigerous cyst with ectopic third molar in the maxillary sinus - A case report. J Clin Diagn Res 2014;8:ZD07-9.
Ko KS, Dover DG, Jordan RC. Bilateral dentigerous cysts - report of an unusual case and review of the literature. J Can Dent Assoc 1999;65:49-51.
Buyukkurt MC, Omezli MM, Miloglu O. Dentigerous cyst associated with an ectopic tooth in the maxillary sinus: A report of 3 cases and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;109:67-71.
Haber R. Not everything in the maxillary sinus is sinusitis: A case of a dentigerous cyst. Pediatrics 2008;121:e203-7.
Shafer WG, Hine MK, Levy BM. Shafer′s Textbook of Oral Pathology. 6 th
Ed., New Delhi, Elsevier India; 2009.
de Souza LB, Gordón-Núñez MA, Nonaka CF, de Medeiros MC, Torres TF, Emiliano GB. Odontogenic cysts: Demographic profile in a Brazilian population over a 38-year period. Med Oral Patol Oral Cir Bucal 2010;15:e583-90.
Baykul T, Dogru H, Yasan H, Cina Aksoy M. Clinical impact of ectopic teeth in the maxillary sinus. Auris Nasus Larynx 2006;33:277-81.
Prabhu SP, Padwa BL, Robson CD, Rahbar R. Dentigerous cyst associated with a displaced tooth in the maxillary sinus: An unusual cause of recurrent sinusitis in an adolescent. Pediatr Radiol 2009;39:1102-4.
Srinivasa Prasad T, Sujatha G, Niazi TM, Rajesh P. Dentigerous cyst associated with an ectopic third molar in the maxillary sinus: A rare entity. Indian J Dent Res 2007;18:141-3.
Sales MA, Cavalcanti MG. Complex odontoma associated with dentigerous cyst in maxillary sinus: Case report and computed tomography features. Dentomaxillofac Radiol 2009;38:48-52.
Kasat VO, Karjodkar FR, Laddha RS. Dentigerous cyst associated with an ectopic third molar in the maxillary sinus: A case report and review of literature. Contemp Clin Dent 2012;3:373-6.
Demirtas N, Kazancioglu HO, Ezirganli S. Ectopic tooth in the maxillary sinus diagnosed with an ophthalmic complication. J Craniofac Surg 2014;25:e351-2.
Ramakrishna A, Lambade P. Dentigerous cyst associated with ectopic canine and a supernumerary tooth: A rare occurrence. J Surg Tech Case Rep 2013;5:85-8.
Onotai LO, da Lilly-Tariah OB. Dentigerous cyst associated with ectopic tooth at the roof of maxillary sinus. Int J Med Med Sci 2013;3:407-10.
Guruprasad Y, Chauhan DS, Kura U. Infected dentigerous cyst of maxillary sinus arising from an ectopic third molar. J Clin Imaging Sci 2013;3 Suppl 1:7.
Asnani S, Mahindra U, Rudagi BM, Kini Y, Kharkar VR. Dentigerous cyst with an impacted third molar obliterating complete maxillary sinus. Indian J Dent Res 2012;23:833-5.
Akyol UK, Salman IA. A case of an extensive dentigerous cyst in the maxillary sinus leading to epiphora and nasal obstruction. J Emerg Med 2012;43:1004-7.
Ngamdu YB, Kodiya AM, Sandabe MB, Garandawa HI, Isa A. Dentigerous cyst associated with ectopic supernumerary canine in the maxillary sinus. J Citation Rep 2012;2:27-30.
Thakur JS, Mohindroo NK, Sharma DR, Minhas RS, Thakur A. Ectopic canine associated with a dentigerous cyst in the maxilla. Ear Nose Throat J 2011;90:e25-7.
Mohan S, Kankariya H, Harjani B, Sharma H. Ectopic third molar in the maxillary sinus. Natl J Maxillofac Surg 2011;2:222-4.
Litvin M, Caprice D, Infranco L. Dentigerous cyst of the maxilla with impacted tooth displaced into orbital rim and floor. Ear Nose Throat J 2008;87:160-2.
Dagistan S, Cakur B, Göregen M. A dentigerous cyst containing an ectopic canine tooth below the floor of the maxillary sinus: A case report. J Oral Sci 2007;49:249-52.
Micozkadioglu SD, Erkan AN. Endoscopic removal of a maxillary dentigerous cyst. B-ENT 2007;3:213-6.
Avitia S, Hamilton JS, Osborne RF. Dentigerous cyst presenting as orbital proptosis. Ear Nose Throat J 2007;86:23-4.
Di Pasquale P, Shermetaro C. Endoscopic removal of a dentigerous cyst producing unilateral maxillary sinus opacification on computed tomography. Ear Nose Throat J 2006;85:747-8.
Tournas AS, Tewfik MA, Chauvin PJ, Manoukian JJ. Multiple unilateral maxillary dentigerous cysts in a nonsyndromic patient: A case report and review of the literature. Int J Pediatr Otorhinolaryngol Extra 2006;1:100-6.
Büyükkurt MC, Tozoglu S, Aras MH, Yolcu U. Ectopic eruption of a maxillary third molar tooth in the maxillary sinus: A case report. J Contemp Dent Pract 2005;6:104-10.
Ustuner E, Fitoz S, Atasoy C, Erden I, Akyar S. Bilateral maxillary dentigerous cysts: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:632-5.
Bajaj MS, Mahindrakar A, Pushker N. Dentigerous cyst in the maxillary sinus: A rare cause of nasolacrimal obstruction. Orbit 2003;22:289-92.
Hasbini AS, Hadi U, Ghafari J. Endoscopic removal of an ectopic third molar obstructing the osteomeatal complex. Ear Nose Throat J 2001;80:667-70.
Takagi S, Koyama S. Guided eruption of an impacted second premolar associated with a dentigerous cyst in the maxillary sinus of a 6-year-old child. J Oral Maxillofac Surg 1998;56:237-9.
Altas E, Karasen RM, Yilmaz AB, Aktan B, Kocer I, Erman Z. A case of a large dentigerous cyst containing a canine tooth in the maxillary antrum leading to epiphora. J Laryngol Otol 1997;111:641-3.
Vele DD, Sengupta SK, Dubey SP, Dokup MK. Cystic lesions of the nasal cavity and the paranasal sinuses: Report of two unusual cases. J Laryngol Otol 1996;110:1157-60.
Freedland ES, Henneman PL. An unusual cause of headache: A dentigerous cyst in the maxillary sinus. Ann Emerg Med 1987;16:1174-6.
Chuong R. Dentigerous cyst involving maxillary sinus: Report of case. J Am Dent Assoc 1984;109:59-60.
Golden AL, Foote J, Lally E, Beideman R, Tatoian J. Dentigerous cyst of the maxillary sinus causing elevation of the orbital floor. Report of a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1981;52:133-6.
Lai YT, Luk YS, Fung KH. Anomalous morphology of an ectopic tooth in the maxillary sinus on three-dimensional computed tomography images. J Radiol Case Rep 2013;7:11-6.
Lamb JF, Husein OF, Spiess AC. Ectopic molar in the maxillary sinus precipitating a mucocele: A case report and literature review. Ear Nose Throat J 2009;88:e6-e11.
Kim KS, Kim JH, Kim HJ. Dentigerous cyst presenting with sinus headache as the primary symptom. Headache 2009;49:919-22.
Abdollahifakhim S, Mousaviagdas M. Ectopic molar with maxillary sinus drainage obstruction and oroantral fistula. Iran J Otorhinolaryngol 2013;25:187-92.
Loubele M, Maes F, Schutyser F, Marchal G, Jacobs R, Suetens P. Assessment of bone segmentation quality of cone-beam CT versus multislice spiral CT: A pilot study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:225-34.
Fuchs T, Kachelriess M, Kalender WA. Technical advances in multi-slice spiral CT. Rev Eur J Radiol 2000;36:69-73.
Bou Serhal C, Jacobs R, Flygare L, Quirynen M, van Steenberghe D. Perioperative validation of localisation of the mental foramen. Dentomaxillofac Radiol 2002;31:39-43.
Sarment DP, Sukovic P, Clinthorne N. Accuracy of implant placement with a stereolithographic surgical guide. Int J Oral Maxillofac Implants 2003;18:571-7.
Scarfe WC, Farman AG, Sukovic P. Clinical applications of cone-beam computed tomography in dental practice. J Can Dent Assoc 2006;72:75-80.
Vercellotti T. Technological characteristics and clinical indications of piezoelectric bone surgery. Minerva Stomatol 2004;53:207-14.
Eggers G, Klein J, Blank J, Hassfeld S. Piezosurgery: An ultrasound device for cutting bone and its use and limitations in maxillofacial surgery. Br J Oral Maxillofac Surg 2004;42:451-3.
Rickert D, Vissink A, Slater JJ, Meijer HJ, Raghoebar GM. Comparison between conventional and piezoelectric surgical tools for maxillary sinus floor elevation. A randomized controlled clinical trial. Clin Implant Dent Relat Res 2013;15:297-302.
Pereira CC, Gealh WC, Meorin-Nogueira L, Garcia-Júnior IR, Okamoto R. Piezosurgery applied to implant dentistry: Clinical and biological aspects. J Oral Implantol 2014;40:401-8.
Rullo R, Addabbo F, Papaccio G, D′Aquino R, Festa VM. Piezoelectric device vs. conventional rotative instruments in impacted third molar surgery: Relationships between surgical difficulty and postoperative pain with histological evaluations. J Craniomaxillofac Surg 2013;41:e33-8.
Slootweg PJ. Carcinoma arising from reduced enamel epithelium. J Oral Pathol 1987;16:479-82.
[Figure 1], [Figure 2]