|Year : 2015 | Volume
| Issue : 2 | Page : 273-277
A rare dentigerous cyst of maxillary central incisor associated with multiple impacted teeth: Case report and review of literature
Pushkar Dahiwal, Sonia Sodhi, Lata Kale, Neha Khambete
Department of Oral Medicine, Diagnosis and Radiology, CSMSS Dental College and Hospital, Aurangabad, Maharashtra, India
|Date of Submission||13-Nov-2014|
|Date of Acceptance||05-Oct-2015|
|Date of Web Publication||21-Nov-2015|
Department of Oral Medicine, Diagnosis and Radiology, CSMSS Dental College and Hospital, Kanchanwadi, Aurangabad, Maharashtra
Source of Support: None, Conflict of Interest: None
| Abstract|| |
We report a case of a 15-year-old male who presented with a chief complaint of swelling in the maxillary anterior region of the jaw since 5 years. Patient gave a history of trauma to the same region 7 years back. On examination, a large intraoral swelling with multiple missing permanent teeth was noted. On radiographic examination, a large corticated radiolucency surrounding the maxillary left central and lateral incisors and canine was seen in the anterior maxilla. Surgical enucleation was carried out and histopathologic examination confirmed diagnosis of an infected dentigerous cyst. The present case report is unique as the cyst enclosed the three permanent maxillary teeth: Central incisor, lateral incisor, and canine.
Keywords: Dentigerous cyst, maxillary permanent central incisor, multiple impacted teeth, odontogenic cyst
|How to cite this article:|
Dahiwal P, Sodhi S, Kale L, Khambete N. A rare dentigerous cyst of maxillary central incisor associated with multiple impacted teeth: Case report and review of literature. J Indian Acad Oral Med Radiol 2015;27:273-7
|How to cite this URL:|
Dahiwal P, Sodhi S, Kale L, Khambete N. A rare dentigerous cyst of maxillary central incisor associated with multiple impacted teeth: Case report and review of literature. J Indian Acad Oral Med Radiol [serial online] 2015 [cited 2020 May 25];27:273-7. Available from: http://www.jiaomr.in/text.asp?2015/27/2/273/170156
| Introduction|| |
Adentigerous cyst can be defined as the one that encloses the crown of an unerupted tooth by expansion of its follicle and is attached to its neck. They are most common developmental odontogenic cysts accounting for more than 24% of the jaw cysts.  Majority of the dentigerous cysts involve the mandibular third molars and the maxillary permanent canines, followed by the mandibular premolars, maxillary third molars, and rarely the central incisors, supernumerary teeth, and mesiodens.  Dentigerous cysts most commonly occur in second and third decades of life and have a male predilection. Studies have shown that the incidence rate of dentigerous cysts involving the maxillary central incisors was 1.5%, as compared to 45.7% involving the mandibular third molar.  Daley et al. reported an incidence rate of 0.1-0.6%, whereas Shear found the incidence to be 1.5%. Mourshed stated that only 1.44% of impacted teeth undergo dentigerous cyst transformation. So dentigerous cysts involving the permanent central incisor are rare. 
| Case Report|| |
The case reported here presented to the Department of Oral Medicine and Radiology with the chief complaint of slow-growing, painless swelling in the maxillary anterior region of the jaw since 5 years. History of present illness revealed that he was apparently normal 7 years back, when he had met with a road traffic accident in which he sustained trauma to the left side of the face. Patient had consulted a general surgeon that time. He neither had any fractures of the dentoalveolar complex, nor did he note avulsion of any tooth. At that time, he had experienced dull, aching pain in the maxillary anterior region of the jaw, which subsided gradually on its own after 1 month. The patient was asymptomatic for 2 years, after which he noticed a small swelling in the same region. The swelling gradually increased to the present size. The medical history was non-contributory.
On general examination, the patient appeared healthy. Extraoral examination revealed facial asymmetry with a solitary, diffused swelling on the left side of the face, extending horizontally from the midline to outer canthus of eye and vertically from the infraorbital margin to corner of the mouth, measuring about 3 × 4 cm in diameter. The surface texture was smooth. The swelling was afebrile, painless, and firm on palpation. On intraoral examination, an expansile lesion was seen in the left maxillary region on the buccal aspect, extending from labial frenum to the distal surface of the 1 st molar obliterating the buccal vestibule. The swelling was firm in consistency and non-tender on palpation. The swelling extended palatally in the periapical region of canine and first premolar. The palatal swelling was soft, non-fluctuant, and non-tender in nature. No bruit or pulsation was palpated. No significant changes were found in periodontal tissues. Hard tissue examination revealed missing permanent left maxillary central and lateral incisor and canine and over-retained root pieces of deciduous left maxillary central and lateral incisor and canine [Figure 1]. A clinical diagnosis of dentigerous cyst was given. On radiographic examination, maxillary lateral occlusal projection showed a unilocular, well-defined radiolucent lesion with hyperostotic borders in the left maxilla, measuring approximately 3 × 3.5 cm in size and surrounding three impacted permanent teeth - central incisor, lateral incisor, and canine [Figure 2]. An orthopantomograph (OPG) revealed a well-defined, corticated radiolucent lesion involving the crown of impacted left maxillary permanent central and lateral incisor and canine in the anterior maxilla, extending from the periapical region of right permanent maxillary central incisor up to the left permanent maxillary 1 st molar region and superiorly toward nasal spine and floor of the nose. There was no evidence of resorption of the roots of associated permanent teeth. Impacted teeth seemed to be arranged in a circular manner in the radiolucency [Figure 3]. Computed tomographic (CT) examination showed a large, well-defined, unilocular expansile lesion with central hypodense areas surrounded by thin cortical bone involving the alveolar process of the maxilla, measuring about 26.9 × 33.1 × 28.9 mm, with multiple haphazardly placed unerupted teeth within (root directed away and crown directed toward the lesion). Inferiorly, the lesion was slightly bulging into the oral cavity with thinning of the cortical bone. Superiorly, it was bulging into the left maxillary sinus [Figure 4]. These findings were in favor of a dentigerous cyst.
|Figure 1: (a) Extraoral photograph and (b) Intraoral photograph of the patient|
Click here to view
|Figure 2: Maxillary lateral occlusal projection showing unilocular, welldefined radiolucent lesion with hyperostotic borders in the left maxilla|
Click here to view
|Figure 3: OPG revealing a well-defi ned, corticated radiolucent lesion involving the crown of impacted left maxillary permanent central and lateral incisor, and canine in the anterior maxilla|
Click here to view
|Figure 4: CT scan: (a-c) Coronal sections, (d and e) Axial sections, and (f) Sagittal section showing hypodense corticated area with multiple impacted teeth|
Click here to view
An aspiration biopsy of the swelling revealed the presence of clear, straw-colored fluid suggestive of an odontogenic cyst. On surgical exposure, the lesion was of cystic quality and was found attached to the crown of maxillary central incisor. The lesion was surgically enucleated and extraction of the impacted teeth was carried out [Figure 5]. The surgical specimen was sent for histopathologic examination. Histopathologic examination confirmed the diagnosis of infected dentigerous cyst [Figure 6]. The patient remained asymptomatic. Clinical and radiographic evaluation after 6 months revealed normal healing with no evidence of recurrence. A prosthodontic consultation was taken for the replacement of the missing teeth. Patient has been rehabilitated with a removable partial denture.
|Figure 5: Surgical exposure of the lesion revealing attachment of cystic lining to the cementoenamel junction of the impacted central incisor|
Click here to view
| Discussion|| |
The exact histopathogenesis of the dentigerous cyst is not known. It is stated that the dentigerous cyst develops around the crown of an unerupted tooth by accumulation of fluid either between the reduced enamel epithelium and enamel or in between the layers of the enamel organ. This fluid accumulation occurs as a result of the pressure exerted by an erupting tooth on an impacted follicle, which obstructs the venous outflow and thereby induces rapid transudation of serum across the capillary wall.  Studies by Ben and Altini indicate that at least two types of dentigerous cysts occur. The first type is developmental in origin and occurs in mature teeth usually as a result of impaction. The second type is inflammatory in origin and occurs in immature teeth as a result of inflammation from a non-vital deciduous tooth or some other source spreading to involve the tooth follicle.  These are diagnosed in the first and early part of the second decade, either on routine radiographic examination or when the patients complain of swelling and pain. In our case, the cyst was probably of inflammatory origin. There are very few cases in literature reporting occurrence of dentigerous cyst associated with multiple impacted teeth. A search of database revealed five cases of such occurrences. [Table 1] gives a summary of such cases. ,,,, Majority of the patients showed mandible as the common site of involvement. None of the case reports had involvement of more than three teeth. In this unique case report, the cyst was found to be enclosing three permanent maxillary teeth: Central incisor, lateral incisor, and canine.
|Table 1: Review of cases of dentigerous cyst associated with multiple impacted teeth|
Click here to view
Radiographically, the dentigerous cyst typically appears as a well-circumscribed, unilocular, usually symmetric radiolucency around the crown of an impacted tooth. An important diagnostic point is that this cyst attaches at the cementoenamel junction.  The internal aspect of the cyst is completely radiolucent except for the crown of the involved tooth. One of the most difficult conditions to distinguish in the differential diagnosis is hyperplastic follicle. Other conditions that must be excluded in the diagnosis are odontogenic keratocyst, ameloblastic fibroma, and cystic ameloblastoma.
Water's, panoramic, and skull radiography are simple and inexpensive methods that can be used in daily practice. The structure of a tooth can be clearly detected on panoramic radiographs. Therefore, panoramic radiographs are preferred over CT. Although the structure of a tooth can be clearly detected on panoramic radiographs, they are inadequate for localizing maxillary ectopic teeth due to their inherently less sharp image and ghost image. A CT scan provides superior bony detail, allowing for visualization of the size and extent of the lesion with determination of orbital or nasal invasion or involvement. Therefore, CT may be more valuable than plain film radiographs, not only for definitive diagnosis but also for evaluation of the associated pathology, exact localization of the ectopic tooth, and proper treatment planning. In our case, spiral CT was used for localization of multiple impacted teeth and knowing the extension of the lesion. 
Unlike other odontogenic cysts, the epithelial cells lining the lumen of the dentigerous cyst possess an unusual ability to undergo metaplastic transition. Occasionally, some untreated dentigerous cysts can develop into an odontogenic tumor (e.g., ameloblastoma) or a malignancy (e.g., oral squamous cell carcinoma).  Marsupialization and surgical enucleation of the cyst may be the treatment of choice. In the present case, surgical enucleation of the cyst was done. Surgical evaluation confirmed that the cystic lining was associated with maxillary central incisors, incorporating lateral incisor and maxillary canine. As the histopathologic appearance of the lining epithelium is not specific, the diagnosis relies on the radiographic and surgical observation of the attachment of the cyst to the cementoenamel junction.
| Conclusion|| |
Thus, as very few cases of dentigerous cysts involving multiple impacted teeth are reported in literature, we intend to add valuable information to those previously reported cases by presenting this very rare case of dentigerous cyst with impacted maxillary central incisor involving multiple impacted teeth.
Declaration of Patient Consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Dr. Rajan Akole is acknowledged for the surgical excision of the lesion.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Daley TD, Wysocki GP. The small dentigerous cyst. A diagnostic dilemma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1995;79:77-81.
Anehosur V. Dentigerous cyst of maxillary sinus with an impacted tooth in an unusual position: A case report. J Oral Maxillofac Surg 2008;7:383-6.
Shear M. Cysts of the Oral Region. 2 nd
ed. Bristol: Wright SG; 1983. p. 56-75, 479-82.
Benn A, Altini M. Dentigerous cysts of inflammatory origin. A clinicopathologic study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1996;81:203-9.
Pramod SR, Shukla JN. Dentigerous cyst of maxilla in a young child. Natl J Maxillofac Surg 2011;2:196-9.
Ivy RH. Multiple dentigerous cysts: With special reference to occurrence in siblings. Ann Surg 1939;109:114-25.
Gonzalez SM, Spalding PM, Payne JB, Giannini PJ. A dentigerous cyst associated with bilaterally impacted mandibular canines in a girl: A case report. J Med Case Rep 2011;5:230.
Agrawal M, Raghavendra PD, Singh B, Agrawal N. Multiple teeth in single dentigerous cyst follicle: A perplexity. Ann Maxillofac Surg 2011;2:187-9.
Rohilla M, Namdev R, Dutta S. Dentigerous cyst involving multiple impacted teeth: A rare case report. J Indian Soc Pedod Prev Dent 2011;29:244-7.
Nishikawa S, Cheng J, Koyano Y, Nomura T, Kato T, Saku T. Dentigerous cyst involving two impacted molars; Report of a case. Oral Med Pathol 1996;1:60-2.
Ziccardi VB, Eggleston TI, Schnider RE. Using fenestration technique to treat a large dentigerous cyst. J Am Dent Assoc 1997;128:201-5.
Khambete N, Kumar R, Risbud M, Kale L, Sodhi S. Dentigerous cyst associated with an impacted mesiodens: Report of 2 cases. Imaging Sci Dent 2012;42:255-60.
Slootweg PJ. Carcinoma arising from reduced enamel epithelium. J Oral Pathol 1987;16:479-82.
Nishikawa S, Cheng J, Koyano Y, Irie T, Nomura T, Kato T, et al
. Dentigerous cyst involving two impacted molars: Report of a Case. Oral Med Pathol 1996;1:60-2.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]