|Year : 2012 | Volume
| Issue : 3 | Page : 168-171
Cystic compound odontome in the anterior maxilla: A rare case report
Ritesh Rambharos Kalaskar, Ashita Ritesh Kalaskar, Chetan Arun Pol, Suvarna Krishna Ghige
Department of Pedodontics, Government Dental College and Hospital, Nagpur, Maharashtra, India
|Date of Submission||29-Nov-2012|
|Date of Acceptance||03-Jan-2013|
|Date of Web Publication||29-Apr-2013|
Ritesh Rambharos Kalaskar
Plot No. 68, Banerjee Layout, Bhagwan Nagar Road, Nagpur, Maharashtra
Source of Support: None, Conflict of Interest: None
Compound odontomes are the most common benign tumors of odontogenic origin. However, cystic compound odontome is rare, and diagnosis based on radiographic appearance is a challenge to overcome. Cystic compound odontome may attain larger size and have the potential to create complications such as root resorption, cortical bone destruction, neoplastic changes like ameloblastoma, retrograde infection, and pus discharge. Herein we report a rare case of cystic compound odontome in the anterior maxilla, which was surgically operated. However, we recommend delaying the surgery till the apical closure of the adjacent tooth to prevent complications like root fracture, damage to developing pulp, and dilacerations.
Keywords: Anterior maxilla, compound odontome, cystic lining, impacted tooth, nolla′s stages, over-retained primary teeth
|How to cite this article:|
Kalaskar RR, Kalaskar AR, Pol CA, Ghige SK. Cystic compound odontome in the anterior maxilla: A rare case report. Indian J Oral Sci 2012;3:168-71
|How to cite this URL:|
Kalaskar RR, Kalaskar AR, Pol CA, Ghige SK. Cystic compound odontome in the anterior maxilla: A rare case report. Indian J Oral Sci [serial online] 2012 [cited 2019 Aug 25];3:168-71. Available from: http://www.indjos.com/text.asp?2012/3/3/168/111183
| Introduction|| |
Odontomes are non-aggressive odontogenic developmental hamartomas consisting chiefly of enamel and dentin with variable amount of pulp and cementum.  They constitute 22% of all odontogenic tumors. , Two types of odontomes are recognized: compound and complex.  Compound odontome is composed of small tooth-like structure, whereas complex type consists of a conglomerate mass of enamel and dentin, which bears no anatomic resemblance to a tooth.  Radiographically, the compound odontome appears as a collection of tooth-like structures of varying sizes and shapes, and is seldom confused with any other lesion. Complex odontome appears as a calcified mass with a radiodensity of tooth structure and may be confused with an osteoma or some highly calcified bone lesion. It is most commonly observed in the anterior maxilla, whereas complex types are frequently located in the premolar and molar regions of both the jaws. , These lesions are more often observed in permanent dentition than in primary dentition.  The etiology of odontome is unknown, but infection, trauma, genetic and environmental factors have been proposed. , Odontomes are usually observed in the first and second decades of life, and detected on routine radiographic examination taken to evaluate the cause of over-retained primary teeth, missing teeth, or displacement of erupted teeth. 
Compound odontome is the most common; however, development of cyst in the follicle of compound odontome and enclosing the same within it is rare. Cystic compound odontome may attain a larger size and create complications, and thus warrants early diagnosis and further management.
| Case Report|| |
A 9-year-old girl was brought to the Department of Pedodontics and Preventive Dentistry because of over-retained primary maxillary right anterior teeth, which was contributing to her poor esthetics. Her medical, dental, and family history was noncontributory. She does not give any history of trauma to orofacial region.
Extraoral examination showed no facial asymmetry. Intraoral examination revealed over-retained primary maxillary right central and lateral incisors and erupted permanent maxillary left central incisor [Figure 1]. Primary maxillary right central and lateral incisors did not show any mobility, whereas primary maxillary left lateral incisor showed grade III mobility. No sign of periapical and periodontal involvement was noted in relation to maxillary anterior teeth.
Intraoral periapical radiograph showed a calcified non-homogeneous irregular radiopaque mass with periapical radiolucent rim causing root resorption of primary maxillary right central incisor (apical-mesial surface). The calcified mass was located occlusal to the impacted permanent maxillary right central and lateral incisors [Figure 2]. Provisional diagnosis of supernumerary was made; however, odontome, periapical cemental dysplasia, and benign cementoblastoma were considered in the differential diagnosis. Patient underwent surgical removal of the diagnosed lesion, along with the extraction of over-retained primary maxillary right central and lateral incisors under local anesthesia. Macroscopically, the surgical specimen showed five calcified masses and one soft tissue mass [Figure 3], which was not coinciding with the radiographic interpretation. Based on thorough clinical and surgical observations, the original diagnosis was changed to compound odontome. The specimen was sent for histopathologic examination to confirm the diagnosis. The histopathologic examination showed longitudinal section and transverse section of mature dentin having dentinal tubules surrounded by enamel matrix which was detached from dentin and little pulp tissue in the pulpal space. The enamel matrix was partially surrounded by reduced enamel epithelium. A cystic epithelial lining abutted against the dental hard tissue. These features were suggestive of cystic compound odontome [Figure 4] and [Figure 5]. Sutures were removed after 1 week and the postoperative healing was uneventful. After 15 days, eruption of permanent maxillary right lateral incisor was evident clinically and the patient was put on recall for every month.
|Figure 2: Intraoral periapical radiograph showing calcified non-homogeneous irregular radioopaque mass and impacted 11|
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|Figure 4: Photomicrograph of compound odontoma showing L.S. and T.S. of mature dentin having dentinal tubules surrounded by enamel matrix and pulp tissue (H and E, ×45)|
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|Figure 5: Photomicrograph shows cystic lining originating from the surrounding fibrous connective tissue of odontome (H and E, ×45)|
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| Discussion|| |
Cystic involvement of compound odontome is very rare. Till date, the majority of the cases reported are of cystic complex odontome. To the best of our knowledge, only four cases of cystic compound odontome have been reported.  The type of cyst developing within the follicle of compound odontome is difficult to predict in the early stages of development. Out of the four cases, calcifying odontogenic cyst was associated with two cases of compound odontome. , Piattelli  and Byung-Cheol  reported cystic compound odontome in pericoronal posterior location and posterior wall of the maxillary sinus, respectively, but they did not specify the cyst [Table 1]. The present case also describes cystic compound odontome in the anterior maxilla, but the type of cyst could not be specified. The case report is exceptional because of the difference in the radiographic, surgical, and histologic diagnosis.
Radiographically, the cystic compound odontome is seldom confused. But when the lesion is in mixed radioopaque-radiolucent stage, it can be confused with the other lesions of the jaws having similar radiographic appearance, age, and location.  In the present case, radiographically the lesion was diagnosed as supernumerary tooth, surgical observation showed compound odontome, and histopathologically cystic compound odontome was diagnosed. The lesion with radiolucent and mixed radioopaque-radiolucent appearance should be evaluated thoroughly to avoid confusion. Age of initiation of the compound odontome in permanent dentition has not been studied in detail. IIker  reported the age of initiation of complex odontome in primary dentition to be between 3 and 7 months. Maxillary primary central incisors complete root formation at 20-24 months. Permanent maxillary central incisors complete crown formation at 4-5 years and begin active eruption at 5-6 years of age. In the present case, maxillary primary central incisor showed pathologic root resorption (mesial-apical 1/3) and the active eruption of permanent maxillary central incisor was hindered by the developing cystic compound odontome. This indicates that the cystic compound odontome might have come into existence between 2 and 3 years of age, and has taken 6-7 years to reach the present size. According to the Nolla's stages of calcification, tooth takes approximately 12-18 months to complete the 3 rd stage of tooth development. 
In the present case, the odontome took 6-7 years to reach the present size (3 rd stage of tooth development), indicating that the rate of growth of odontome is approximately five times slower than the normal tooth development. This relationship between the rate of development of odontome and that of the developing tooth can be helpful in planning the surgical phase. Further research regarding the age of initiation and rate of growth of compound odontome is indicated.
Usually cystic compound odontome is asymptomatic, non-aggressive, slow growing, and measures between 1 and 3 cm in diameter.  But occasionally, this lesion can attain larger size and create complications such as tooth displacement, resorption, cortical bone destruction, neoplastic changes like ameloblastoma, retrograde infection, and pus discharge.  In the present case, although the lesion was not more than 1 × 1 cm in diameter, resorption of the root of primary maxillary right central incisor was evident. Therefore every child should undergo periodic radiographic investigation from early primary dentition period to evaluate the presence of odontome and its associated complications. However, early discovery of cystic compound odontome does not warrant immediate surgical intervention. Its removal is to be delayed till the calcification of the adjacent permanent tooth in order to avoid complications due to surgery, such as hypoplasia, odontoma-like malformations, removal of tooth bud, and alteration in the position of tooth bud. , We further recommend delaying the surgery till the apical closure of the adjacent tooth (depending on the location) to prevent complications such as root fracture, damage to developing pulp, and dilacerations.
| Conclusion|| |
Though cystic compound odontome is a rare lesion found in the oral cavity, its early diagnosis and management is important to prevent its potential complications such as hypoplasia, odontoma-like malformations, root fracture, damage to developing pulp, dilacerations, etc., For this, routine radiographic examination should be conducted for every child from early primary dentition period. Also, more emphasis should be given to correctly determine the relationship of rates of development of odontome with that of developing tooth, so as to plan the surgical phase using superior methods of diagnostics such as computerized tomography, magnetic resonance imaging, and Digital Volumetric Tomography (DVT).
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]