Compound-complex odontoma: A case report of a rare variant
Nishath Khanum, Mahesh Mysore Shivalingu, Naresh Lingaraju, Srisha Basappa
Department of Oral Medicine and Radiology, Farooqia Dental College and Hospital, Mysore, Karnataka, India
Date of Submission 22-Aug-2014
Date of Acceptance 29-Mar-2015
Date of Web Publication 22-Apr-2015
Nishath Khanum
Department of Oral Medicine and Radiology, Farooqia Dental College and Hospital, Mysore - 570 021, Karnataka
The odontoma is a benign tumor containing all the various component tissues of the teeth. It is the most common odontogenic tumor representing 67% of all odontogenic tumors. Odontomas are considered to be developmental anomalies (hamartomas) rather than true neoplasms. Based on the degree of morphodifferentiation or on the basis of their resemblance to normal teeth, they are divided into compound and complex odontomas. The compound odontoma is composed of multiple, small tooth-like structures. The complex odontoma consists of a conglomerate mass of enamel and dentin, which bears no anatomic resemblance to a tooth. They are usually diagnosed on routine radiological examinations in the second decade of life and are often slow growing and non-aggressive in nature. Here, we report a case of rare, unusually large, compound-complex odontoma, located in the left anterior maxilla of a 13-year-old male patient.
Keywords: Compound complex odontoma, hamartoma, odontogenic tumor, radiopacities
Khanum N, Shivalingu MM, Lingaraju N, Basappa S. Compound-complex odontoma: A case report of a rare variant. J Indian Acad Oral Med Radiol 2014;26:463-6
Khanum N, Shivalingu MM, Lingaraju N, Basappa S. Compound-complex odontoma: A case report of a rare variant. J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2019 Jul 19];26:463-6. Available from: http://www.jiaomr.in/text.asp?2014/26/4/463/155668
The term odontoma was used earlier to describe any odontogenic origin tumour. In 1867, Paul Broca coined the term odontoma. He defined the term odontoma as 'tumors formed by the overgrowth of transitory or complete dental tissues. [1] Because the odontogenic cells do not reach the normal state of morphodifferentiation, the enamel and dentin are formed in an abnormal pattern. Although unknown, the etiology is said to include local trauma or infection. A gene mutation or postnatal interference with the genetic control of tooth development is also one of the suggested causes. [2] Odontomas are slow growing and often non-aggressive in nature. With the mean age at the time of diagnosis being 14 years, odontomas are usually detected during the first two decades of life. [3]
The World Health Organization (WHO) classifies odontomas into compound and complex odontomas. Complex odontomas are less common than the compound variety, and the latter occurs more commonly in the maxilla, having a predilection for the incisor-canine region without gender bias. [2] The complex odontoma is more common in the mandibular posterior teeth region and has a female predilection. [4] However, rarely, lesions may show features of both compound and complex odontomas. The treatment of choice is surgical removal of the lesion, followed by histopathological examination to confirm the diagnosis.
A 13-year-old male patient visited the department of Oral Medicine and Radiology, with a chief complaint of swelling in the upper left back teeth region since 1 year. The swelling was gradually and constantly increasing in size to reach the present size. No history of pus, blood or watery discharge or any other secondary changes were noted.
On local extraoral examination, a solitary, ill-defined swelling was present in the left middle 1/3 rd of the face. On intraoral hard tissue examination, 23, 24 and 25 were clinically missing and 63 was retained with pre-shedding mobility. There was a well-defined solitary oval swelling present in relation to the left buccal alveolar bone, measuring about 2 × 3 cm, extending from the distal aspect of 22 to the mesial aspect of 26. The mucosa over the swelling was normal with no pus, blood or watery discharge. There was no evidence of ulcer or growth noted on the mucosa. On palpation, the swelling was non-tender, hard in consistency with a smooth surface and well-defined borders. Expansion of the buccal cortical plate was noted [Figure 1].
Figure 1: Intraoral photograph
The panoramic radiograph (OPG) revealed a well-defined mixed radiopaque-radiolucent lesion measuring about 3 × 3 cm in diameter in the left maxilla, surrounded by a thin radiolucent rim. The lesion extended superiorly up to the floor of the maxillary sinus and the nasal cavity and inferiorly to the alveolar crest, medially to the distal root surface of 22 and laterally to the mesial surface of 26. The missing premolars were pushed distally near the mesial root surface of 26 and the canine was placed near the infra-orbital margin [Figure 2]. The occlusal radiograph also showed a solitary, well-defined, mixed radiopaque-radiolucent lesion measuring about 3 × 3 cm in the left posterior maxilla, surrounded by a thin radiolucent rim. The impacted 24 and 25 were displaced distally and palatally in relation to 26 [Figure 3].
Figure 2: Preoperative OPG showing the mixed radiopaque-radiolucent lesion
Figure 3: Maxillary occlusal view
Based on the history and clinical examination, a provisional diagnosis of dentigerous cyst was made. Differential diagnosis included adenomatoid odontogenic tumor, odontoma, and keratocystic odontogenic tumor. Based on the radiographic appearance a diagnosis of complex odontoma was made. The tumor was surgically removed under local anesthesia [Figure 4]. The gross specimen consisted of an irregular mass of calcified tissue as well as multiple teeth-like structures [Figure 5]. The post-operative OPG revealed complete enucleation of the tumor [Figure 6].
Figure 4: Intraoperative photograph showing the exposed odontoma
Figure 5: Specimen after surgical removal
Figure 6: Post-operative OPG
On histopathological examination, the sections revealed calcified structures like enamel, dentin, and cementum which were intermingled with pulp-like tissues in few areas. The calcified structures were arranged regularly in few areas, whereas, in few areas it was haphazardly arranged. Few odontogenic islands were seen in a fibro-vascular connective tissue [Figure 7] and [Figure 8]. Based on the appearance of the gross specimen and the histopathological examination a diagnosis of compound-complex odontoma was made, which is a rare entity.
Figure 7: Histopathological section showing enamel and dentin-like structure
Figure 8: Histopathological section showing haphazardly arranged enamel and dentin
Rather than being true neoplasms, odontomas are hamartomatous lesions or malformations. [2],[3] They constitute 22% of all odontogenic tumours of the jaw making them the most common benign odontogenic tumours. [4] Majority of the complex odontomas reported in literature measure around 1-2 cm in diameter. However, in the present case, the odontoma measured about 3 × 3 cm, which is very rare. The anterior maxilla is the common site of occurrence of compound odontomas, whereas the complex odontomas frequently occur in the posterior mandibular region. [2],[4]
The present case is unique in that it has combined features of both compound and complex odontoma. Very few cases of compound - complex odontoma have been reported in literature. [5] In a review of 160 cases of odontomas by Tekkesin et al., of all the investigated cases, 99 were complex, 57 were compound, and only four were mixed odontomas. This further confirms the rare occurrence of the mixed odontoma, that is the compound - complex odontoma. [6]
Although the etiology of odontomas remains unknown, local trauma, infection, family history and genetic mutation have been suggested as possible causes. Compound odontomas usually do not cause any bony expansion, whereas complex odontomas often cause slight or even marked bony expansion. Pathological changes such as impaction, malpositioning, aplasia, malformation and devitalization of the adjacent teeth can be caused by 70% of the odontomas. [7] The lesions are usually asymptomatic and are invariably discovered on routine radiographic examinations. The radiological appearance of odontomas depends on their stage of development and degree of mineralization and is divided into three stages. [2] Since the present case had a mixed radiopaque radiolucent lesion, we considered the present lesion to be in the intermediate stage of development.
Cases of large complex odontomas erupting into the oral cavity and also infected complex odontomas have been reported. When odontomas occur in association with missing teeth, it is a rare occurrence. Early diagnosis will facilitate removal of the cause of eruption disturbances, which is important in the developing dental arch. For early detection of odontomas, panoramic radiography and a proper clinical examination are important. Complications can be prevented by surgical excision followed by histopathological analysis. A careful follow-up of the case, both clinically and radiographically is necessary to assess the eruption of the unerupted or impacted teeth. The odontomas have a favorable prognosis with rare chances of relapse.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]
Khanum N
Shivalingu MM
Lingaraju N
Basappa S
Compound complex odontoma
hamartoma
odontogenic tumor
radiopacities
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