|Year : 2015 | Volume
| Issue : 2 | Page : 99-102
Erupted complex composite odontoma: Report of two atypical cases
Preeti Tomar Bhattacharya1, Soumyabrata Sarkar1, Tanya Khaitan1, Arpita Kabiraj2
1 Department of Oral Medicine and Radiology, Haldia Institute of Dental Sciences and Research, Haldia, West Bengal, India
2 Department of Oral Pathology and Microbiology, Haldia Institute of Dental Sciences and Research, Haldia, West Bengal, India
|Date of Web Publication||11-Dec-2015|
Preeti Tomar Bhattacharya
Department of Oral Medicine and Radiology, Haldia Institute of Dental Sciences and Research, Haldia - 721 645, West Bengal
Source of Support: None, Conflict of Interest: None
Odontomas are nonaggressive, hamartomatous developmental malformations of odontogenic origin. They are considered one of the most common odontogenic lesions composed by diverse dental tissues. They may interfere with the eruption of an associated tooth and are more prevalent in the posterior mandible. The eruption of a complex odontoma into the oral cavity is rare. Here, we report such two rare cases of gigantic erupted complex composite odontomas.
Keywords: Cementoid, dentinoid, hamartoma, mutation
|How to cite this article:|
Bhattacharya PT, Sarkar S, Khaitan T, Kabiraj A. Erupted complex composite odontoma: Report of two atypical cases. J Dent Allied Sci 2015;4:99-102
|How to cite this URL:|
Bhattacharya PT, Sarkar S, Khaitan T, Kabiraj A. Erupted complex composite odontoma: Report of two atypical cases. J Dent Allied Sci [serial online] 2015 [cited 2020 Jan 26];4:99-102. Available from: http://www.jdas.in/text.asp?2015/4/2/99/171546
| Introduction|| |
Odontomas are hamartomatous growths of dental tissues such as enamel, dentin, cementum and pulp that comprise about 22% of all odontogenic tumors. According to the World Health Organization classification of 2005, there are two types of odontomas: Complex (an amorphous conglomeration of dental tissues consisting of enamel, dentin, cementum, pulp, and enamel organ) and compound odontomas (many separate, small, tooth-like denticles in a structural and more orderly pattern including rudimentary teeth).  Eruption of complex composite odontoma (CO) in the oral cavity is extremely rare. As per the review of Amado Cuesta et al. on 61 cases of odontomas, only 1.6% of complex odontomas erupt into the oral cavity.  Here, we present two such cases where complex COs erupted in the oral cavity.
| Case Reports|| |
A 30-year-old female patient reported with a complaint of painful hard mass in the lower right back jaw region since 1 year. The pain was sudden in onset, dull, intermittent, aggravated on chewing and relieved on medication. Intraoral examination revealed well-defined yellowish tooth like mass in the right pterygomandibular raphae region measuring approximately 2 cm × 1 cm in size. The surface over the mass appeared smooth. On palpation, the mass was tender and bony hard in consistency [Figure 1]. Orthopantomograph revealed well-defined nonhomogeneous radiopaque tooth like mass [marked A in [Figure 2] measuring 3 cm × 2 cm distal to edentulous region i.r.t. 47 extending up to ascending ramus of the mandible that was surrounded by radiolucent shadow [marked B in [Figure 2] lined by a thin radiopaque sclerotic border [marked C in [Figure 2].
A young female patient presented with an asymptomatic hard mass in the lower right back teeth region since 6 months. The patient complained of constant irritation of tongue. Intraoral examination revealed the presence of a well-defined yellowish jagged calcified mass on the right alveolar region i.r.t. 46, 47, 48 measuring approximately 3.2 cm × 2.5 cm in size. On palpation, the mass was nontender, bony hard in consistency and covered with calculus deposits [Figure 3]. Orthopantomograph revealed well-defined nonhomogenous radiopaque mass [marked A in [Figure 4] roughly 4 cm × 3 cm in dimension seen extending from the distal aspect of 46 to the ramus of mandible posteriorly surrounded by radiolucent halo [marked B in [Figure 4] and lined by a thin sclerotic border [marked C in [Figure 4]. Below the radiopaque mass, malformed and horizontally impacted 47 [marked D in [Figure 4] and vertically impacted 48 [marked E in [Figure 4] were also seen in close proximity to the inferior alveolar canal. Resorption of distal root of 46 was also eminent.
Based on the history, clinical findings and radiological examination, a provisional diagnosis of erupted complex odontoma was obtained for both the cases. The aforementioned cases had undergone surgical removal of the calcified masses, and the specimens were sent for histopathological analysis.
The photomicrographs of case 1 showed the irregular arrangement of dentin [Figure 5]. The photomicrographs of case 2 revealed multiple areas of calcified areas having tubular structures resembling dentin (dentinoid) [Figure 6]. At some locations, there were intensely hematoxyphilic calcified areas resembling cementum (cementoid). Scanty collagenous tissue was also seen in between calcified tissues [Figure 7]. Based on the above features, final diagnosis of complex odontoma was obtained.
|Figure 6: Photomicrograph of case 2 showing multiple dentinal tubules (×10)|
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|Figure 7: Photomicrograph of case 2 showing cementum and collagenous masses (×10)|
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Therefore, the patients were being followed up after 2 months and 1 year postoperatively and successful wound healing was noted.
| Discussion|| |
The word "odontoma" stands for a tumor of odontogenic origin and "composite" for something made up of many parts or elements. The term "odontoma" was initially used by Paul Broca in 1867 and classified by Thoma and Goldman in 1946. There are three clinical variants of odontomas-central (intraosseous), peripheral (extraosseous), and erupted odontomas.  The odontoma occurring in the soft tissue overlying the teeth bearing portion is the peripheral odontoma while an erupted odontoma is the erupted counterpart of intraosseous odontoma in the oral cavity.
The etiological theories of CO vary from pathological processes such as inadequate growth space, infection, trauma, mature ameloblasts, dental lamina remnants, and odontoblastic hyperactivity to genetic mutation. Furthermore, Gardner and Hermann syndromes are allegedly associated with odontomas.
Genetic factors control odontoma formation through one or more processes:
- By inheritance of abnormal genes: According to Hitchin, odontomas may be inherited postnatally through a mutant gene with genetic control of tooth development. 
- By mutation in the genes: Mutations involving LHX8 and PARP1 genes have also been implicated in the morphogenesis of compound and complex odontomas. ,
- By interference with the mechanism by which gene control tooth formation and form: It has been found in various researches that similar genes control the formation of normal teeth as well as abnormal dental components in odontogenic tumors. The abnormal expression of these genes leads to the formation of an odontogenic tumor. 
Odontomas are more frequently noted in permanent dentition and slightly more in males. They have limited growth potential, and the largest dimension is usually up to 3 cm. Bagewadi et al. reported an erupted complex odontoma that measured 3.5 cm × 4 cm.  Similarly, a large erupted complex odontomas of 3 cm × 2 cm has been described by Vengal et al.  Interestingly, mention of a huge maxillary lesion causing erosion of infraorbital rim has also been found in literature.  Both the current cases were equal or larger in 3 cm diameter thus falling among scarce massive erupted complex odontomas.
Clinically, the erupted odontomas resemble a chunk of solidified calculus over the occlusal surfaces of teeth. They might also be mistaken as a necrotic bone of osteomyelitis in the cases where they erupt until the level of the alveolar ridge. The attempt at their removal without appropriate diagnosis may be troublesome to the patient as well as the clinician. The odontomas irrespective of their eruptive behavior can get infected due to the replacement of large portions of the normal bone with avascular tumor tissue. Cystic degeneration of odontomas can also set in as a substantial number of cases testified the coexistence of odontomas with a dentigerous cyst. , Further complication like the evolution of ameloblastoma from the dentigerous cyst is also not a distant possibility.  Such impediments warrant earliest diagnosis and removal of this lesion.
By and large the majority of patients are asymptomatic amid few cases reporting with halitosis, tongue or cheek irritation, pain, swelling and pus discharge. In the first case, the patient presented with pain whereas in the second case the patient complained of constant irritation of tongue. Bagewadi et al. reported for the first time a lesion causing ulceration on the buccal mucosa.  Facial asymmetry due to the progression of a massive maxillary complex odontoma has also been encountered. , Ludwig's angina secondary to infection of an erupted complex odontoma has also been described. 
The lack of periodontal ligament in odontoma rules out its eruption mechanism being similar to that of normal teeth. Three eruption theories have been advocated in literature i.e., pressure resorption, bony remodeling and traumatic. The increase in the size of odontoma and subsequent pressure may lead to sequestration of overlying bone and resultant occlusal movement or eruption of an odontoma.  Apart from the above cause, bony remodeling may also play a vital role in odontoma eruption. Immunocytochemical studies have suggested that reduced enamel epithelium and dental follicle initiate cellular activities that result in recruitment of osteoclasts to the dental follicle. A path of least resistance is created by secretion of proteases by reduced enamel epithelium. It has been postulated that pressure resorption theory is prevalent in older individuals whereas bone remodeling plays a significant role at a younger age. 
Radiographically, three stages of complex odontomas formation have been elaborated in literature. The initial or first stage is of a radiolucent lesion due to the absence of calcification of dental tissues. In second or intermediate stage, the lesion is incomplete or partially calcified and third or mature stage comprises of a nonhomogeneous predominantly radiopaque lesion with varying densities of hard dental tissues which are surrounded by a radiolucent halo lined by a thin radiopaque sclerotic border. Each one of the presented cases showed mature or the third stage of radiographic appearance.
Microradiography is also a beneficial modality in establishing a definite diagnosis. In this technique, various histological features are recognized by their varying degrees of radiopacities and radiolucencies. In addition, the resolution of microradiographic images was found to be comparable to histological images. 
The radiographic differential diagnosis of benign cementoblastoma, periapical cemental dysplasia, cement-ossifying fibroma and ameloblastic fibro-odontoma should be considered for an intraossoeus complex odontoma. A benign cementoblastoma is seen as a well-defined radiopacity surrounded by a radiolucent rim that is attached to a tooth root apex. Cemento-ossifying fibroma is characterized by diffuse less dense radiopaque lesion when it is mature, not surrounded by a radiolucent rim and not associated with impacted teeth. Furthermore, odontomas occur more in young individuals as compared to cemento-ossifying fibromas. In periapical cemental dysplasia, classically multiple radiopacities centered in the periapical regions of the teeth are noted and has a wider uneven sclerotic border. Ameloblastic fibro-odontoma is more radiolucent as compared to odontoma, has numerous small mature dental tissues dispersed within the lesion and has predilection for older individuals. The complex odontoma typically shows accumulation of disorganized dental tissues in its center. 
Conservative surgical enucleations were performed of the reported cases and were recurrence free 1 year posttreatment.
| Conclusion|| |
Complex CO is an agglomerate of all dental tissues which show abnormal morphodifferentiation with normal histodifferentiation. The authors attempted to elaborate on two cases of large erupted complex odontomas in the right mandibular posterior region of the oral cavity associated with eruption disturbances. The oral diagnosticians should be sentient of clinical features, radiographic manifestations, and treatment options of this benign lesion to avoid untoward outcomes.
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| References|| |
Barnes L, Eveson JW, Reichart P, Sidransky D. World Health Organization Classification of Tumors. Pathology and Genetics of E558 Head and Neck Tumors. Lyon: IARC Press; 2005. p. 310.
Amado Cuesta S, Gargallo Albiol J, Berini Aytés L, Gay Escoda C. Review of 61 cases of odontoma. Presentation of an erupted complex odontoma. Med Oral 2003;8:366-73.
Junquera L, de Vicente JC, Roig P, Olay S, Rodríguez-Recio O. Intraosseous odontoma erupted into the oral cavity: An unusual pathology. Med Oral Patol Oral Cir Bucal 2005;10:248-51.
Hitchin AD. The aetiology of the calcified composite odontomes. Br Dent J 1971;130:475-82.
Kim JY, Jeon SH, Park JY, Suh JD, Choung PH. Comparative study of LHX8 expression between odontoma and dental tissue-derived stem cells. J Oral Pathol Med 2011;40:250-6.
Nozaki T, Watanabe M, Morita J, Ohura K, Kusama K. Expression of PARP1 in odontoma. J Meikai Dent Med 2006;35:1-6.
Papagerakis P, Peuchmaur M, Hotton D, Ferkdadji L, Delmas P, Sasaki S, et al.
Aberrant gene expression in epithelial cells of mixed odontogenic tumors. J Dent Res 1999;78:20-30.
Bagewadi SB, Kukreja R, Suma GN, Yadav B, Sharma H. Unusually large erupted complex odontoma: A rare case report. Imaging Sci Dent 2015;45:49-54.
Vengal M, Arora H, Ghosh S, Pai KM. Large erupting complex odontoma: A case report. J Can Dent Assoc 2007;73:169-73.
Murphy C, O'Connell JE, Cotter E, Kearns G. Management of large erupting complex odontoma in maxilla. Case Rep Pediatr 2014;2014:963962.
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.
Astekar M, Manjunatha BS, Kaur P, Singh J. Histopathological insight of complex odontoma associated with a dentigerous cyst. BMJ Case Rep 2014;2014. pii: bcr2013200316.
Arunkumar KV, Vijaykumar, Garg N. Surgical management of an erupted complex odontoma occupying maxillary sinus. Ann Maxillofac Surg 2012;2:86-9.
Bertolai R, Acocella A, Sacco R, Agostini T. Submandibular cellulitis (Ludwig's angina) associated to a complex odontoma erupted into the oral cavity. Case report and literature review. Minerva Stomatol 2007;56:639-47.
Kaneko M, Fukuda M, Sano T, Ohnishi T, Hosokawa Y. Microradiographic and microscopic investigation of a rare case of complex odontoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;86:131-4.
Wood NK, Goaz PW, Lehnert J. Mixed radiolucent-radiopaque lesions associated with teeth. In: Wood NK, Goaz PW, editors. Differential Diagnosis of Oral and Maxillofacial Lesions. Singapore: Harcourt Brace & Company Asia Pte Ltd.; 1998. p. 289-314.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]