Complex Odontoma in Both the Jaws: A Rare Case Report

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1 /jp-journals Adit Srivastava et al CASE REPORT Complex Odontoma in Both the Jaws: A Rare Case Report Adit Srivastava, AG Annaji, Sanjay B Nyamati, Govind Singh, GC Shivakumar, S Sahana ABSTRACT Odontomas are the most common odontogenic tumors. They are usually asymptomatic and are often discovered during routine radiography. Complex odontomas appear as irregular calcified masses that bear no similarity to teeth. We report a rare case of complex odontoma in maxilla and mandible in an adult patient with clinical, radiological, histological features and treated surgically, with its review of the literature. Keywords: Complex, Computed tomography, Maxilla/Mandible, Odontoma, Radiopaque. How to cite this article: Srivastava A, Annaji AG, Nyamati SB, Singh G, Shivakumar GC, Sahana S. Complex Odontoma in Both the Jaws: A Rare Case Report. J Orofac Res 2012;2(1): Source of support: Nil Conflict of interest: None declared INTRODUCTION Paul Broca was the first person to use the term odontoma in He defined the term odontoma as tumors formed by the overgrowth of transitory or complete dental tissues. 1 Odontomas are slow-growing asymptomatic neoplasms represent 22% of the odontogenic tumors found in jaws. 2 They develop from epithelial and mesenchymal components of the dental apparatus, producing enamel and dentin. 3 They can occur anywhere in the mandible or maxilla without apparent predilection for age or sex, although they usually are found in conjunction with primary teeth. Odontomas frequently can inhibit the eruption of adjacent teeth. 4 Radiologically odontomas are classified as complex, compound and cystic. Complex odontomas are less common than the compound variety in the ratio 1:2. 5 Complex odontomas in turn are found in the posterior mandibular sector, overimpacted teeth, and can reach several centimeters in size. These lesions manifest as a radiopaque solid mass with occasional nodular elements, and surrounded by a fine radiolucent rim. The lesions are unilocular and are separated from the normal bone by a well-defined corticalization line. No individual tooth-like structures are seen. 6 A complex odontoma presents as an amorphous conglomeration of dental tissues consisting of enamel, dentin, cementum, pulp and enamel organ. 7 The treatment of choice is surgical removal of the lesion, followed by histopathological study to confirm the diagnosis. We present an interesting and a rare case of complex odontoma in maxilla and mandible of an adult patient. 56 CASE REPORT A 41-year-old woman reported to department of oral medicine and radiology with a complaint of swelling in the upper left and lower right back jaw region since 4 years. She gave a history of swelling in the lower left jaw for which she had undergone surgery 6 years back. Intraoral examination revealed that two hard swellings were present in the upper left posterior quadrant (Fig. 1A) extending from 23 to 27, and other one in the lower right posterior quadrant (Fig. 1B) from 42 to 47. Both the swellings were measuring approximately 3 5 cm, with bicortical expansion with displacement of adjacent tooth. It was bony hard in consistency with intact overlying mucosa. Spacing was present between 25 and 26, and 45 and 46. The mandibular left quadrant was missing. Fig. 1A: Intraoral photograph of maxillary left quadrant Fig. 1B: Intraoral photograph of mandibular right quadrant

2 JOFR Complex Odontoma in Both the Jaws: A Rare Case Report Intraoral periapical radiograph revealed a well-defined radiopaque mass in between the roots of 25 to 27, (Fig. 2A) and an ill-defined radiopaque mass in between 45 and 46. (Fig. 2B) Cross-sectional mandibular occlusal radiograph showed bicortical expansion (Fig. 3), panoramic radiograph (Fig. 4) showed the radiopaque mass surrounded by thin radiolucent rim extending from 25 to 26, with pushing the floor of the maxillary sinus, and also radiopaque mass surrounded by thin radiolucent rim extending from 45 to 46, with inclination and tilting of adjacent teeth without causing any destruction of mandibular canal, and absence of left mandible from symphysis to subcondylar region. Axial view (Fig. 5A) of computed tomography (CT) of the jaws showed expansile hyperdense mass in the maxillary left premolar region. The coronal view (Fig. 5B) of CT showed an expansile calcified mass in the mandibular premolar region, and in the maxillary left region showed the lesion was not involving the maxillary sinus. Considering Fig. 3: Mandibular occlusal radiograph Fig. 4: Orthopantomograph Fig. 2A: Intraoral periapical radiograph of maxillary left quadrant the clinical and radiologic presentations, a diagnosis of complex odontoma was determined. Differential diagnosis of ameloblastic fibro-odontoma and other fibro-osseous lesions were considered. The patient underwent surgery with general anesthesia. A mucoperiosteal flap was opened; the tumor was enucleated along with the extraction of involved teeth 24, 25, 26 and 45, 46. The areas were curetted well and lining was removed. The cavity was applied with carnoy s solution and packed with gauge, the flap was sutured in place. The specimen (Fig. 6) was sent to histopathological examination (Fig. 7) which revealed mostly irregular dentin, cementum, enamel and spaces containing loose fibrous connective tissue, which confirmed the diagnosis of complex odontoma. DISCUSSION Fig. 2B: Intraoral periapical radiograph of mandibular right quadrant Odontoma is the most common type of odontogenic tumor, although some authors prefer to refer to it as hamartoma, not a true tumor. 5 Complex odontomas tend to occur in the posterior region of the jaw and compound odontomas are more common in the anterior maxilla. 8,9 The main unique feature of our case is that the complex odontoma is in both Journal of Orofacial Research, January-March 2012;2(1):

3 Adit Srivastava et al maxilla and mandible in the premolar region. They may be discovered at any age, although less than 10% are found in patients over 40 years of age, our patient was 41 years. Although they are commonly asymptomatic, clinical indicators of odontoma may include retention of deciduous teeth, noneruption of permanent teeth, pain, expansion of the cortical bone and tooth displacement. In the present case, there was bicortical expansion and displacement of teeth. Other symptoms include anesthesia in the lower lip and swelling in the affected area. According to WHO classification, odontomas can be divided into three groups are as follows: Complex odontoma: When the calcified dental tissues are simply arranged in an irregular mass bearing no morphologic similarity to rudimentary teeth. 2. Compound odontoma: Composed of all odontogenic tissue in an orderly pattern that results in many teeth like structures, but without morphological resemblance of normal teeth. 3. Ameloblastic fibro-odontoma: Consists of varying amount of calcified dental tissues and dental papilla like tissues, that later component resembling an ameloblastic fibroma. The ameloblastic fibro-odontoma is considered as an immature precussor of complex odontoma. Clinically, odontomas are either complex or compound, and are classified as follows: 11 Intraosseous: These odontomas occur inside the bone and may erupt (erupted odontoma) into the oral cavity. Extraosseous or peripheral: Odontomas occurring in the soft tissue covering the tooth-bearing portions of the jaws. The odontoma presents as a well-defined radiopacity situated in bone, but with a density that is greater than bone and equal to or greater than that of a tooth. It contains foci of variable density. A radiolucent halo, typically surrounded by a thin sclerotic line, surrounds the radiopacity. The radiolucent zone is the connective tissue capsule of a normal tooth follicle. The thin sclerotic line resembles the corticated border seen in a normal tooth crypt. The developmental stages can be identified based on radiologic features and the degree of calcification of the lesion at the time of diagnosis. 11,12 The first stage is characterized by radiolucency due to the absence of dental tissue calcification, the second or intermediate stage shows partial calcification and the third or classically radiopaque stage exhibits predominant tissue calcification with the surrounding radiolucent halo. 13,14 In our case, we present a complex odontoma, which should be differentiated from cementoblastoma, osteoid osteoma and fibro-osseous lesions, such as cemento-ossifying fibroma. Additional radiographic evaluation with CT was necessary to determine the extension and features of the lesion because the two-dimensional limitations of periapical and panoramic images did not allow complete visualization of the maxillofacial complex. In this regard, CT images are necessary not only for evaluation of the lesion itself, but also for localization of associated pathoses and proper treatment planning. 15 In our case, CT images allowed better depiction of the hyperdense mass nearing the floor of the maxillary sinus. These features, based on CT images, were critical for the therapeutic approach, allowing perfect planning for the surgical procedure and elucidation of diagnosis. In cases when obstruction of sinus drainage is evident, one should be completely aware of serious 58 Fig. 5A: Axial view of computed tomography Fig. 5B: Coronal view of computed tomography

4 JOFR Complex Odontoma in Both the Jaws: A Rare Case Report CONCLUSION As for our knowledge there is no such reported case of complex odontoma occurring in both the jaws in an adult patient. Since our patient is from a remote place, there were no previous records of the hemimandibulectomy of the left side, we assume that it could have been any cyst or a tumor. CT is the gold standard, especially for ruling out any suspicion of associated paranasal sinus, orbital involvement, and revealing the spread and intracranial extension of cysts and tumors of the oral cavity. Fig. 6: Surgical specimen Fig. 7: Histological photograph complications, such as orbital infections, epidural and subdural empyema, meningitis, cerebritis, cavernous sinus thrombosis, brain abscess and death. 16 Odontomas have been associated with trauma during primary dentition as well as with inflammatory and infectious processes, hereditary anomalies (Gardner syndrome, Hermann s syndrome), odontoblastic hyperactivity and alterations in the genetic components responsible for controlling dental development. 17 According to Hitchin, 18 he suggested that odontomas are inherited through a mutant gene or interference, possibly postnatal, with genetic control of tooth development. The recommended treatment of odontoma is through conservative approach. 10 Since, enucleation and curettage of odontomas are curative, chances of recurrence are less. If any portion of the lesion is left unexcised such residual odontomas may remain unchanged throughout. Very rarely the wound may get infected after an incomplete removal, since the avascular odontoma portion acts like foreign body. 19 REFERENCES 1. Shafer, Hue, Levy. Cysts and tumors of the Jaws. In a textbook of oral pathology (4th ed). WB Saunders Company, p Bhaskar SN, Oral pathology in the dental office: Survey of 20,575 biopsy specimens. JADA 1968;76: Shafer WG, Hine MH, Levy BM. A textbook of oral pathology, (3rd ed). Philadelphia, WB Saunders Co 1974: Baden E. Odontogenic tumors. Pathology Annual 1971;6: Cohen DM, Bhattacharyya I. Ameloblastic fibroma, ameloblastic fibroodontoma, and odontoma. Oral Maxillofac Surg Clin North Am 2004;16(3): Philipsen H, Reichart P, Praetorius F. Mixed odontogenic tumours and odontomas. Considerations on interrelationship. Review of the literature and presentation of 134 new cases of odontomas. Oral Oncol 1997;32: Reichart PA, Philipsen HP. Compound odontoma, in odontogenic tumors and lesions. Quintessence 2004: Neville BW, Damm DD, Allen CM, Bouquot JF. Odontogenic cysts and tumors. In: Oral and maxillofacial pathology (2nd ed). Philadelphia (PA): WB Saunders 2002: Mupparapu M, Singer SR, Rinaggio J. Complex odontoma of unusual size involving the maxillary sinus: Report of a case and review of CT and histopathologic features. Quintessence Int 2004;35(8): Kramer IRH, Pindborg JJ, Shear M. Histological typing of odontogenic tumours (2nd ed). Berlin: Springer-Verlag 1992: Junquera L, de Vincente JC, Roig P, Olay S, Rodriguez-Recio O. Intraosseous odontoma erupted into the oral cavity: An unusual pathology. Med Oral Patol Oral Cir Bucal 2005;10(3): Worth HM, Odontomas and cysts of the jaws. In: Principles and practice of oral radiographic interpretation. Chicago: Year Book Medical 1963: Wood NK, Goaz PW, Lehnert J. Mixed radiolucent radiopaque lesions associated with teeth. In: Wood NK, Goaz PW (Eds). Differential diagnosis of oral and maxillofacial lesions. Singapore: Harcourt Brace and Company Asia Pte Ltd 1998: Guinta JL, Kaplan MA. Peripheral soft tissue odontomas. Oral Surg Oral Med Oral Pathol 1990;69(3): 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: Kim IK, Kim JR, Jang KS, Moon YS, Park SW. Orbital abscess from an odontogenic infection. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103: Ragalli CC, Ferreria JL, Blasco F. Large erupting complex odontoma. Int J Oral Maxillofac Surg 2000;29(5): Journal of Orofacial Research, January-March 2012;2(1):

5 Adit Srivastava et al 18. Hitchin AD. The aetiology of the calcified composite odontomas. Brit Dent J 1971;30(11): Marx RE, Stern D. Odontogenic tumor. Oral and maxillofacial pathology. Quintessence 2003;14: ABOUT THE AUTHORS Adit Srivastava (Corresponding Author) Assistant Professor, Department of Oral Medicine and Radiology Faculty of Dental Sciences, IMS, Banaras Hindu University Varanasi , Uttar Pradesh, India, Phone: , Fax: , dr.adit69@gmail.com; shiva21375@yahoo.in AG Annaji Reader, Department of Oral Medicine and Radiology, VS Dental College, Bengaluru, Karnataka, India Sanjay B Nyamati Professor and Head, Department of Oral Medicine and Radiology Triveni College of Dental Sciences, Bilaspur, Chhattisgarh, India Govind Singh Senior Lecturer, Department of Oral Medicine and Radiology, Sardar Patel Post Graduate Institute of Dental and Medical Sciences Lucknow, Uttar Pradesh, India GC Shivakumar Associate Professor, Department of Oral Medicine and Radiology People s Dental Academy, Bhopal, Madhya Pradesh, India S Sahana Reader, Department of Public Health Dentistry, People s College of Dental Sciences and Research Centre, Bhopal, Madhya Pradesh India 60