|Year : 2016 | Volume
| Issue : 2 | Page : 171-174
Odontoameloblastoma: A rare case with unusual presentation
Supreet Jain1, Mathod C Shashikanth2, Priyanka Agrawal3, Rahul Singh4
1 Department of Oral Medicine and Radiology, New Horizon Dental College and Research Institute, Bilaspur, Chhattisgarh, India
2 Department of Oral Medicine and Radiology, Rungta College of Dental Sciences and Research, Bhilai, Chhattisgarh, India
3 Private Practice, Raipur, Chhattisgarh, India
4 Department of Orthodontics and Dentofacial Orthopedics, New Horizon Dental College and Research Institute, Bilaspur, Chhattisgarh, India
|Date of Submission||19-Nov-2015|
|Date of Acceptance||21-Nov-2016|
|Date of Web Publication||02-Dec-2016|
Jain Handloom, Jain Furniture, Telipara, Near Bus Stand, Bilaspur - 495 001, Chhattisgarh
Source of Support: None, Conflict of Interest: None
| Abstract|| |
The odontoameloblastoma (OA), also known as ameloblastic odontoma, is a very rare odontogenic tumor that is similar to ameloblastoma in its locally aggressive behavior. OA includes odontogenic ectomesenchyme in addition to odontogenic epithelium that resembles an ameloblastoma both in structure and in behavior. Its clinical presentation, however, often mimics the more innocuous odontoma, and hence, the recognition of its aggressive nature is commonly only ascertained after its histopathologic diagnosis following enucleation. This paper presents a rare case of OA with unusual clinical and radiological features.
Keywords: Ameloblastic odontoma, odontoameloblastoma, odontogenic tumor
|How to cite this article:|
Jain S, Shashikanth MC, Agrawal P, Singh R. Odontoameloblastoma: A rare case with unusual presentation. J Indian Acad Oral Med Radiol 2016;28:171-4
|How to cite this URL:|
Jain S, Shashikanth MC, Agrawal P, Singh R. Odontoameloblastoma: A rare case with unusual presentation. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2020 Nov 23];28:171-4. Available from: https://www.jiaomr.in/text.asp?2016/28/2/171/195133
| Introduction|| |
Odontoameloblastoma (OA) is an extremely rare mixed odontogenic tumor characterized by the simultaneous occurrence of an ameloblastoma and composite odontoma. , It is also known as ameloblastic odontoma.  There is proliferation of tissue of the odontogenic apparatus in an unrestrained pattern, including complete morphodifferentiation, as well as apposition and even calcification.  According to the World Health Organization (WHO), it is a neoplasm that includes odontogenic ectomesenchyme in addition to odontogenic epithelium that resembles an ameloblastoma in both structure and behavior. Because of the presence of odontogenic ectomesenchyme, inductive changes take place leading to the formation of dentin and enamel in parts of tumor. , The term OA was included in the 1971 WHO histological classification of odontogenic tumors.  The first case of OA was described by Thoma in 1944, and to date less than 50 cases of OA have been reported in the literature. , We present a rare case of OA involving the anterior mandible mimicking a complex odontoma and a brief review of the related literature.
| Case Report|| |
A 13-year-old male patient reported to the Department of Oral Medicine and Radiology, with a chief complaint of irregularly placed teeth in the lower front teeth region. Patient gave no relevant medical, dental, and family history. Intraoral examination revealed crowding of mandibular anteriors [Figure 1]. There was no swelling or vestibular obliteration. Panoramic radiograph revealed radiopaque lesion between 32 and 33 extending from alveolar crest to apex of 33 of approximately 5 × 8 mm in measurement, round to oval in shape, scalloped margin and surrounded by a radiolucent rim. Internal structure appeared to be radiopaque. There was distal tilting of the root tip of 33 and mesial tilting of 32 [Figure 2].
|Figure 2: Panoramic image showing radiopaque mass between the roots of 32 and 33|
Click here to view
On the basis of radiological examination, diagnosis made was complex odontome and differential diagnosis of focal cemento-osseous dysplasia, ossifying fibroma, calcifying epithelial odontogenic tumor was given. Routine hematological examinations were carried out, and the values were within normal range. Tumor was excised under local anesthesia with all aspectic measures, and the specimen was sent for histopathological examination [Figure 3]a and b.
Hematoxylin and eosin (H and E) stain of biopsy specimen showed fibrocellular stroma with few cell layer-thick epithelium lined by ameloblast-like cells and stellate reticulum [Figure 4]. Decalcified section of the biopsy specimen showed irregular dentinal tubules, few areas of calcification and primitive connective tissue stroma [Figure 5]. On the basis of histopathological findings, final diagnosis of odontoameloblastoma was made. Postoperative follow-up after 6 months showed uneventful healing [Figure 6]a and b.
|Figure 4: Hematoxylin and eosin stain of biopsy specimen showing fibrocellular stroma with few cell layer-thick epithelium lined by ameloblasts-like cell and stellate reticulum |
Click here to view
|Figure 5: Decalcified section of biopsy specimen showed irregular dentinal tubules, few areas of calcification, and primitive connective tissue stroma|
Click here to view
|Figure 6: (a) Postoperative intraoral picture after 6 month follow-up (b) postoperative panoramic view after 6 month follow-up|
Click here to view
| Discussion|| |
OA or ameloblastic odontoma has generated significant controversy and confusion in literature, as evidenced by the reporting of ameloblastic fibroodontomas, developing odontoma, and OAs under the term ameloblastic odontoma.  It is an aggressive odontogenic neoplasm having an incidence of 0.5%.  OA occurs between 6 months to 40 years of age, predominantly in young patients, appearing under 15 years of age and has a predilection for males. ,,
No studies have been reported establishing the pathogenesis of this neoplasm. Nevertheless, some theoretical explanations have been presented.  Thompson et al. suggested that the proliferation of epithelium could induce the mesenchymal tissue to form hamartomatous mineralized dental tissue.  Mosqueda-Taylor et al. suggested that the ameloblastoma and odontoma can develop separately and then collide. 
Clinically, OA is a slow growing painless mass that expands the alveolus and vestibular cortex, courses with a delayed or absence of permanent teeth eruption, and disturbances in occlusion. Mandible is more involved than the maxilla with predilection for molar-premolar region. , In the present case, there were no clinical signs and symptoms seen. There was only crowding in the lower anteriors. The pathology was diagnosed radiologically, which was involving mandibular anterior region. Various authors have reported the lesion to be associated with swelling. ,,,,
Radiographically, the lesion shows central destruction of the bone, expansion of cortical plates, and unilocular or multilocular radiolucent area containing dense radioopacities either as solid masses or as smaller particles, which may or may not bear full resemblance to tooth form. In some instances, a single irregular radioopaque mass of calcified tissue is present which may be confused with composite odontoma. It commonly exhibits a well-defined margin, displacing the surrounding erupted teeth rather than producing root resorption. ,, Mosca et al. also reported similar radiographic appearance with cortical plate expansion, which was not present in our case. 
In the presented case, pathology was radiopaque with lobulated margin and surrounded by radiolucent rim giving resemblance to composite odontoma. There was distal tilting of the root tip of 33 and mesial tilting of 32 with no resorption. A differential diagnosis of compound or complex odontomas, osteoma, osteosclerosis, condensing osteitis, cementoma, ossifying fibroma, cementoossifying fibroma and ameloblastic fibroodontoma must be considered. 
Histopathology of OA consists of cells and tissues in a complex distribution, including columnar, squamous, undifferentiated epithelial cells, as well as ameloblasts, enamel and enamel matrix, dentin, osteodentin, dentinoid, osteoid material, stellate reticulum-like tissues, dental papilla, bone, cementum, as well as stromal connective tissue. Many structures resembling normal or atypical tooth germ may be found, with or without the presence of calcified dental tissues. There are sheets of typical ameloblastomas of one or another recognized type, usually basal cell, follicular, or plexiform. Few mitotic figures are present. ,
Because of their rarity, controversy exists in the treatment of OA. The effective methods of treatment are en-bloc resection or complete resection of the affected part of bone irrespective of the size of the lesion.  OA has potential for recurrence.
| Conclusion|| |
OA is a very rare entity and only few cases have been reported. It has similarity to other odontogenic lesions, and therefore a preoperative diagnosis is difficult to achieve based only on the clinical and radiographic features of the lesion. Oral radiologists should be aware of the existence of these odontogenic tumors to properly diagnose the lesion. Proper treatment and follow-up of patients must be done to prevent further complications.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Rajendran R, Sivapathasundaram B, editors. Shafer's Textbook of Oral Pathology. 5 th
ed. New Delhi: Elsevier; 2006.
Neville BW, Damn DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 2 nd
ed. Philadelphia: Saunders; 2002.
Siniscalchi EN, Catalfamo L, Rinaldo F, Ieni A, Tuccari G, De Ponte FS. Odontoameloblastoma: Five years follow up of a surgical case and review of literature. Eur Rev Med Pharmacol Sci 2012;16:125-8.
Kumar K, George GB, Padiyath S, Rupak S. Odontoameloblastoma: A diagnostic dilemma for maxillofacial radiologist. Int J Odontostomat 2013;7:203-6.
White SC, Pharoah MJ. Oral radiology: Principles and interpretation. 6 th
ed. St. Louis: Mosby Elsevier; 2009.
Mosca RC, Marques MM, Barbosa SC, Marcucci, Oliveira JX, Lascala CA. Odontoameloblastoma: Report of two cases. Indian J Dent Res 2009;20:230-4.
Sapru BBL, Dasgupta MD, Rajaram CT. Odontoameloblastoma: A rare odontogenic tumor. Med J Armed Forces India 2001;57:333-4.
Dive A, Khandekar S, Bodhade A, Dhobley A. Odontoameloblastoma. J Oral Maxillofac Pathol 2011;15:60-4.
Martín-Granizo-López R, López-García-Asenjo J, De Pedromarina M, Domínguez-Cuadrado L. Odontoameloblastoma: A case report and a review of the literature. Med Oral 2004;9:340-4.
Thompson IO, Phillips VM, Ferreira R, Housego TG. Odontoameloblastoma: A case report. Br J Oral Maxillofac Surg 1990;28:347-9.
Mosqueda-Taylor A, Carlos-Bregni R, Ramirez-Amador V, Palma-Guzman JM, Esquivel-Bonilla D, Hernandez-Rojas LA. Odontoameloblastoma: Clinico-pathologic study of three cases and critical review of the literature. Oral Oncol 2002;38:800-5.
Wood NK, Goaz PW. Differential diagnosis of oral and maxillofacial lesions. 5 th
ed. St. Louis: Mosby Elsevier; 1997.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]