Odontoameloblastoma: Case Report of a Rare Tumor

“Odontoameloblastoma: case report of a rare tumor”

Ahrens A. GSM*, Bressi T.**

* D.D. Studio Dentistico Dr. Ahrens Alessandro, Nocciano (Pe) – Italy,

** M.D. ENT Department, University hospital, Perugia, Italy

Abstract

Odontoameloblastoma is a rare odontogenic tumor, characterised by the simultaneous occurrence of an ameloblastoma and a compound or complex odontoma in the same tumoral mass(epithelial and mesenchymal cells), which rises up usually in the bone of maxilla. The WHO introduced the term of Odontoameloblastoma in 1971 and actually less then 30 cases are described in literature.This tumor has a male to female ratio 1.8:1 and usually occurs in the second decade.

We report the case of a 16 years old female.

Introduction

Odontoameloblastoma is a very rare tumor with epithelial and mesenchimal components, usually involving the bone of maxilla and mimicking an odontoma. Due to this mimicking it requires a histological analysis to set up a diagnosis. AS described in 1944 by Thoma ed al. (1) the odontoameloblastoma is an ameloblastoma with evident signs of focal differentiation into an odontoma.

Case Report

In 2001 a 16 years old female come to our attention for the compliant of a lack of eruption of the 4.5 and 4.6 and for a gingival painful swelling.. The medical history of the patient was negative for other diseases.

The oral examination showed a good oral hygiene, absence of decays or periodontal disease, except a reddish area in the region of 4.5 and 4.6, and a low grade displacement of 4.7.

Was also reported the absence of 4.5 and 4.6 and an enlargement of the cortex.

To better evaluate the oral structures we required at first a panoramic rx that revealed a well defined of radiolucency with a teeth-similar and radiopaque structures inside.

To better appreciate this radiolucent area we proceeded with a periapical rx that showed an intraosseus lesion of about 2 cm, non involving the Inferior Alveolar Nerve, with a well defined margin and some radiolucid and radiopaque structures inside.

We also reported a displacement of the roots of 4.7 without signs of resorption.

After the evaluation of the case we decided to go on with the surgical approach for the complete removal of the lesion.

Surgery time for the enucleation started with a vestibular fullthickness envelope extended from 4.4 to 4.7 to identify and isolate the mental nerve and the lesion. The lesion was easily identified after the perforation of the cortex.The second step was the removal of the lesion and the curettage of the bone cavity and the filling with deprotheinized bovine bone and the remodellation of the area interested by the lesion.

The envelope was sewed with a 4-0 sintetic suture ( Poliglicolic acid covered with policaprolate - co-glicolide) and the patient was administered with antibiotics (claritromicin RM) 500 mg once a day for 5 days. To preserve the second molar (4.7) from mobility and also to save the post-extractive space we inserted a space manteiner.

The specimen underwent histologic analysis that revealed a picture of odontoma with areas of ameloblastic cells. The microscopical report evidenced odontogenic epithelial cells proliferating in nests and strands. The stroma around these nests showed fibromyxoiod areas with cementum surrounded by ameloblastic cells.

Due to the risk of recurrence we started a periodical follow-up control which included clinical examination and TC for three years. Actually there is non evidence of recurrence.

Discussion

The polymorphic histologic aspect of odontogenic tumors makes it difficult to set a 100% diagnosis. Many authors( 2-18) tried to described exactly this kind of tumor and also to define it.

What has been actually defined is that odontoameloblastoma rises up as a slow-growing painless mass, expanding the vestibular cortex, associated to the absence of eruption of the permanent teeth in the involved area.

The radiologic picture is characterised by a multilocular radiolucency with raiopaque areas related to dental tissues. It presents a well defined margin and shows displacing of surrounding teeth.

Histologically the odontoamelobastoma shows areas of odontogenic epithelium surrounded by mesenchymal components like enamel, dentine and cementum forming structures.

It is important to set a differential diagnosis with ameloblastic fibro-odontoma, with che CEOT ( calcifying epithelial odontogenic tumor) and odontoma.

The ameloblastic fibro-odontoma usually affects adult subjects with a slow-growing mass without bone infiltration.

The CEOT is a benign but locally aggressive neoplasm. It’s origins are actually uncertain because some authors decribe it as arising from reduced enamel epithelium of an associated unerupted tooth; by the other side other authors describe it as arising from the stratum intermedium of the tooth bud. Commonly occurs in the molar region of the mandibula and with lower frequency in premolar and anterior region. Most frequently that patients are grouped in the middle age but there is a wide range of case from 8 to 92 years, without prevalence of sex.

In about the 50% of cases the lesion is associated with a unerupted or impacted tooth.

Histologically the CEOT presents islands of eosinophilic epithelial cells infiltrating the bony trabeculae. The cells present nuclear hypercromatism and pleomorphism. Another typical aspect of CEOT is the presence of liesegang rings (concentric spherical calcifications).

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