Bilateral adenomatoid odontogenic tumour of the maxilla in a 2-year-old female—the report of a rare case and review of the literature

Abstract

Multifocal odontogenic lesions are uncommon and have only been observed in conditions associated with known genetic mutations. To the best of our knowledge, only two cases of multifocal adenomatoid odontogenic tumours (AOT) have previously been reported in the literature. In this study, we report the first case of a bilateral, separate AOT in the maxilla in the midline in a 2-year-old female. The patient presented with bilateral expansile masses in the maxilla on either side of the midline which had been present for 6 months. She was asymptomatic and had occasional difficulty in breathing. The tumour was diagnosed as AOT and was surgically enucleated along with the associated teeth. The patient recovered well and has been on recall for 5 years. The follow-up panoramic radiograph made a fortnight ago revealed evidence of three new radio-opaque lesions with an associated tooth in the region of the anterior mandible, the premolar region of the right maxilla and the molar region of the left maxilla. To acquire additional information about AOT, all reports regarding AOT cited in 'PubMed' from 1995 onward were reviewed and the incidence, clinical features, radiographic features and management of AOT are discussed in this study.

Adenomatoid odontogenic tumor: An updated analysis of the cases reported in the literature

“…There are some cases described in the literature as multiple AOTs . These cases are extremely rare and can be associated with the Schimmelpenning syndrome .…”

An Unusual Case of Extra Follicular Adenomatoid Odontogenic Tumor: A Case Report.

“…The pathogenesis of AOT is not clear and a number of hypotheses have been proposed. Theoretically, it is said to arise from the enamel organ, the epithelial lining of the dentigerous cyst, epithelial rests of Malassez of the deciduous or permanent tooth, or remnants of the dental lamina and may show an ameloblastic phenotype 4 . However, Philipsen et al also argued that all AOT variants show identical histological features and therefore it points towards a common origin from the dental lamina or its remains.…”

Case reports of a typical extrafollicular adenomatoid odontogenic tumor of maxilla

“…Displacement of the teeth associated with tumor is more common. 12 Radiographically AOT presents as a well demarcated, unilocular radiolucency that generally exhibits a smooth or corticated border. Most lesions are around 1 to 3 cm in greatest diameter and associated with impacted tooth.…”