Abstract

The first detailed description of calcifying epithelial odontogenic tumor (CEOT) are ascribed to Jens Pindborg, but this tumor was described some years previously. Subsequently, CEOT was included in the 1971 WHO classification of odontogenic tumors and a since then number of variants have been described, which have added confusion to the diagnostic criteria. We aimed to survey the literature on the variants of CEOT, in parallel with a review of our single institution experience of CEOTs. Cases identified were collated, including available clinical, radiological and histological information and then reviewed, taking into account changes in the understanding and classifications of odontogenic tumors since initial diagnosis. We identified 26 cases from 1975 to 2017 for which histological material was available. Of these, only 13 (50%) showed the “classic” histological appearance, whilst two cases were identified as recognized variants. In 11 cases, other diagnoses or a differential diagnosis were preferred, with no agreed diagnosis in four of these. The proliferation fraction (Ki67) in the 10 cases tested was 2.1% ± 0.18. These findings illustrate the diagnostic challenges in this group of tumors and highlight the gaps in knowledge. Techniques, such as EWSR1 gene cytogenetic analysis, may be helpful in cases with clear cells. However, in other areas of controversy, including the non-calcifying and Langerhans cell rich variants, further investigation, perhaps utilizing sequencing technologies may be needed to refine the classification. Owing to the relative rarity of these lesions it would be beneficial if future work could be pursued as an international collaboration.

Highlights

  • Introduction and Review of the LiteratureJens Pindborg described the calcifying epithelial odontogenic tumor (CEOT), a rare epithelial odontogenic tumor, in detail in 1958 [1]

  • We aimed to review all of our diagnoses of CEOT in the diagnostic archive and review them in light of the three WHO classifications published during this time (1991, 2005 and 2017) and the current literature on this entity

  • Thirty two cases had been coded as CEOT in the diagnostic database from 1975 to 2017

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Summary

Introduction

Jens Pindborg described the calcifying epithelial odontogenic tumor (CEOT), a rare epithelial odontogenic tumor, in detail in 1958 [1]. The number of cases has continued to increase and, to date, more than 362 cases have been reported [10]. According to this recent review of published cases, there was an almost equal distribution among males and females and the peak age of occurrence of central lesions was in the 3rd and 4th decades, similar to that presented in our recent series of odontogenic tumors [11]. Most presentations are intraosseous but in 1966, Pindborg described an extra-osseous/ peripheral CEOT [12]

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