CEOT Variants or Entities: Time for a Rethink? A Case Series with Review of the Literature

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.

Calcifying epithelial odontogenic tumour series with unique clinical and histopathological features

“…Approximately 388 cases of CEOT have been reported in English literature till the present time. 6 For such a rare entity, our case series of six cases during a 20 year period can be a fair addition to the accumulating knowledge about CEOT. This study has tried to correlate the clinical, radiological and histopathological features of this unique tumour amid a vast number of pathological mimics.…”

Comparative Analysis Between Dentinogenic Ghost Cell Tumor and Ghost Cell Odontogenic Carcinoma: A Systematic Review

“…Spindle, basaloid, and epidermoid differentiation patterns have also been reported in ameloblastoma and ameloblastic carcinoma, a spectrum of lesions with epithelium parenchymal similarities to GCONs [8,9,[40][41][42][43]. Scattered or abundant clear cells were also reported in ameloblastomas and calcifying epithelial odontogenic tumors [41,44]. Lesions with a mixture of ameloblastomatous epithelium with an adenoidlike component have been previously termed as adenoid ameloblastoma [45,46], with some tumors presenting ghost cells and/or dentinoid material [45][46][47][48][49].…”

Non-calcifying Langerhans Cell Rich Variant of Calcifying Epithelial Odontogenic Tumor and Amyloid Rich Variant of Central Odontogenic Fibroma: A Unique Entity or a Spectrum?

“…The diagnosis of COF, a mesenchymal tumor, seems not to be representative of the histopathological features of the disease possessing the proliferating odontogenic epithelial cells. This controversy in diagnosis has also been reported [24] and encountered even after Dr. Pindborg had defined CEOT and still included one NCLC-CEOT case in his review article describing various CEOT histopathologic patterns [5]. It is also possible that NCLC-CEOT/AR-COF is in the COF/CEOT spectrum and has a unique clinical presentation due to its specific tumor location (Figure 6).…”

“…All the cases from case reports or case series named NCLC-CEOT [9,[16][17][18][19][20] or AR-COF [13-15, 21, 22] were reviewed. The cases included in review articles focusing on NCLC-CEOT or AR-COF and review articles including information about the NCLC-CEOT or AR-COF variants were also reviewed [22][23][24][26][27][28][29]. The inclusion criteria were the case reports and case series with convincing histopathological photographs for confirming the diagnosis (reviewed by two board-certified oral pathologists) and containing detailed clinical information for further comparisons.…”

“…The inclusion criteria were the case reports and case series with convincing histopathological photographs for confirming the diagnosis (reviewed by two board-certified oral pathologists) and containing detailed clinical information for further comparisons. The exclusion criteria were the cases without convincing histopathological photographs; peripheral (extraosseous) CEOT or COF; and those that lack detailed clinical information for further comparison [24,[26][27][28][29][30]. The duplicated cases were also excluded.…”