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Non-Calcifying/Langerhans Cell-Rich Calcifying Epithelial Odontogenic Tumour: A Critical Review of the Rare and Distinctive Entity

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Abstract

Background

The study aims to analyse the non-calcifying/Langerhans cell rich (NCLC) subtype of calcifying epithelial odontogenic tumour (CEOT). 

Method

The features of cases of the NCLC subtype of CEOT noted in the English literature by PubMed as well as 3 new cases were reviewed.

Results

Overall, twenty-one cases were noted. Many were women in the fourth to sixth decades (male-to-female ratio =1 to 2). Radiologically, the lesion is often unilocular with resorption of the affected teeth. Nineteen of the 21 cases occurred in the maxilla, especially the anterior portion. On pathological examination, epithelial cells are noted in non-calcifying amyloid-rich fibrous stroma. The main differential diagnosis is the amyloid subtype of central odontogenic fibroma. Immunohistochemical studies revealed the tumour epithelial cells were positive for cytokeratins and p63 and contained CD1a, S-100, and langerin-positive Langerhans cells. On a median follow-up of 2 years, one patient had a recurrence one year after curettage.

Conclusion

The NCLC subtype of CEOT is unique as it contains significant numbers of Langerhans cells and has clinicopathological features distinctive from classic CEOT.

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Data Availability

All the data available in the study are in the paper.

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Acknowledgements

The authors would like to thank the staff in the Department of Oral Pathology, Hospital of Stomatology, Sun Yat-sen University for preparing the histology sections.

Funding

No funding was obtained for this study.

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Author notes

  1. Nan Xie and Zhipei Chen contributed equally to this paper.

Authors and Affiliations

  1. Department of Oral Pathology, Hospital of Stomatology, Sun Yat-Sen University, Guangzhou, Guangdong, China

    Nan Xie, Zhipei Chen, Jingbin Huo & Xiaohua Chen

  2. Department of Radiology, Hospital of Stomatology, Sun Yat-Sen University, Guangzhou, Guangdong, China

    Yi Liu

  3. School of Medicine and Dentistry and Menzies Health Institute Queensland, Gold Coast, QLD, 4222, Australia

    Alfred King-yin Lam

  4. Pathology Queensland, Gold Coast University Hospital, Southport, QLD, 4215, Australia

    Alfred King-yin Lam

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  1. Nan Xie
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Contributions

N.X. wrote the main script text Y.L, J.H, X.C contributes to figures A.L. concept and edit the main manuscript. All authors reviewed the manuscript

Corresponding author

Correspondence to Alfred King-yin Lam.

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This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. The authors declare that they have no conflict of interest.

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Xie, N., Chen, Z., Liu, Y. et al. Non-Calcifying/Langerhans Cell-Rich Calcifying Epithelial Odontogenic Tumour: A Critical Review of the Rare and Distinctive Entity. Head and Neck Pathol 17, 1011–1020 (2023). https://doi.org/10.1007/s12105-023-01602-5

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