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Published in: Diagnostic Pathology 1/2014

Open Access 01-12-2014 | Research

A clinicopathologic study on calcifying epithelial odontogenic tumor: with special reference to Langerhans cell variant

Authors: Yan Chen, Ting-Ting Wang, Yan Gao, Tie-Jun Li

Published in: Diagnostic Pathology | Issue 1/2014

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Abstract

Background

Calcifying epithelial odontogenic tumour (CEOT) is a rare benign odontogenic tumour, and its Langerhans cell variant is even rarer. Due to the limited number of recorded cases, the biological behaviour and histogenesis of the Langerhans cell variant of CEOT are not yet fully understood. Thus, the correlation between conventional CEOT and the Langerhans cell variant remains to be clarified.

Material (cases)

Eight cases of CEOT including 2 cases of Langerhans cell variant were clinicopathologically studied and the English language literature was reviewed. Langerhans cells were detected in 2 cases of conventional CEOT and in 2 cases of Langerhans cell variant by immunohistochemistry.

Results and findings

In the 6 cases of conventional CEOT, 5 tumours involved the premolar and molar region and the anterior portion of the mandible was affected in 1 case. Four patients were followed for 2–7 years and did not show any sign of recurrence. A review of the English language literature revealed 5 cases; combined with the present 2 new cases, a total of 7 cases of Langerhans cell variant of CEOT were collected. The patients were all Asian. Six tumours occurred in the maxilla and 1 in mandible; all mainly involved the anterior region of the jaws. Five patients were followed for 2-10 years and did not show any evidence of recurrence. Langerhans cells can be seen in both the conventional and the Langerhans cell variant of CEOT; however, increased numbers of Langerhans cells are seen in the latter.

Conclusions

Although the Langerhans cell variant of CEOT is a rare entity and behaves similarly to the conventional type, it could show unique clinical and histologic features that may pose problems for differential diagnosis.
Appendix
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Literature
1.
go back to reference Pindborg JJ: Calcifying epithelial odontogenic tumors. Acta Pathol Microbiol Scand Suppl. 1955, 111: 71- Pindborg JJ: Calcifying epithelial odontogenic tumors. Acta Pathol Microbiol Scand Suppl. 1955, 111: 71-
2.
go back to reference Pindborg JJ: A calcifying epithelial odontogenic tumor. Cancer. 1958, 11 (4): 838-843. 10.1002/1097-0142(195807/08)11:4<838::AID-CNCR2820110423>3.0.CO;2-5.PubMedCrossRef Pindborg JJ: A calcifying epithelial odontogenic tumor. Cancer. 1958, 11 (4): 838-843. 10.1002/1097-0142(195807/08)11:4<838::AID-CNCR2820110423>3.0.CO;2-5.PubMedCrossRef
3.
go back to reference Philipsen HP, Reichart PA: Calcifying epithelial odontogenic tumour: biological profile based on 181 cases from the literature. Oral Oncol. 2000, 36 (1): 17-26. 10.1016/S1368-8375(99)00061-5.PubMedCrossRef Philipsen HP, Reichart PA: Calcifying epithelial odontogenic tumour: biological profile based on 181 cases from the literature. Oral Oncol. 2000, 36 (1): 17-26. 10.1016/S1368-8375(99)00061-5.PubMedCrossRef
4.
go back to reference Anavi Y, Kaplan I, Citir M, Calderon S: Clear-cell variant of calcifying epithelial odontogenic tumor: clinical and radiographic characteristics. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003, 95 (3): 332-339. 10.1067/moe.2003.8.PubMedCrossRef Anavi Y, Kaplan I, Citir M, Calderon S: Clear-cell variant of calcifying epithelial odontogenic tumor: clinical and radiographic characteristics. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003, 95 (3): 332-339. 10.1067/moe.2003.8.PubMedCrossRef
5.
go back to reference Germanier Y, Bornstein MM, Stauffer E, Buser D: Calcifying epithelial odontogenic (pindborg) tumor of the mandible with clear cell component treated by conservative surgery: report of a case. J Oral Maxillofac Surg. 2005, 63 (9): 1377-1382. 10.1016/j.joms.2005.05.298.PubMedCrossRef Germanier Y, Bornstein MM, Stauffer E, Buser D: Calcifying epithelial odontogenic (pindborg) tumor of the mandible with clear cell component treated by conservative surgery: report of a case. J Oral Maxillofac Surg. 2005, 63 (9): 1377-1382. 10.1016/j.joms.2005.05.298.PubMedCrossRef
6.
go back to reference Asano M, Takahashi T, Kusama K, Iwase T, Hori M, Yamanoi H, Tanaka H, Moro I: A variant of calcifying epithelial odontogenic tumor with Langerhans cells. J Oral Pathol Med. 1990, 19 (9): 430-434. 10.1111/j.1600-0714.1990.tb00874.x.PubMedCrossRef Asano M, Takahashi T, Kusama K, Iwase T, Hori M, Yamanoi H, Tanaka H, Moro I: A variant of calcifying epithelial odontogenic tumor with Langerhans cells. J Oral Pathol Med. 1990, 19 (9): 430-434. 10.1111/j.1600-0714.1990.tb00874.x.PubMedCrossRef
7.
go back to reference Takata T, Ogawa I, Miyauchi M, Ijuhin N, Nikai H, Fujita M: Non-calcifying Pindborg tumor with Langerhans cells. J Oral Pathol Med. 1993, 22 (8): 378-383. 10.1111/j.1600-0714.1993.tb01093.x.PubMedCrossRef Takata T, Ogawa I, Miyauchi M, Ijuhin N, Nikai H, Fujita M: Non-calcifying Pindborg tumor with Langerhans cells. J Oral Pathol Med. 1993, 22 (8): 378-383. 10.1111/j.1600-0714.1993.tb01093.x.PubMedCrossRef
8.
go back to reference Slootweg PJ: Bone and cementum as stromal features in Pindborg tumor. J Oral Pathol Med. 1991, 20 (2): 93-95. 10.1111/j.1600-0714.1991.tb00897.x.PubMedCrossRef Slootweg PJ: Bone and cementum as stromal features in Pindborg tumor. J Oral Pathol Med. 1991, 20 (2): 93-95. 10.1111/j.1600-0714.1991.tb00897.x.PubMedCrossRef
9.
go back to reference Seim P, Regezi JA, O’Ryan F: Hybrid ameloblastoma and calcifying epithelial odontogenic tumor: case report. J Oral Maxillofac Surg. 2005, 63 (6): 852-855. 10.1016/j.joms.2005.02.024.PubMedCrossRef Seim P, Regezi JA, O’Ryan F: Hybrid ameloblastoma and calcifying epithelial odontogenic tumor: case report. J Oral Maxillofac Surg. 2005, 63 (6): 852-855. 10.1016/j.joms.2005.02.024.PubMedCrossRef
10.
go back to reference Wang L, Wang SZ, XMC: Langerhans cells containing calcifying epithelial odontogenic tumor: report of two cases and review of the literature. Oral Oncology EXTRA. 2006, 42: 144-146. 10.1016/j.ooe.2005.10.006.CrossRef Wang L, Wang SZ, XMC: Langerhans cells containing calcifying epithelial odontogenic tumor: report of two cases and review of the literature. Oral Oncology EXTRA. 2006, 42: 144-146. 10.1016/j.ooe.2005.10.006.CrossRef
11.
go back to reference Wang YP, Lee JJ, Wang JT, Liu BY, Yu CH, Kuo RC, Chiang CP: Non-calcifying variant of calcifying epithelial odontogenic tumor with Langerhans cells. J Oral Pathol Med. 2007, 36 (7): 436-439. 10.1111/j.1600-0714.2007.00533.x.PubMedCrossRef Wang YP, Lee JJ, Wang JT, Liu BY, Yu CH, Kuo RC, Chiang CP: Non-calcifying variant of calcifying epithelial odontogenic tumor with Langerhans cells. J Oral Pathol Med. 2007, 36 (7): 436-439. 10.1111/j.1600-0714.2007.00533.x.PubMedCrossRef
12.
go back to reference Barnes L, EJ RP, Sidransky: World Health Organization Classification of Tumours. Pathology & Genetics Head and Neck Tumours. 2005, Lyon: IARC Pres Barnes L, EJ RP, Sidransky: World Health Organization Classification of Tumours. Pathology & Genetics Head and Neck Tumours. 2005, Lyon: IARC Pres
13.
go back to reference Luo HY, Li TJ: Odontogenic tumors: a study of 1309 cases in a Chinese population. Oral Oncol. 2009, 45 (8): 706-711. 10.1016/j.oraloncology.2008.11.001.PubMedCrossRef Luo HY, Li TJ: Odontogenic tumors: a study of 1309 cases in a Chinese population. Oral Oncol. 2009, 45 (8): 706-711. 10.1016/j.oraloncology.2008.11.001.PubMedCrossRef
14.
go back to reference Vap DR, Dahlin DC, Turlington EG: Pindborg tumor: the so-called calcifying epithelial odontogenic tumor. Cancer. 1970, 25 (3): 629-636. 10.1002/1097-0142(197003)25:3<629::AID-CNCR2820250319>3.0.CO;2-H.PubMedCrossRef Vap DR, Dahlin DC, Turlington EG: Pindborg tumor: the so-called calcifying epithelial odontogenic tumor. Cancer. 1970, 25 (3): 629-636. 10.1002/1097-0142(197003)25:3<629::AID-CNCR2820250319>3.0.CO;2-H.PubMedCrossRef
15.
go back to reference Franklin CD, Pindborg JJ: The calcifying epithelial odontogenic tumor. A review and analysis of 113 cases. Oral Surg Oral Med Oral Pathol. 1976, 42 (6): 753-765. 10.1016/0030-4220(76)90098-0.PubMedCrossRef Franklin CD, Pindborg JJ: The calcifying epithelial odontogenic tumor. A review and analysis of 113 cases. Oral Surg Oral Med Oral Pathol. 1976, 42 (6): 753-765. 10.1016/0030-4220(76)90098-0.PubMedCrossRef
16.
go back to reference Demian N, Harris RJ, Abramovitch K, Wilson JW, Vigneswaran N: Malignant transformation of calcifying epithelial odontogenic tumor is associated with the loss of p53 transcriptional activity: a case report with review of the literature. J Oral Maxillofac Surg. 68 (8): 1964-1973. Demian N, Harris RJ, Abramovitch K, Wilson JW, Vigneswaran N: Malignant transformation of calcifying epithelial odontogenic tumor is associated with the loss of p53 transcriptional activity: a case report with review of the literature. J Oral Maxillofac Surg. 68 (8): 1964-1973.
17.
go back to reference Kawano K, Ono K, Yada N, Takahashi Y, Kashima K, Yokoyama S, Yanagisawa S: Malignant calcifying epithelial odontogenic tumor of the mandible: report of a case with pulmonary metastasis showing remarkable response to platinum derivatives. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007, 104 (1): 76-81. 10.1016/j.tripleo.2006.04.014.PubMedCrossRef Kawano K, Ono K, Yada N, Takahashi Y, Kashima K, Yokoyama S, Yanagisawa S: Malignant calcifying epithelial odontogenic tumor of the mandible: report of a case with pulmonary metastasis showing remarkable response to platinum derivatives. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007, 104 (1): 76-81. 10.1016/j.tripleo.2006.04.014.PubMedCrossRef
18.
go back to reference Gopalakrishnan R, Simonton S, Rohrer MD, Koutlas IG: Cystic variant of calcifying epithelial odontogenic tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006, 102 (6): 773-777. 10.1016/j.tripleo.2005.09.029.PubMedCrossRef Gopalakrishnan R, Simonton S, Rohrer MD, Koutlas IG: Cystic variant of calcifying epithelial odontogenic tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006, 102 (6): 773-777. 10.1016/j.tripleo.2005.09.029.PubMedCrossRef
19.
go back to reference Ficarra G, Hansen LS, Stiesmeyer EH: Intramural calcifying epithelial odontogenic tumor. Int J Oral Maxillofac Surg. 1987, 16 (2): 217-221. 10.1016/S0901-5027(87)80134-0.PubMedCrossRef Ficarra G, Hansen LS, Stiesmeyer EH: Intramural calcifying epithelial odontogenic tumor. Int J Oral Maxillofac Surg. 1987, 16 (2): 217-221. 10.1016/S0901-5027(87)80134-0.PubMedCrossRef
20.
go back to reference Poomsawat S, Punyasingh J: Calcifying epithelial odontogenic tumor: an immunohistochemical case study. J Mol Histol. 2007, 38 (1): 103-109. 10.1007/s10735-007-9082-9.PubMedCrossRef Poomsawat S, Punyasingh J: Calcifying epithelial odontogenic tumor: an immunohistochemical case study. J Mol Histol. 2007, 38 (1): 103-109. 10.1007/s10735-007-9082-9.PubMedCrossRef
Metadata
Title
A clinicopathologic study on calcifying epithelial odontogenic tumor: with special reference to Langerhans cell variant
Authors
Yan Chen
Ting-Ting Wang
Yan Gao
Tie-Jun Li
Publication date
01-12-2014
Publisher
BioMed Central
Published in
Diagnostic Pathology / Issue 1/2014
Electronic ISSN: 1746-1596
DOI
https://doi.org/10.1186/1746-1596-9-37

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