Open Access Case report

A pigmented calcifying cystic odontogenic tumor associated with compound odontoma: a case report and review of literature

Phuu P Han1, Hitoshi Nagatsuka1*, Chong H Siar2, Hidetsugu Tsujigiwa1, Mehmet Gunduz1, Ryo Tamamura1, Silvia S Borkosky1, Naoki Katase1 and Noriyuki Nagai1

Author Affiliations

1 Department of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Okayama 700-8525, Japan

2 Department of Oral Pathology, Oral Medicine and Periodontology, Faculty of Dentistry, University of Malaya, 50603 Kuala Lumpur, Malaysia

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Head & Face Medicine 2007, 3:35  doi:10.1186/1746-160X-3-35

Published: 25 September 2007

Abstract

Background

Pigmented intraosseous odontogenic lesions are rare with only 47 reported cases in the English literature. Among them, pigmented calcifying cystic odontogenic tumor, formerly known as calcifying odontogenic cyst, is the most common lesion with 20 reported cases.

Methods

A case of pigmented calcifying cystic odontogenic tumor associated with odontoma occurring at the mandibular canine-premolar region of a young Japanese boy is presented with radiographic, and histological findings. Special staining, electron microscopic study and immunohistochemical staining were also done to characterize the pigmentation.

Results

The pigments in the lesion were confirmed to be melanin by Masson-Fontana staining and by transmission electron microscopy. The presence of dendritic melanocytes within the lesion was also demonstrated by S-100 immunostaining.

Conclusion

The present case report of pigmented calcifying cystic odontogenic tumor associated with odontoma features a comprehensive study on melanin and melanocytes, including histochemical, immunohistochemical and transmission electron microscopic findings.