J Korean Assoc Oral Maxillofac Surg.  2014 Aug;40(4):199-203. 10.5125/jkaoms.2014.40.4.199.

Clear cell odontogenic carcinoma mimicking a cystic lesion: a case of misdiagnosis

Affiliations
  • 1Department of Oral and Maxillofacial Surgery, College of Dentistry, Yonsei University, Seoul, Korea. omsnam@yuhs.ac
  • 2Department of Oral Pathology, College of Dentistry, Yonsei University, Seoul, Korea.
  • 3Oral Cancer Research Institute, College of Dentistry, Yonsei University, Seoul, Korea.

Abstract

Clear cell odontogenic carcinoma (CCOC) is a rare jaw tumor that was classified as a malignant tumor of odontogenic origin in 2005 by the World Health Organization because of its aggressive and destructive growth capacity and metastasis to the lungs and lymph nodes. We report a case of a 66-year-old female who had swelling, incision and drainage history and a well-defined unicystic radiolucent lesion that was comparable to a cystic lesion. At first, the patient received decompression, and the lesion size decreased. Three months after decompression, cyst enucleation was performed. The pathologic result indicated that the lesion was CCOC. In this report we emphasize that patients with painful cystic lesions in addition to jaw enlargement and loosening teeth should be considered for the possibility of malignancy.

Keyword

Mouth neoplasms; Head and neck neoplasms

MeSH Terms

Aged
Decompression
Diagnostic Errors*
Drainage
Female
Head and Neck Neoplasms
Humans
Jaw
Lung
Lymph Nodes
Mouth Neoplasms
Neoplasm Metastasis
Tooth
World Health Organization

Figure

  • Fig. 1 Panoramic radiograph showing a defined radiolucent lesion in the right maxillary region (arrow).

  • Fig. 2 Computed tomography image at first visit showed well defined and low attenuated cystic lesion on the right maxillary region.

  • Fig. 3 Two month after decompression, a panoramic radiograph showed that a lesion size decreased, compared with previous radiograph (arrow).

  • Fig. 4 Three-month follow-up cone-beam computed tomography image after decompression.

  • Fig. 5 Three-dimensional reconstruction image, image at first visit is on the left (lesion is indicated with black arrows) and image at 3-months after decompression is on the right (lesion is indicated with white arrows).

  • Fig. 6 Histopathologic findings. A. Histopathologic examination of the previous enucleation specimen revealed tumorous proliferation of epithelial nests infiltrating the stroma, indicating malignancy (H&E staining, ×100). B. Cuboidal shaped odontogenic cells were also noted among the tumorous clear cells (H&E staining, B: ×200, inset box: ×200). C. In the resected specimen, the tumor cells (T) infiltrated to the bone marrow of the maxilla (H&E staining, ×100). D. Lymphovascular permeation of tumor cells (arrowheads) (H&E staining, ×200).

  • Fig. 7 Magnetic resonance imaging after enucleation of the lesion. The top image is T1-weighted and lower image is T2-weighted. Magnetic resonance images show a well-defined cystic mass.

  • Fig. 8 There was no evidence of local recurrence on eight month follow-up panoramic radiograph after operation.


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