J Korean Assoc Oral Maxillofac Surg.  2013 Aug;39(4):182-187.

Expansile keratocystic odontogenic tumor in the maxilla: immunohistochemical studies and review of literature

• Byun JH
• Kang YH
• Choi MJ
• Park BW

Abstract

Keratocystic odontogenic tumors (KCOT) - previously termed odontogenic keratocysts (OKC) - are characterized by aggressive behavior and a high rate of recurrence. Histopathologically, the basal layer of KCOT shows a higher cell proliferation rate and increased expression of anti-apoptosis genes. Clinically, KCOT is frequently involved in the mandibular posterior region but is not common in the posterior maxilla. However, it should be noted that due to its expansive characteristics, KCOT involved near the maxillary sinus could easily expand to an enormous size and occupy the entire maxilla. To achieve total excision of these expanded cystic tumors in the maxilla, a more aggressive approach would be needed. In this report, we describe two cases of expansile KCOT involving the entire unilateral maxilla and maxillary sinus; they were completely excised using the Weber-Ferguson approach, showing no evidence of recurrence during the follow-up period of more than two years. In immunohistochemical analyses of the tumor specimens, p53 and p63 showed strong expression, and B-cell lymphoma 2 (BCL2) and MKI67 (Ki-67) showed moderate or weak expression, however, detection of BCL2-associated X protein (BAX) was almost negative. These data indicate that expansile KCOT possesses increased anti-apoptotic activity and cell proliferation rate but decreased apoptosis. These properties of KCOT may contribute to tumor enlargement, aggressive behavior, and high recurrence rate.

Keyword

Keratocytic odontogenic tumor; Odontogenic keratocyst; Immunohistochemistry

MeSH Terms

Apoptosis
bcl-2-Associated X Protein
Cell Proliferation
Follow-Up Studies
Immunohistochemistry
Lymphoma, B-Cell
Maxilla
Maxillary Sinus
Odontogenic Cysts
Odontogenic Tumors
Recurrence
bcl-2-Associated X Protein

Figure

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  Fig. 1 Preoperative radiologic views of case 1. The panoramic view showed the large bone with the destroyed cystic lesion from the anterior maxilla to the entire left side of the maxilla and maxillary sinus. Partial dental root resorption of the posterior teeth involved was observed, but tooth deviation was not detected. In computed tomography view, homogenous cystic lesion was expanded to buccal and palatal bony wall with partial perforation of cortical bone.

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  Fig. 2 Intraoperative view and postoperative routine radiograms of case 1. The expansile maxillary cystic tumor was completely excised with Weber-Ferguson's approach, and all teeth involved were retained. Panorama and Water's view 1 year after surgery; there was no evidence of recurrence, showing normal sinus function.

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  Fig. 3 Preoperative panorama and computed tomography views of case 2. Large cystic lesion with cortical bone expansion on the entire left side of the maxilla and maxillary sinus was detected. Partial root resorption of the teeth involved was observed similar to case 1.

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  Fig. 4 Intraoperative views, tumor specimen, and postoperative radiograms and clinical view of case 2. The expansile maxillary cystic tumor was completely excised via Weber-Ferguson's approach, and there was no clinical evidence of any recurrence or complication for follow-up period.

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  Fig. 5 Histological and immunohistochemical (IHC) analysis of tumor specimens of case 1 (A) and case 2 (B). In H&E-stained slides of two cases, thin lining epithelium and parakeratin formation in the lumen side of the tumors were observed, indicating that the tumor was KCOT. In the IHC analysis of the specimens, MKI67 (Ki-67) was moderately expressed in case 1 and weakly reacted in case 2; tumor protein 53 (p53) and tumor protein 63 (p63) were strongly expressed in the basal and suprabasal layers of the tumor epithelium in two cases. B-cell lymphoma 2 (BCL2) was moderately expressed in the stromal and epithelial layers of tumors, whereas BCL2-associated X protein (BAX) was almost negatively detected in the specimens in two cases. Scale bar=100 µm.

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