J Korean Assoc Oral Maxillofac Surg.  2011 Dec;37(6):520-523. 10.5125/jkaoms.2011.37.6.520.

Keratoameloblastoma of the maxilla: a case report

• Won JH
• Na HY
• Kim HS
• Kim J
• Nam W
• Cha IH
• Kim HJ

• KMID: 2136991
• DOI: http://doi.org/10.5125/jkaoms.2011.37.6.520

Abstract

A keratoameloblastoma is a histologically variant of the ameloblastoma group, which varies in size and contains keratin material in the fibrous connective tissue among cystic lesions. A keratoameloblastoma is a rare disease with only 13 cases reported in the literature since Pindborg's first report in 1970. A 41-year-old man visited, complaining of pus discharged from the right maxilla. He had been diagnosed with an odontogenic keratocyst and was treated with cyst enucleation in the past. The clinical and radiology examination found evidence of recurrence and finally diagnosed him with keratoameloblastoma after enucleation and biopsy. This report discusses the clinical, radiological and histological characteristics of keratoameloblastoma and its treatment. In addition, we report another case of keratoameloblastoma that had transformed from an odontogenic keratocyst.

Keyword

Keratoameloblastoma; Maxilla; Papilliferous keratoameloblastoma; Odontogenic keratocyst

MeSH Terms

Adult
Ameloblastoma
Biopsy
Connective Tissue
Humans
Keratins
Maxilla
Odontogenic Cysts
Rare Diseases
Recurrence
Suppuration
Keratins

Figure

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  Fig. 1. The panoramic radiograph showing radiolucent lesion occupying right maxilla. Ji-Hoon Won et al: Keratoameloblastoma of the maxilla: a case report. J Korean Assoc Oral Maxillofac Surg 2011

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  Fig. 2. Axial and coronal computed tomography (CT) view. A. The axial CT scan shows well-defined and low attenuated cystic lesion about 3.5 cm sized in the right maxilla posterior region. Also, we can find marked expansion of the maxilla with perforation of cortical plates. B. The coronal scan shows elevation and thickening of right sinus mucosa of floor. The lesion is involved with nasal cavity. Ji-Hoon Won et al: Keratoameloblastoma of the maxilla: a case report. J Korean Assoc Oral Maxillofac Surg 2011

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  Fig. 3. Postoperative panoramic radiograph taken 2 months after decompression of cystic lesion. The lesion become smaller than preoperative panoramic radiograph. Ji-Hoon Won et al: Keratoameloblastoma of the maxilla: a case report. J Korean Assoc Oral Maxillofac Surg 2011

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  Fig. 4. Postoperative computed tomography taken 3 months after decompression of cystic lesion. The surgical site exhibits bone formation without evidence of disease. Also, the lesion is separated from nasal cavity and mucosa. Ji-Hoon Won et al: Keratoameloblastoma of the maxilla: a case report. J Korean Assoc Oral Maxillofac Surg 2011

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  Fig. 5. A. Typical epithelial lining of an odontogenic keratocyst (1996. 3.). B, C, D. Follicular epithelial structure containing layers of parakeratin. The follicle is lined by ameloblast-like colummar cells exhibiting hyperchromatism, palisading, reversed polarity. Several solid keratinized epithelial follicles present in stroma (2009. 6) (H&E staining, A: ×40, B, C, D: ×200). Ji-Hoon Won et al: Keratoameloblastoma of the maxilla: a case report. J Korean Assoc Oral Maxillofac Surg 2011

Reference

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