Calcifying odontogenic cyst combined with odontogenic keratocyst: report of a case and review of the literature

Abstract

Introduction and importance: Odontogenic lesions composed of two or more separate odontogenic cysts are uncommon. The calcifying odontogenic cyst (COC) and odontogenic keratocyst (OKC) are odontogenic cysts that exhibit some neoplastic features. Concurrence of these lesions is extremely rare. We report a rare case of COC combined with OKC in a patient and review the relevant literature systemically.

Case presentation: In this article, we present a case report of an 18-year-old boy patient with no medical history whose chief complaint was facial asymmetry. A painless swelling in the right side of the posterior mandible was observed. Histopathological and radiographic investigations revealed a combined odontogenic lesion composed of inflamed OKC with COC.

Clinical discussion: Long-lasting debates on the true nature of OKC and COC, whether cystic or neoplastic, has been existed so far. Their genetic-derived mechanisms can justify the concurrent occurrence of these cysts in a patient. The treatment plan for this lesion included decompression followed by enucleation, curettage, and application of Carnoy's solution.

Conclusion: We reported a rare case of an inflamed OKC with COC-like features. Based on the literature, the coincidence of these two odontogenic cysts has been reported for the third time here.

Keywords: Calcifying odontogenic cyst; Keratocystic odontogenic tumor; Mixed odontogenic tumor; Odontogenic cyst; Odontogenic keratocyst.

Chart 1

PRISMA flowchart containing the search strategy and the respective selection process.

Fig. 1

A. Panoramic imaging reveals a unilocular radiolucency in the posterior area extending from the first to the third molar, B. CBCT imaging demonstrates a well-defined border in the right mandibular body, which expands the buccal and lingual cortex.

Fig. 2

A. Histopathologic feature demonstrates a cystic lesion lined by parakeratinized stratified squamous epithelium with palisaded basal cell layer which detached from underlying connective tissue, B. the junction of epithelium and underlying connective tissue is flattened, and the surface of the epithelium shows corrugation.

Fig. 3

The photomicrograph field of sections reveals aggregations of the ghost cells and calcifications in the underlying connective tissue.

Fig. 4

The high-power field of sections shows the cyst's lining with a hyperchromatic basal cell layer and columnar cells with a reverse polarity appearance.

Fig. 5

No recurrence of the lesion according to the A. panoramic post-op, and B. photographic post-op images one year after treatment.