Nasopalatine duct cyst with sebaceous differentiation: a rare case report with literature review

BACKGROUND: The aim of this study was to report a rare case of nasopalatine duct cyst with sebaceous differentiation. Further, a systematic search of the literature was performed to identify studies reporting patients with intraosseous jaw cysts with sebaceous differentiation. CASE PRESENTATION: A 55-year-old Korean man was referred to our hospital because of a cystic lesion of the anterior maxilla. Radiologic examination revealed a well-circumscribed radiolucent lesion in the anterior maxilla. Histology showed a respiratory columnar and cuboidal epithelium-lined cyst. Transition from the ciliated columnar epithelium to stratified squamous epithelium with sebaceous differentiation was observed. Based on these findings, the final diagnosis was nasopalatine duct cyst with sebaceous differentiation. A systematic search of the literature was performed to identify studies reporting patients with intraosseous jaw cysts with sebaceous differentiation. There were 24 cases of sebaceous differentiation in the epithelium of the cysts including 2 odontogenic keratocysts, 8 orthokeratinized odontogenic cysts, 8 dentigerous cysts, 1 radicular cyst, and 2 glandular odontogenic cysts. However, no case reports describing the occurrence of nasopalatine duct cysts with sebaceous differentiation have been reported. CONCLUSION: This first case report of nasopalatine duct cysts with sebaceous differentiation could provide insight into the diagnostic process of cystic lesions with sebaceous differentiation.