CASE REPORT   

European Journal of Oral and Maxillofacial Surgery 2022 December;6(3):132-6

DOI: 10.23736/S2532-3466.22.00294-6

Copyright © 2022 EDIZIONI MINERVA MEDICA

language: English

Adult-onset xanthogranuloma manifested in maxillary sinus: a case report

Masataka UEHARA ¹ ✉, Daisei YASUDA ²

¹ Unit of Oral and Maxillofacial Surgery, Shimonoseki City Hospital, Shimonoseki, Japan; ² Unit of Pathology, Shimonoseki City Hospital, Shimonoseki, Japan

The xanthogranuloma is a member of the non-Langerhans cell histiocytosis group. Typically, xanthogranuloma is a disease of childhood/adolescence, manifesting as cutaneous lesions. We present a rare case of xanthogranuloma that manifested in a 68-year-old Japanese woman’s maxillary sinus. Computed tomography revealed a massive lesion occupying the right maxillary sinus protruding into the orbit. An aggregation of histiocytes was observed in an incisional-biopsy hematoxylin and eosin-stained specimen: the immunohistochemical examination of the histiocytes revealed the CD68 but not CAM5.2 expression. The tumor was histologically diagnosed as xanthogranuloma without evidence of malignancy. Because enblock tumor enucleation may cause an inferior deviation of the globe, a two-stage surgery was performed: 1) reduction of the tumor; and 2) total enucleation of the tumor. This strategy successfully induced regeneration of the floor of the orbit and has controlled the tumor for 32 months after the second-stage surgery.

KEY WORDS: Adult; Xanthogranuloma, juvenile; Maxillary sinus