• Yong Xia, Zongchang Song, Xinlei Zhang, Xinhong Guan, Guifang Tan, Yi Le, Shuang Liu, Hui Xue, Jing Li, Yajun Zhang, Jing Chen, Huajuan Jiang, Xia Jiang, Yanxia Cheng, Chuchu Zhou, Xu Sha, and Jin-Xin Lou.
• Department of Oncology, Shanghai University Affiliated Mengchao Cancer Hospital, Shanghai, China.
• Medicine (Baltimore). 2023 Sep 22; 102 (38): e35225e35225.

RationaleGhost cell odontogenic carcinoma is a rare malignant odontogenic carcinoma characterized by the presence of ghost cells. It has a nonspecific clinical and radiographic presentation and can be locally destructive and invasive, sometimes with distant metastases. However, no effective systemic therapy is currently recommended for such patients.Patient ConcernsThe patient has been unable to undergo surgery or radiotherapy again. Therefore, he was referred to our department for a more aggressive, multimodal systematic treatment program.DiagnosesThe histopathological examination was morphologically suggestive of ghost cell odontogenic carcinomas.InterventionsWe report a case of locally invasive primary inoperable odontogenic shadow cell carcinoma in a 31-year-old Chinese man who achieved treatment with Toripalimab and chemotherapy, followed by Toripalimab maintenance therapy after 6 cycles.OutcomesHe achieved partial remission after treatment. The quality of life significantly improved after treatment. There were no grade 3/4 treatment-related adverse events during treatment.LessonsThis case presented that Toripalimab and chemotherapy may be a safe and effective systemic therapy for ghost cell odontogenic carcinoma.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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