Remarkable short-term regression of a posterior mediastinum metastasis from primary intraosseous carcinoma treated with nivolumab: a case report

Publication date: Available online 26 November 2018

Source: Journal of Oral and Maxillofacial Surgery

Author(s): Masashi Kimura, Yukihiro Shiraki, Kenichiro Ishibashi, Masahiro Umemura

Abstract

Primary intraosseous carcinoma (PIOC, NOS) is a rare odontogenic malignancy that is thought to arise from the odontogenic epithelium. In addition, some cases arise in odontogenic cysts or other benign precursors. There are no established treatment protocols for PIOC because of its rarity. In particular, no standard therapy has been established for patients with recurrent or metastatic PIOC. Here we report a case in which the use of nivolumab showed a significant treatment response in metastatic PIOC. The patient was a 71-year-old male with PIOC. We performed segmental mandibulectomy with modified radical neck dissection following neoadjuvant chemotherapy, including two cycles of paclitaxel, cetuximab, and carboplatin and one cycle of docetaxel, cisplatin, and 5-fluorouracil. The lesion rapidly enlarged during neoadjuvant chemotherapy, and surgical specimens demonstrated no histological therapeutic effects of chemotherapy. Three months after the surgery, contrast-enhanced CT scans showed posterior mediastinal lymph node metastasis. Treatment with nivolumab was initiated, and significant clinical improvement was noted after two cycles. Although it was a short-term treatment, we observed remarkable effects and no adverse events during and after nivolumab treatment. Nivolumab may be an attractive treatment option for metastatic PIOC that is resistant to traditional chemotherapy and target therapy, including cetuximab. Further investigation of this treatment is warranted.

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