Pembrolizumab in a Patient With a Metastatic CASTLE Tumor of the Parotid

Pembrolizumab in a Patient With a Metastatic CASTLE Tumor of the Parotid

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Carcinoma showing thymus-like elements (CASTLE) is a rare tumor, most commonly found in the thyroid Production Editing Controllers gland.Here we report a case of CASTLE tumor localized to the parotid gland, recognized in retrospect after a late manifestation of symptomatic pleural carcinomatosis.The original tumor in the parotid gland was treated by surgery followed by radiotherapy.Ten years later, a metastatic disease with recurrent pleural effusions occurred.Pleural carcinomatosis was strongly positive for CD5, CD117, and p63 as was the original tumor of the parotid, which allowed the diagnosis of a CASTLE tumor.

Additionally, the pleural tumor expressed throwing stars high levels of programmed death ligand 1 (PD-L1), and the patient underwent treatment with the monoclonal PD-L1 inhibitor pembrolizumab achieving a partial remission.To the best of our knowledge, this is the first patient with a metastatic CASTLE tumor treated with a PD-L1 inhibitor.