Lorenz Lisa, von Rappard Joscha, Arnold Walter, Mutter Nicole, Schirp Udo, Scherr Andreas, Jehle Andreas Werner
Department of Internal Medicine, Hirslanden Klinik St. Anna, Lucerne, Switzerland.
Department of Pathology, Cantonal Hospital Lucerne, Lucerne, Switzerland.
Front Oncol. 2019 Aug 14;9:734. doi: 10.3389/fonc.2019.00734. eCollection 2019.
Carcinoma showing thymus-like elements (CASTLE) is a rare tumor, most commonly found in the thyroid 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 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.
具有胸腺样成分的癌（CASTLE）是一种罕见肿瘤，最常见于甲状腺。本文报告1例局限于腮腺的CASTLE肿瘤病例，该病例在出现有症状的胸膜癌转移的晚期表现后经回顾性诊断得以确诊。腮腺原发病灶行手术治疗并辅助放疗。10年后，出现伴有反复胸腔积液的转移性疾病。胸膜癌转移灶与腮腺原发病灶一样，CD5、CD117和p63均呈强阳性，据此确诊为CASTLE肿瘤。此外，胸膜肿瘤表达高水平的程序性死亡配体1（PD-L1），该患者接受了单克隆PD-L1抑制剂帕博利珠单抗治疗，病情获得部分缓解。据我们所知，这是首例接受PD-L1抑制剂治疗的转移性CASTLE肿瘤患者。
