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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.

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. [ABSTRACT FROM AUTHOR]

Spindle epithelial tumor with thymus-like differentiation (SETTLE) is a rare malignant thyroid tumor showing thymic or related branchial pouch differentiation. The tumors are composed predominantly of spindle cells along with focal epithelial component and ductular formations. SETTLE occurs in young patients, with indolent growth and a tendency to develop delayed blood-borne metastases. We herein report two cases of SETTLE with a follow-up period of 64 months and 30 months, respectively.

The aim of the present study was to summarize the clinical characteristics, diagnosis, treatment and prognosis of carcinoma showing thymus-like differentiation of the thyroid (CASTLE). A search of the relevant literature was conducted, which identified 82 cases of CASTLE reported to date. A review of the clinical and auxiliary examination data, treatment, pathological findings and follow-up of these cases was performed. The clinical manifestations of CASTLE varied and did not demonstrate specificity. During ultrasound examination, CASTLE was described as a solid and hypoechoic mass, and the echo was typically observed to be heterogeneous. In computed tomography (CT), CASTLE was demonstrated to be a well-defined, soft tissue density mass without calcification, which appeared enhanced following administration of contrast medium. In emission CT, CASTLE was revealed as a 'cold nodule'. A total of 78 patients underwent radical surgery, and 12 patients experienced recurrence. Lymph node metastasis did not promote recurrence, and postoperative radiation was not able to reduce the recurrence rate (P=0.144 and 1.000, respectively). The median follow-up time was 14 months (range, 1-312 months). Typical immunohistochemical examinations demonstrated the specimens to be positive for cluster of differentiation (CD)5, CD117, cytokeratin 19, epithelial membrane antigen and tumor protein 63, and negative for calcitonin, thyroglobulin and thyroid transcription factor-1. Thyroid CASTLE is a rare malignancy whose exact diagnosis relies on pathological examination, particularly immunohistochemistry, since preoperative examinations are usually unable to provide an exact diagnosis. Radical resection is the primary treatment for CASTLE, which presents favorable prognosis. [ABSTRACT FROM AUTHOR]

The aim of the present study was to summarize the clinical characteristics, diagnosis, treatment and prognosis of carcinoma showing thymus-like differentiation of the thyroid (CASTLE). A search of the relevant literature was conducted, which identified 82 cases of CASTLE reported to date. A review of the clinical and auxiliary examination data, treatment, pathological findings and follow-up of these cases was performed. The clinical manifestations of CASTLE varied and did not demonstrate specificity. During ultrasound examination, CASTLE was described as a solid and hypoechoic mass, and the echo was typically observed to be heterogeneous. In computed tomography (CT), CASTLE was demonstrated to be a well-defined, soft tissue density mass without calcification, which appeared enhanced following administration of contrast medium. In emission CT, CASTLE was revealed as a 'cold nodule'. A total of 78 patients underwent radical surgery, and 12 patients experienced recurrence. Lymph node metastasis did not promote recurrence, and postoperative radiation was not able to reduce the recurrence rate (P=0.144 and 1.000, respectively). The median follow-up time was 14 months (range, 1-312 months). Typical immunohistochemical examinations demonstrated the specimens to be positive for cluster of differentiation (CD)5, CD117, cytokeratin 19, epithelial membrane antigen and tumor protein 63, and negative for calcitonin, thyroglobulin and thyroid transcription factor-1. Thyroid CASTLE is a rare malignancy whose exact diagnosis relies on pathological examination, particularly immunohistochemistry, since preoperative examinations are usually unable to provide an exact diagnosis. Radical resection is the primary treatment for CASTLE, which presents favorable prognosis.