Background: CASTLE (Carcinoma showing thymus-like differentiation) in thyroid gland is a rare disease with favorable prognosis. Treating with surgery and adjuvant radiation therapy has been reported to be able to improve local controlling and long – term survival rates. In this report, we give an account of a clinical case of thyroid gland CASTLE and include relevant literature review in diagnosis and treatment of this disease.Case presentation: A 60-year-old female patient diagnosed with Castle in 2015, had a total thyroidectomy and hormone maintenance. After 5 years, the patient was diagnosed with recurrence, late stage and incapable of surgery. Due to the refusal of radiation therapy, the patient is followed and is currently stable after 15 months.Conclusions: CASTLE is a rare disease, diagnosis is based on postoperative pathology and immunohistochemistry analysis, especially upon CD5 marker. In the case of relapse, treatment includes surgery and radiation therapy. In some cases, follow-up is also an acceptable option for the patient.
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