Therapeutic options for metastatic insular thyroid carcinoma (ITC) are very limited. We present the case of a 39-year-old woman with refractory metastatic ITC who responded to oxaliplatin plus irinotecan chemotherapy. She was initially diagnosed with a locally advanced and metastatic to lung ITC and underwent near-total thyroidectomy. After surgery she was treated and failed to respond first to radioiodine and subsequently to liposomal doxorubicin plus paclitaxel combination chemotherapy and was offered palliative external irradiation for metastases to bone and the brain. Eleven months postdiagnosis the patient became severely dyspnoic as a result of progression of lung metastases and elected to be treated with oxaliplatin plus irinotecan as a secondline chemotherapy. She experienced relief of her dyspnea shortly after treatment initiation and an objective response of her lung metastases was documented after the third course of treatment. Treatment continued for six cycles and tumor remission lasted for 5 months. Toxicity was mild. Confirmatory testing of oxaliplatin-irinotecan combination in case series or single cases of metastatic ITC is warranted.
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