A 3-year-old boy presented with multiple brain metastases 21 months after the resection of stage II Wilms' tumor. Metastasis to other organs was not found. He was treated by total removal of a large metastatic tumor in the left temporal lobe and post-operative radiotherapy and chemotherapy. He has been in complete remission for 20 months after surgery. Cerebral metastasis from Wilms' tumor without systemic metastases is very rare. It is speculated that brain metastases occurred in this patient because most of the anticancer agents used in the primary treatment for Wilms' tumor were not able to cross the blood brain barrier.

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