Sunitinib (SU011248; Sutent) is a new small molecule that inhibits members of the split-kinase domain family of receptor tyrosine kinases (RTKs), with established antitumor activity in renal cancer. In the current report, we describe a patient with a solitary brain metastasis from renal cell carcinoma who achieved partial response of the cerebral lesion following treatment with sunitinib. To the best of our knowledge, this is the first report of sunitinib activity in brain metastases from kidney cancer. A limited number of publications support the hypothesis that small tyrosine kinase inhibitors may cross the blood-brain barrier. Although the role of sunitinib in advanced renal carcinoma has been evaluated through prospective trials, the efficacy of the drug in patients with brain metastases has not been explored, since patients with cerebral lesions were excluded in those studies. Thus, we believe that accumulating evidence from personal experience or limited reports could be useful. Moreover, in our case, sunitinib was found to be safe, leading to considerable shrinkage of the brain metastasis without any serious adverse events or central nervous system toxicities. We consider this observation to be important, given the absence of data regarding the activity of the drug in this particular clinical setting.