Introduced in 1988 by Kaneko and colleagues, selective ophthalmic arterial infusion of chemotherapeutic drug has recently gained more interest among retinoblastoma experts worldwide. The report showed that the procedure could be repeated up to 12 treatments without serious side effects. We report a 4-year-old girl with bilateral retinoblastoma. The left eye was enucleated for the group E disease. The right eye started with 3 retinal tumors (group C) was treated with systemic chemotherapy plus local therapy. Seven months after the last cycle of chemotherapy, the tumor recurred close to the fovea. Systemic chemotherapy was reinitiated without success. To avoid aggressive cryotherapy and external-beam radiotherapy, selective ophthalmic arterial infusion of chemotherapeutic drugs was performed for 15 sessions. The tumor responded partially without evidence of drug-induced retinal toxicity by the electroretinogram. Minor irregularities of the inner wall of supraclinoid portion of the internal carotid artery were observed only at the sixth session. Narrowing of the vascular lumen occurred on the last 2 sessions. We demonstrate that this technique when performed repeatedly could result in the anatomic changes of the small blood vessel. Careful follow-up is necessary for early detection of any serious consequences.