Vincristine has a high neurotoxicity level. If given intrathecally by accident, it can cause ascending radiculomyeloencephalopathy, which is almost always fatal. The authors report a rare case in which vincristine was accidentally injected intrathecally into a 32-year-old man. The patient, who had Burkitt lymphoma, was neurologically intact, and it is likely that his survival was made possible due to aggressive neurosurgical therapy. After immediate cerebrospinal fluid (CSF) aspiration, external ventricular and lumbar drains were placed for CSF irrigation, which was continued for 6 days. This CSF irrigation was combined with 1) the intrathecal administration of fresh-frozen plasma to bind the vincristine and 2) an intravenous antineurotoxic therapy involving pyridoxine, folic acid, and glutamic acid. The patient's first sensorimotor deficits occurred after 2 days, led to an incomplete sensorimotor dysfunction below T-9 within the next 17 days, but progressed no further. Supported by the scarce data culled from the reviewed literature, the authors hypothesize that prolonged CSF irrigation combined with antineurotoxic therapy contributed to the patient's satisfactory outcome. In conclusion, accidental intrathecal vincristine injection requires emergency and adequate neurosurgical therapy.