Vincristine is an anti-neoplastic agent for intravenous use. Inadvertent intrathecal administration of vincristine tends to follow a predictable clinical sequence of a progressive ascending radiculomyeloencephalopathy, usually fatal in outcome. We report a case of a 10-year-old girl who suffered an inadvertent intrathecal vincristine administration. We have outlined the management strategy used and her consequent neurological progress. She has survived but with a significant neurological deficit. Her outcome is, however, less severe than in previous literature reports; this may in part be attributable to early aggressive neurological management. This case also serves to highlight the tragic consequences of human error when handling chemotherapeutic agents of this nature.