A 16-year-old girl developed right middle cerebral artery infarction and deep venous thrombosis of the lower extremities in association with circulating lupus-like anticoagulant. Currently, she is functionally independent with no further vascular insults and is being treated with sodium warfarin. This patient illustrates that cerebral ischemia can occur in association with lupus anticoagulant in the pediatric population. This entity should be considered and appropriate screening tests performed in young patients with unexplained ischemic stroke or transient ischemic attack.