Antiphospholipid antibody (APLA) syndrome is a major risk factor for arterial and venous thrombosis. Surgical risks in patients known to suffer from APLA syndrome are usually related to haemorrhage or to thrombo-embolic events, such as deep venous thrombosis and pulmonary emboli. The rare published reports of patients with APLA syndrome undergoing neurosurgical procedures relate to haemostatic complication, with none alerting to the peri-operative risk of stroke in these patients. We present a case of a peri-operative stroke in a patient undergoing resection of a foramen magnum meningioma. We discuss the association of peri-operative stroke and APLA syndrome and emphasize the high risk it imposes for neurosurgical procedures in these patients. In addition, we suggest an anticoagulation treatment algorithm for APLA syndrome patients undergoing craniotomies.