Benign intracranial hypertension is a rare complication of systemic lupus erythematosus often attributed to cerebral sinus thrombosis which impairs venous drainage and cerebrospinal fluid outflow. We report the case of a woman with a primary antiphospholipid syndrome who developed benign intracranial hypertension with no actual evidence of venous cerebral thrombosis and with no other possible cause for this clinical manifestation than high titres of anticardiolipin antibodies and a lupus anticoagulant.