Cerebral venous thromboembolism is a rare cerebrovascular disorder reported in acute promyelocytic leukemia, witch is more known for bleeding complications as the leading cause of early mortality. We report a case of 28 years old Moroccan woman, diagnosed with cerebral venous thrombosis as an initial presentation of acute promyelocytic leukemia. As soon as the diagnostic was made, treatment by all trans retinoic acid combined to conventional chemotherapy concomitant to systemic anticoagulation were started. This combination of treatment was sufficient to obtain complete remission and led to a complete disappearance of the thrombosis as well as the neurological signs without squeal. This case highlights the importance of a rigorous and complete investigation that some thromboembolic events call for.