A 26-year-old woman suffered concomitant inferior vena cava, iliac, and femoral vein thrombosis and cerebral venous thrombosis. Ten days before symptom onset she had started using an oral contraceptive that contained low-dose ethynilestradiol and gestodene. Both protein C deficiency and protein C activated resistance were detected. To our knowledge, the association of cerebral, caval, and ilio-femoral-popliteal venous thrombosis has not been described previously. The severity of the clinical features could be a consequence of the two combined thrombophilic mechanisms and of the continuation of the oral contraceptive. A thrombophilic disorder should be considered in young patients with thromboembolic disease. Because of the high prevalence of the genetic deficiency causing protein C activated resistance, it is probably worthwhile to perform general screening before prescription of oral contraceptives.