Inflammatory bowel disease (IBD) including ulcerative colitis and Crohn's disease puts patients at high risk of thromboembolism accidents. These patients may take infliximab for active and fistulating Crohn's disease, which can also increase the risk of thrombosis. Deep vein thrombosis (DVT) and pulmonary thromboembolism (PTE) are more common among these patients, but cerebrovascular, mesenteric, portal and retinal veins can also be affected. In this paper, we report a case of isolated right Labbe vein thrombosis after infliximab therapy for Crohn's disease. To the best of our knowledge, our patient is the first case report of isolated cortical vein thrombosis following administration of rituximab for Crohn's disease.