Background: Although thromboembolic complications are common in adult patients with malignant diseases, cerebral venous sinus thrombosis has been rarely described in cancer afflicted pediatric and adolescent population. Case history: A 16-year-old adolescent girl referred for complaints of pain and swelling on her left leg. On physical examination, a solid tibial mass was discovered. After the diagnosis of Ewing sarcoma with a tru-cut biopsy, chemotherapy protocol consisting of cisplatin, ifosfamide, adriamycine, and vincristine was started. During the first course of the treatment, the patient expressed headache, diplopia, and ptosis. Contrast-enhanced magnetic resonance (MR) images and MR angiography showed superior sagittal and transverse sinus thromboses. After anticoagulant therapy, the thromboses disappeared within 1.5 months. She received her chemotherapy protocol with the anticoagulant prophylaxis. After a follow-up period of 12 months, she is still in a good neurological recovery without any sequel. Conclusion: Children and adolescents with cancer should be monitored closely for thrombotic complications. We discuss this uncommon case to draw attention to the importance of early diagnosis and adequate treatment of intracranial thrombosis in childhood cancer, and we review the relevant literature. © Springer-Verlag 2008.