A 21-year-old man with homozygous sickle cell disease maintained on hydroxyurea for 1 year developed thrombosis of the superior sagittal, right transverse, and right sigmoid dural sinuses with a large venous infarct. He was successfully treated with endovascular thrombolysis. Investigation did not reveal any inherited or acquired hypercoagulable state. This patient however had consistently elevated hemoglobin levels both at the time of the initial event and on follow up. One year later he developed symptomatic avascular necrosis of the right hip. High hemoglobin levels resulting from hydroxyurea therapy may have contributed to development of complications in this patient.
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