A 39-year-old woman with aplastic anemia suffered from progressive visual disturbance and headache. Funduscopic examination revealed pronounced bilateral papilledema. Lumbar puncture disclosed an elevated opening CSF pressure with normal biochemical and cellular findings. Brain MRI was normal. After transfusion of blood products, papilledema and her symptoms were improved. We conclude that the development of pseudotumor cerebri is related to aplastic anemia resulting from bone marrow failure.