Acquired amegakaryocytic thrombocytopenia (AAT) is a rare disorder, characterized by severe thrombocytopenia and selective, marked decrease or absence of megakaryocytes in the bone marrow. We describe a 29-year-old female with adult onset Still's disease preceding a diagnosis of AAT and autoimmune hemolytic anemia, which was successfully treated with cyclosporine. This is the first case of AAT in a patient with adult onset Still's disease.