We report the case of a 16-month-old patient with chronic immune thrombocytopenia (ITP) patient who experienced delayed treatment-free response (TFR) after romiplostim treatment. He received intravenous immunoglobulin every month to maintain a platelet count above 20,000/μL for 2 years. Thereafter, he received rituximab and cyclosporine as second-line therapy, with no response, followed by romiplostim. After 4 weeks of treatment, the platelet count was maintained above 50,000/μL. Following 7 months of treatment, he discontinued romiplostim, and the platelet count decreased. His platelet counts remained above 50,000/μL, without any bleeding symptoms, 2 years after romiplostim discontinuation. This is the first report of TFR after romiplostim treatment in pediatric chronic ITP.
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BACKGROUND Acute idiopathic thrombocytopenic purpura(ITP) is one of the common hematologic disorders in children. Bone marrow aspiration(BMA) is often performed in children with acute ITP to rule out leukemia, aplastic anemia or other hematologic diseases. However, whether BMA is needed in children with typical clinical and hematological features of acute ITP have been questioned. This study was performed to examine the proper indication of BMA in acute childhood ITP. MATERIALS AND METHODS: The medical records and BMA reports of children with the provisional diagnosis of acute ITP were reviewed from January 1984 to December 2000. Patients were divided into two groups, one with typical and another with atypical clinical and hematological features of acute ITP. Typical acute ITP group was characterized by the history of previous viral infection, well being appearance, no hepatosplenomegaly, no lymphadenopathy, normal Hb, WBC, neutrophil count and peripheral blood smear except thrombocytopenia. A platelet count of 50x109/L or lower was the cutoff level. RESULTS: Total 120 children with the provisional diagnosis of acute ITP were included. One hundred eighteen of them were confirmed to have acute ITP by BMAs. Of these, 66 had typical and 54 had atypical features. All of typical features and 52 of 54 with atypical features of acute ITP were confirmed to have acute ITP by BMAs. Two patients with atypical features of acute ITP were diagnosed as aplastic anemia and myelodyspalstic syndrome, respectively, by BMAs. CONCLUSION: This study concludes that BMA is not needed for the children with typical features of acute ITP but it is needed for the children with atypical features of acute ITP to rule out other hematologic disorders.