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 Blood, 15 June 2007, Vol. 109, No. 12, pp. 5157-5159.
Prepublished online as a Blood First Edition Paper on March 7, 2007; DOI 10.1182/blood-2007-02-065805.

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CLINICAL TRIALS AND OBSERVATIONS

Brief Report

Fatal agranulocytosis after deferiprone therapy in a child with Diamond-Blackfan anemia

Jan-Inge Henter1, and Jonas Karlén1

1 Childhood Cancer Research Unit, Department of Woman and Child Health, Karolinska University Hospital, Karolinska Institutet, Stockholm, Sweden

A 10-year-old girl with steroid-resistent Diamond-Blackfan anemia (DBA) developed agranulocytosis 9 weeks after chelation with deferiprone was initiated (45 mg/kg daily, 60% of recommended dose) in addition to her ordinary deferoxamine therapy. The blood counts, checked weekly, dropped markedly between weeks 8 and 9. She rapidly developed a septicemia and was admitted with high fever (40.9°C), white blood cell count 0.4 x 109/L, absolute neutrophil count 0.1 x 109/L and platelets 114 x 109/L. She was administered broad spectrum antibiotics, G-CSF (10 microgram/kg daily) and corticosteroids but remained neutropenic and died 6 weeks after admission. Bone marrow examination day 23 revealed areas with low cellularity (around 30%), but also areas with infiltrates of T cells; granulopoiesis and erythropoiesis were scarce. We conclude that weekly neutrophil monitoring is not sufficient to avoid fatal agranulocytosis. We suggest that deferiprone not be prescribed to DBA patients unless the clinical indications are particularly strong, and that the risk of agranulocytosis in thalassemia patients be carefully considered.


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