Literature DB >> 17344464

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

Jan-Inge Henter1, Jonas Karlén.   

Abstract

A 10-year-old girl with steroid-resistant 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 degrees C), white blood cell count 0.4 x 10(9)/L, absolute neutrophil count 0.1 x 10(9)/L and platelets 114 x 10(9)/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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Year:  2007        PMID: 17344464     DOI: 10.1182/blood-2007-02-065805

Source DB:  PubMed          Journal:  Blood        ISSN: 0006-4971            Impact factor:   22.113


  18 in total

1.  Agranulocytosis due to deferiprone: a case report with cytomorphological and functional bone marrow examination.

Authors:  Nicoletta Masera; Luisa Tavecchia; Daniela Valentina Longoni; Oscar Maglia; Andrea Biondi; Giuseppe Masera
Journal:  Blood Transfus       Date:  2011-07-18       Impact factor: 3.443

Review 2.  An update on iron chelation therapy.

Authors:  Erika Poggiali; Elena Cassinerio; Laura Zanaboni; Maria Domenica Cappellini
Journal:  Blood Transfus       Date:  2012-06-27       Impact factor: 3.443

Review 3.  World health dilemmas: Orphan and rare diseases, orphan drugs and orphan patients.

Authors:  Christina N Kontoghiorghe; Nicholas Andreou; Katerina Constantinou; George J Kontoghiorghes
Journal:  World J Methodol       Date:  2014-09-26

Review 4.  How I treat Diamond-Blackfan anemia.

Authors:  Adrianna Vlachos; Ellen Muir
Journal:  Blood       Date:  2010-07-22       Impact factor: 22.113

5.  The Stomatological Complications of Diamond-Blackfan Anemia: A Case Report.

Authors:  Rita Fabiane Teixeira Gomes; Maria Cristina Munerato
Journal:  Clin Med Res       Date:  2016-02-10

6.  Agranulocyosis in Beta Thalassemia Major Patients treated with Oral Iron Chelating Agent (Deferiprone).

Authors:  Yasser Wali; Azza Al Shidhani; Shahina Daar
Journal:  Oman Med J       Date:  2008-10

Review 7.  Diamond Blackfan anemia treatment: past, present, and future.

Authors:  Anupama Narla; Adrianna Vlachos; David G Nathan
Journal:  Semin Hematol       Date:  2011-04       Impact factor: 3.851

8.  Safety and efficacy of iron chelation therapy with deferiprone in patients with transfusion-dependent thalassemia.

Authors:  Saumya S Jamuar; Angeline H M Lai
Journal:  Ther Adv Hematol       Date:  2012-10

9.  Agranulocytosis with deferiprone treatment of superficial siderosis.

Authors:  Nikhil Huprikar; Marisa Gossweiler; Maureen Callaghan; Paul Bunge
Journal:  BMJ Case Rep       Date:  2013-08-07

Review 10.  Critical Issues in Diamond-Blackfan Anemia and Prospects for Novel Treatment.

Authors:  Hojun Li; Harvey F Lodish; Colin A Sieff
Journal:  Hematol Oncol Clin North Am       Date:  2018-06-05       Impact factor: 3.722

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