American Journal of Hematology 77:299–302 (2004)
Rapid Irreversible Encephalopathy Associated With Anti-D Immune Globulin Treatment for Idiopathic Thrombocytopenic Purpura Kenneth Christopher,1* Clare Horkan,2 Ilie T. Barb,3 Christian Arbelaez,4 Travis A. Hodgdon,5 and Paul C. Yodice5 1
Department of Medicine, Renal Division, Brigham and Women’s Hospital, Harvard Medical School, Boston, Massachusetts 2 Department of Radiology, Beth Israel Deaconess Hospital, Harvard Medical School, Boston, Massachusetts 3 Department of Medicine, Division of Cardiovascular Medicine, Brigham and Women’s Hospital, Harvard Medical School, Boston, Massachusetts 4 Department of Emergency Medicine, Rhode Island Hospital, Brown Medical School, Providence, Rhode Island 5 Department of Medicine, Division of Critical Care Medicine, The Miriam Hospital, Brown Medical School, Providence, Rhode Island
Intravenous Rho (D) immune globulin (IV RhIG, WinRho SDF) has been shown to be a safe treatment for idiopathic thrombocytopenic purpura (ITP). Common side effects of IV RhIG include mild hemolysis, febrile reaction, and headache. Significant hemolysis with renal impairment following IV RhIG has been reported. We report a case of irreversible encephalopathy 48 hr following an infusion of IV RhIG for treatment of ITP. Am. J. Hematol. 77:299–302, 2004. ª 2004 Wiley-Liss, Inc. Key words: immune globulin; encephalopathy
INTRODUCTION
Intravenous Rho (D) immune globulin (IV RhIG, WinRho SDF; Nabi, Boca Raton, FL) has been shown to be a safe treatment for idiopathic thrombocytopenic purpura (ITP) [1]. The mechanism of IV RhIG in the treatment of ITP is not completely understood. In the treatment of ITP, IV RhIG is given to D-positive patients to coat the erythrocytes. It is postulated that in nonsplenectomized patients, splenic macrophage Fc receptors are bound by IV RhIG-coated erythrocytes, allowing antibody-coated platelets to escape the reticuloendothelial system [2]. The mechanism of IV RhIG function is likely related to sequestration of antibody-sensitized red cells. In an animal model of ITP, administration of anti-CD24 IgG antibodies ameliorates immune thrombocytopenia and blocks reticuloendothelial system function [3]. In humans, IV RhIG is shown in to increase platelet counts to greater than 20 103/mL2 in over 70% of D positive nonsplenectomized patients [4]. Common side effects of IV RhIG include mild hemolysis, febrile reaction, and headache. Significant hemolysis ª 2004 Wiley-Liss, Inc.
with renal impairment following IV RhIG has been reported [5]. To our knowledge, this is the first report of irreversible encephalopathy 48 hr following an infusion of IV RhIG for ITP. CASE REPORT
A 62-year-old male with a history of coronary artery disease presented to his physician with easy bruising. On evaluation, the patient was found to have platelet count of 27 103/mm3 and hemoglobin of 15 g/dL. The patient was subsequently diagnosed with ITP. Over the next 5 weeks, the patient was treated with prednisone 80 mg QD followed by *Correspondence to: Kenneth Christopher, M.D., Laboratory of Molecular Immunology, Brigham and Women’s Hospital, PBB170, 75 Francis Street, Boston, MA 02115. E-mail:
[email protected] Received for publication 26 July 2003; Accepted 19 May 2004 Published online in Wiley InterScience (www.interscience.wiley.com). DOI: 10.1002/ajh.20189
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Case Report: Christopher et al.
danazol 400 mg QD and a prednisone taper without sustained increase in the platelet count. IV RhIG therapy for refractory ITP was started for a platelet count of 10 103/mm3. The patient was D antigen positive. The patient received 4.98 mg IV RhIG (50 mg/kg) over 6 min. During and immediately following the infusion, the patient felt well. One hour following the infusion, the patient presented to the emergency department with general malaise, chills, and low back pain radiating to the bilateral inguinal folds. Significant laboratory data on presentation to the emergency department showed an LDH of 1018 IU/ L (normal 296–558 IU/l), total bilirubin of 1.9 mg/dL (normal
