J Korean Med Sci.  2010 Nov;25(11):1676-1679. 10.3346/jkms.2010.25.11.1676.

Improvement in Erythropoieis-stimulating Agent-induced Pure Red-cell Aplasia by Introduction of Darbepoetin-alpha When the Anti-erythropoietin Antibody Titer Declines Spontaneously

• Lee H
• Yang J
• Kim H
• Kwon JW
• Oh KH
• Joo KW
• Kim YS
• Ahn C
• Han JS
• Kim S

• KMID: 1779243
• DOI: http://doi.org/10.3346/jkms.2010.25.11.1676

Abstract

Anti-erythropoietin antibodies usually cross-react with all kinds of recombinant erythropoietins; therefore, erythropoiesis-stimulating agent (ESA)-induced pure red-cell aplasia (PRCA) is not rescued by different ESAs. Here, we present a case of ESA-induced PRCA in a 36-yr-old woman with chronic kidney disease, whose anemic condition improved following reintroduction of darbepoetin-alpha. The patient developed progressive, severe anemia after the use of erythropoietin-alpha. As the anemia did not improve after the administration of either other erythropoietin-alpha products or erythropoietin-beta, all ESAs were discontinued. Oxymetholone therapy failed to improve the transfusion-dependent anemia and a rechallenge with ESAs continuously failed to obtain a sustained response. However, her anemia improved following reintroduction of darbepoetin-alpha at 3 yr after the initial diagnosis. Interestingly, anti-erythropoietin antibodies were still detectable, although their concentration was too low for titration. In conclusion, darbepoetin-alpha can improve ESA-induced PRCA when the anti-erythropoietin antibody titer declines and its neutralizing capacity is lost.

Keyword

Red-Cell Aplasia, Pure; Kidney Failure, Chronic; Erythropoietin, Recombinant; Darbepoetin-alfa

MeSH Terms

Adult
Anemia/drug therapy/etiology
Antibodies/*blood/immunology
Bone Marrow Cells/pathology
Drug Hypersensitivity/immunology
Erythropoietin/*analogs & derivatives/therapeutic use
Erythropoietin, Recombinant/adverse effects/*immunology/therapeutic use
Female
Glomerulonephritis, IGA/complications
Hematinics/adverse effects/immunology/*therapeutic use
Humans
Kidney Failure, Chronic/complications
Oxymetholone/therapeutic use
Red-Cell Aplasia, Pure/chemically induced/*drug therapy/immunology

Figure

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  Fig. 1 Bone marrow biopsy findings. (A) Bone marrow section, The cellularity is 0-20% which is hypocellular for age. Trilineage hematopoiesis is markedly decreased, and the decrease of erythropoiesis is remarkable. Plasma cells, lymphocytes and eosinophils are unremarkable. Foreign cells and granulomata are absent, H&E stained, ×100. (B) A megakaryocyte and myeloid precursor cells are observed, but no erythroid precursor cells are observed, H&E stained, ×1,000.

• 

  Fig. 2 Clinical course of the patient by time. The gray line is a schematic representation of the patient's hemoglobin (Hb) levels (conversion factor from g/dL to g/L, ×10) from June 2000 to April 2009. One red triangle means transfusion of two units of packed red blood cells. Hemodialysis (HD) was initiated in January 2002. Oxymetholone therapy was given from June 2003 to July 2003. The double-headed arrows below the graph indicate the durations of erythropoiesis-stimulating agent use, represented by the following colors: green arrow, Epokine; blue arrows, Espogen; violet arrows, Recormon; yellow arrows, Aranesp.

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