Pure red cell aplasia associated with imatinib-treated FIP1L1-PDGFRA positive chronic eosinophilic leukemia

Intern Med. 2010;49(12):1195-200. doi: 10.2169/internalmedicine.49.3178. Epub 2010 Jun 15.

Abstract

A 28-year-old man with marked eosinophilia is described. FIP1L1/PDGFRA mRNA showed multiple alternatively-spliced fusion transcripts. Sequencing analysis showed that the deduced DNA breakpoints were intron 10 in the FIP1L1 gene and exon 12 in the PDGFRA gene. Then, a diagnosis of chronic eosinophilic leukemia (CEL) was made. Whereas the response to the treatments with prednisolone and hydroxyurea were unsatisfactory, treatment with imatinib showed a rapid decrease of eosinophils. The hemoglobin level also dropped and bone marrow examination showed pure red cell aplasia. Continued administration of very low dose imatinib (100 mg every 5 days) led to and maintained complete molecular remission, with good tolerability.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Benzamides
  • Chronic Disease
  • Humans
  • Hypereosinophilic Syndrome / complications
  • Hypereosinophilic Syndrome / drug therapy
  • Hypereosinophilic Syndrome / genetics*
  • Imatinib Mesylate
  • Male
  • Oncogene Proteins, Fusion / biosynthesis*
  • Oncogene Proteins, Fusion / genetics
  • Piperazines / therapeutic use*
  • Pyrimidines / therapeutic use*
  • Receptor, Platelet-Derived Growth Factor alpha / biosynthesis*
  • Receptor, Platelet-Derived Growth Factor alpha / genetics
  • Red-Cell Aplasia, Pure / drug therapy
  • Red-Cell Aplasia, Pure / etiology
  • Red-Cell Aplasia, Pure / genetics*
  • mRNA Cleavage and Polyadenylation Factors / biosynthesis*
  • mRNA Cleavage and Polyadenylation Factors / genetics

Substances

  • Benzamides
  • FIP1L1 protein, human
  • Oncogene Proteins, Fusion
  • Piperazines
  • Pyrimidines
  • mRNA Cleavage and Polyadenylation Factors
  • Imatinib Mesylate
  • FIP1L1-PDGFRA fusion protein, human
  • Receptor, Platelet-Derived Growth Factor alpha