RAP-011, an activin receptor ligand trap, increases hemoglobin concentration in hepcidin transgenic mice

Am J Hematol. 2015 Jan;90(1):8-14. doi: 10.1002/ajh.23856. Epub 2014 Oct 25.

Abstract

Over expression of hepcidin antimicrobial peptide is a common feature of iron-restricted anemia in humans. We investigated the erythroid response to either erythropoietin or RAP-011, a "murinized" ortholog of sotatercept, in C57BL/6 mice and in hepcidin antimicrobial peptide 1 over expressing mice. Sotatercept, a soluble, activin receptor type IIA ligand trap, is currently being evaluated for the treatment of anemias associated with chronic renal disease, myelodysplastic syndrome, β-thalassemia, and Diamond Blackfan anemia and acts by inhibiting signaling downstream of activin and other Transforming Growth Factor-β superfamily members. We found that erythropoietin and RAP-011 increased hemoglobin concentration in C57BL/6 mice and in hepcidin antimicrobial peptide 1 over expressing mice. While erythropoietin treatment depleted splenic iron stores in C57BL/6 mice, RAP-011 treatment did not deplete splenic iron stores in mice of either genotype. Bone marrow erythroid progenitors from erythropoietin-treated mice exhibited iron-restricted erythropoiesis, as indicated by increased median fluorescence intensity of transferrin receptor immunostaining by flow cytometry. In contrast, RAP-011-treated mice did not exhibit the same degree of iron-restricted erythropoiesis. In conclusion, we have demonstrated that RAP-011 can improve hemoglobin concentration in hepcidin antimicrobial peptide 1 transgenic mice. Our data support the hypothesis that RAP-011 has unique biologic effects which prevent or circumvent depletion of mouse splenic iron stores. RAP-011 may, therefore, be an appropriate therapeutic for trials in human anemias characterized by increased expression of hepcidin antimicrobial peptide and iron-restricted erythropoiesis.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Activin Receptors, Type II / chemistry
  • Animals
  • Biological Transport
  • Blood Cell Count
  • Erythroid Precursor Cells / drug effects
  • Erythropoiesis / drug effects*
  • Erythropoietin / pharmacology
  • Female
  • Hemoglobins / analysis*
  • Hepcidins / genetics*
  • Immunoglobulin G / chemistry
  • Iron / blood
  • Iron / metabolism*
  • Ligands
  • Mice, Inbred C57BL
  • Mice, Transgenic
  • Recombinant Fusion Proteins / pharmacology*
  • Spleen / metabolism

Substances

  • ACE-011
  • ActRIIA-mIgG2aFc fusion protein
  • Hamp protein, mouse
  • Hemoglobins
  • Hepcidins
  • Immunoglobulin G
  • Ligands
  • Recombinant Fusion Proteins
  • Erythropoietin
  • Iron
  • Activin Receptors, Type II
  • activin receptor type II-A