Mechanism underlying severe deficiency of plasma ADAMTS-13 activity in immune thrombotic thrombocytopenic purpura

J Thromb Haemost. 2024 May;22(5):1358-1365. doi: 10.1016/j.jtha.2024.02.003. Epub 2024 Feb 14.

Abstract

Background: Immune-mediated thrombotic thrombocytopenic purpura is caused by autoantibodies against ADAMTS-13, a plasma enzyme that cleaves von Willebrand factor. However, the mechanism resulting in severe deficiency of plasma ADAMTS-13 activity remains controversial.

Objectives: To determine the mechanism of autoantibody-mediated severe deficiency of plasma ADAMTS13 activity in immune-mediated thrombotic thrombocytopenic purpura.

Methods: Fluorescence resonance energy transfer-VWF73 was used to determine plasma ADAMTS-13 activity. Enzyme-linked immunosorbent assay (ELISA) was used to determine anti-ADAMTS-13 immunoglobulin G. ELISA and capillary electrophoresis-based Western blotting were employed to assess plasma ADAMTS-13 antigen.

Results: We showed that plasma ADAMTS-13 antigen levels varied substantially in the samples collected on admission despite all showing plasma ADAMTS-13 activity of <10 IU/dL (or <10% of normal level) using either ELISA or Western blotting. More severe deficiency of plasma ADAMTS-13 antigen (<10%) was detected in admission samples by ELISA than by capillary Western blotting. There was a significant but moderate correlation between plasma ADAMTS-13 activity and ADAMTS-13 antigen by either assay method, suggesting that severe deficiency of plasma ADAMTS-13 activity is not entirely associated with low levels of ADAMTS-13 antigen.

Conclusion: We conclude that severe deficiency of plasma ADAMTS-13 activity primarily resulted from antibody-mediated inhibition, but the accelerated clearance of plasma ADAMTS-13 antigen via immune complexes may also contribute significantly to severe deficiency of plasma ADAMTS-13 activity in a subset of patients with acute immune-mediated thrombotic thrombocytopenic purpura.

Keywords: ADAMTS-13; TTP/HUS; autoantibodies; capillary Western blotting; von Willebrand factor.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • ADAM Proteins* / blood
  • ADAM Proteins* / deficiency
  • ADAM Proteins* / immunology
  • ADAMTS13 Protein* / blood
  • ADAMTS13 Protein* / immunology
  • Adult
  • Aged
  • Autoantibodies* / blood
  • Blotting, Western
  • Enzyme-Linked Immunosorbent Assay*
  • Female
  • Fluorescence Resonance Energy Transfer
  • Humans
  • Immunoglobulin G / blood
  • Male
  • Middle Aged
  • Purpura, Thrombotic Thrombocytopenic* / blood
  • Purpura, Thrombotic Thrombocytopenic* / diagnosis
  • Purpura, Thrombotic Thrombocytopenic* / enzymology
  • Purpura, Thrombotic Thrombocytopenic* / immunology
  • von Willebrand Factor / analysis
  • von Willebrand Factor / metabolism

Substances

  • ADAMTS13 Protein
  • ADAMTS13 protein, human
  • Autoantibodies
  • ADAM Proteins
  • Immunoglobulin G
  • von Willebrand Factor