Acquired von Willebrand Syndrome Hiding Inherited von Willebrand Disease Can Explain Severe Bleeding in Patients With Aortic Stenosis

Arterioscler Thromb Vasc Biol. 2020 Sep;40(9):2187-2194. doi: 10.1161/ATVBAHA.120.314656. Epub 2020 Jul 9.

Abstract

Objective: Aortic stenosis may be complicated by an acquired von Willebrand syndrome that rarely causes significant bleeding, raising the question of why it does so in a few cases. To seek an explanation, we studied 5 severe bleeder aortic stenosis patients in a cohort of 49 patients, using the flowchart for inherited von Willebrand disease. Approach and Results: All 5 patients were lacking in large and intermediate VWF (von Willebrand factor) multimers, 3 had reduced plasma and platelet VWF levels, and none showed PFA100 closure. Two patients (those with most multimers missing) also had a short VWF half-life. Genetic analyses on the 3 patients with reduced platelet VWF levels revealed that one carried both the c.1164C>G and the c.7880G>A mutations, and another carried the c.3390C>T mutation, while the third had one of the 2 VWF alleles relatively less expressed than the other (25% versus 75%). No genetic alterations emerged in the other 2 patients. Successful replacement of the stenotic aortic valve, performed in the 2 patients with VWF mutations, did not correct their abnormal VWF multimer picture-unlike what happened in the aortic stenosis patients without bleeding symptoms.

Conclusions: Our findings suggest that acquired von Willebrand syndrome can develop in patients with hitherto-undiagnosed inherited von Willebrand disease. Since von Willebrand disease is the most common bleeding disorder, this possibility should be considered in aortic stenosis patients-especially those with a more severe bleeding history and more disrupted VWF laboratory patterns-because they risk hemorrhage during aortic valve replacement.

Keywords: acquired von Willebrand syndrome; aortic valve; aortic valve stenosis; mutation; von Willebrand diseases; von Willebrand factor.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aged
  • Aged, 80 and over
  • Aortic Valve Stenosis / complications
  • Aortic Valve Stenosis / diagnostic imaging
  • Aortic Valve Stenosis / surgery*
  • Biomarkers / blood
  • Blood Vessel Prosthesis Implantation / adverse effects*
  • Case-Control Studies
  • Female
  • Genetic Predisposition to Disease
  • Half-Life
  • Hemostasis* / genetics
  • Humans
  • Male
  • Mutation
  • Phenotype
  • Postoperative Hemorrhage / blood
  • Postoperative Hemorrhage / diagnosis
  • Postoperative Hemorrhage / etiology*
  • Predictive Value of Tests
  • Protein Multimerization
  • Protein Stability
  • Proteolysis
  • Risk Assessment
  • Risk Factors
  • Severity of Illness Index
  • Time Factors
  • Treatment Outcome
  • von Willebrand Diseases / blood
  • von Willebrand Diseases / complications*
  • von Willebrand Diseases / diagnosis
  • von Willebrand Diseases / genetics
  • von Willebrand Factor / genetics
  • von Willebrand Factor / metabolism*

Substances

  • Biomarkers
  • von Willebrand Factor