Marfan syndrome with antineutrophil cytoplasmic antibody-associated systemic vasculitis presenting as severe anaemia and haematuria after the Bentall procedure

Eur J Cardiothorac Surg. 2013 Aug;44(2):379-81. doi: 10.1093/ejcts/ezt071. Epub 2013 Feb 22.

Abstract

One month previously, a 28-year old male underwent an emergency modified Bentall procedure because of Marfan syndrome with acute aortic dissection Stanford Class A. Computed tomography of the chest did not reveal severe graft stenosis of the anastomosis. To explore the cause of anaemia, renal dysfunction and macroscopic haematuria, the patient was tested for antineutrophil cytoplasmic antibody (ANCA)-associated systemic vasculitis (AASV). Antimyeloperoxidase antibodies (MPO)-ANCA and antiproteinase 3 antibodies (PR3)-ANCA were strongly positive. Corticosteroid therapy was applied, followed by cyclophosphamide and azathioprine. In response to treatment, the MPO-ANCA and PR3-ANCA levels gradually decreased, proteinuria was alleviated and haemoglobin levels returned to normal after 6 months. This is the first report to highlight haemolytic anaemia and AASV with Marfan syndrome after surgery for aortic dissection.

Keywords: Anaemia; Antineutrophil cytoplasmic antibody-associated systemic vasculitis; Haematuria; Marfan syndrome.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Anemia / diagnosis*
  • Antibodies, Antineutrophil Cytoplasmic / analysis
  • Aortic Aneurysm / surgery
  • Aortic Dissection / surgery
  • Blood Vessel Prosthesis Implantation / adverse effects
  • Blood Vessel Prosthesis Implantation / methods*
  • Hematuria / diagnosis*
  • Humans
  • Male
  • Marfan Syndrome / blood
  • Marfan Syndrome / diagnosis*
  • Marfan Syndrome / surgery*
  • Systemic Vasculitis / blood
  • Systemic Vasculitis / diagnosis*

Substances

  • Antibodies, Antineutrophil Cytoplasmic