Fulvene-5 potently inhibits NADPH oxidase 4 and blocks the growth of endothelial tumors in mice

J Clin Invest. 2009 Aug;119(8):2359-65. doi: 10.1172/JCI33877. Epub 2009 Jul 13.

Abstract

Hemangiomas are the most common type of tumor in infants. As they are endothelial cell-derived neoplasias, their growth can be regulated by the autocrine-acting Tie2 ligand angiopoietin 2 (Ang2). Using an experimental model of human hemangiomas, in which polyoma middle T-transformed brain endothelial (bEnd) cells are grafted subcutaneously into nude mice, we compared hemangioma growth originating from bEnd cells derived from wild-type, Ang2+/-, and Ang2-/- mice. Surprisingly, Ang2-deficient bEnd cells formed endothelial tumors that grew rapidly and were devoid of the typical cavernous architecture of slow-growing Ang2-expressing hemangiomas, while Ang2+/- cells were greatly impaired in their in vivo growth. Gene array analysis identified a strong downregulation of NADPH oxidase 4 (Nox4) in Ang2+/- cells. Correspondingly, lentiviral silencing of Nox4 in an Ang2-sufficient bEnd cell line decreased Ang2 mRNA levels and greatly impaired hemangioma growth in vivo. Using a structure-based approach, we identified fulvenes as what we believe to be a novel class of Nox inhibitors. We therefore produced and began the initial characterization of fulvenes as potential Nox inhibitors, finding that fulvene-5 efficiently inhibited Nox activity in vitro and potently inhibited hemangioma growth in vivo. In conclusion, the present study establishes Nox4 as a critical regulator of hemangioma growth and identifies fulvenes as a potential class of candidate inhibitor to therapeutically interfere with Nox function.

Publication types

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

MeSH terms

  • Angiopoietin-2 / physiology
  • Animals
  • Cyclopentanes / pharmacology*
  • Endothelial Cells / metabolism
  • Enzyme Inhibitors / pharmacology*
  • Hemangioma / drug therapy*
  • Hemangioma / pathology
  • Intracellular Signaling Peptides and Proteins
  • Mice
  • NADPH Oxidase 4
  • NADPH Oxidases / antagonists & inhibitors*
  • NADPH Oxidases / genetics
  • NADPH Oxidases / physiology
  • Proteins / genetics
  • Vascular Endothelial Growth Factor A / antagonists & inhibitors

Substances

  • Angiopoietin-2
  • Cyclopentanes
  • Enzyme Inhibitors
  • Intracellular Signaling Peptides and Proteins
  • Nrarp protein, mouse
  • Proteins
  • Vascular Endothelial Growth Factor A
  • fulvene-5
  • NADPH Oxidase 4
  • NADPH Oxidases
  • Nox4 protein, mouse