Mice with defects in HB-EGF ectodomain shedding show severe developmental abnormalities

J Cell Biol. 2003 Nov 10;163(3):469-75. doi: 10.1083/jcb.200307035. Epub 2003 Nov 3.

Abstract

Heparin-binding EGF-like growth factor (HB-EGF) is first synthesized as a membrane-anchored form (proHB-EGF), and its soluble form (sHB-EGF) is released by ectodomain shedding from proHB-EGF. To examine the significance of proHB-EGF processing in vivo, we generated mutant mice by targeted gene replacement, expressing either an uncleavable form (HBuc) or a transmembrane domain-truncated form (HBdeltatm) of the molecule. HB(uc/uc) mice developed severe heart failure and enlarged heart valves, phenotypes similar to those in proHB-EGF null mice. On the other hand, mice carrying HBdeltatm exhibited severe hyperplasia in both skin and heart. These results indicate that ectodomain shedding of proHB-EGF is essential for HB-EGF function in vivo, and that this process requires strict control.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Cell Surface Extensions / genetics
  • Cell Surface Extensions / metabolism
  • Epidermal Growth Factor / deficiency*
  • Epidermal Growth Factor / genetics
  • Gene Targeting
  • Heart Defects, Congenital / genetics*
  • Heart Defects, Congenital / metabolism
  • Heart Defects, Congenital / pathology
  • Heart Valves / abnormalities
  • Heart Valves / pathology
  • Heparin-binding EGF-like Growth Factor
  • Hyperplasia / genetics*
  • Hyperplasia / metabolism
  • Hyperplasia / pathology
  • Intercellular Signaling Peptides and Proteins
  • Mice
  • Mice, Mutant Strains
  • Mutation / genetics
  • Protein Processing, Post-Translational / genetics
  • Protein Structure, Tertiary / genetics
  • Skin Abnormalities / genetics*
  • Skin Abnormalities / metabolism
  • Skin Abnormalities / pathology
  • Solubility

Substances

  • Hbegf protein, mouse
  • Heparin-binding EGF-like Growth Factor
  • Intercellular Signaling Peptides and Proteins
  • Epidermal Growth Factor