Murine Surf4 is essential for early embryonic development

PLoS One. 2020 Jan 24;15(1):e0227450. doi: 10.1371/journal.pone.0227450. eCollection 2020.

Abstract

Newly synthesized proteins co-translationally inserted into the endoplasmic reticulum (ER) lumen may be recruited into anterograde transport vesicles by their association with specific cargo receptors. We recently identified a role for the cargo receptor SURF4 in facilitating the secretion of PCSK9 in cultured cells. To examine the function of SURF4 in vivo, we used CRISPR/Cas9-mediated gene editing to generate mice with germline loss-of-function mutations in Surf4. Heterozygous Surf4+/- mice exhibit grossly normal appearance, behavior, body weight, fecundity, and organ development, with no significant alterations in circulating plasma levels of PCSK9, apolipoprotein B, or total cholesterol, and a detectable accumulation of intrahepatic apoliprotein B. Homozygous Surf4-/- mice exhibit embryonic lethality, with complete loss of all Surf4-/- offspring between embryonic days 3.5 and 9.5. In contrast to the milder murine phenotypes associated with deficiency of known SURF4 cargoes, the embryonic lethality of Surf4-/- mice implies the existence of additional SURF4 cargoes or functions that are essential for murine early embryonic development.

Publication types

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

MeSH terms

  • Alleles
  • Animals
  • Apolipoproteins B / blood
  • Apolipoproteins B / metabolism
  • CRISPR-Cas Systems / genetics
  • Cholesterol / blood
  • Embryonic Development*
  • Gene Editing
  • Heterozygote
  • Membrane Proteins / genetics*
  • Membrane Proteins / metabolism
  • Mice
  • Mice, Knockout
  • Proprotein Convertase 9 / blood
  • Proprotein Convertase 9 / metabolism

Substances

  • Apolipoproteins B
  • Membrane Proteins
  • Surf4 protein, mouse
  • Cholesterol
  • Proprotein Convertase 9