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LRRC58 defines an E3 ubiquitin ligase complex sensitive to cysteine abundance - PubMed

4 hours ago
  • #ubiquitin ligase
  • #cysteine homeostasis
  • #neurodevelopment
  • LRRC58 is identified as a substrate adaptor for a Cullin-RING E3 ubiquitin ligase complex that responds to cysteine levels.
  • In high cysteine conditions, LRRC58 is ubiquitinated and degraded by the proteasome, inhibiting its activity.
  • During cysteine deprivation, LRRC58 stabilizes to target CDO1, a key enzyme in cysteine catabolism, for degradation.
  • CDO1 degradation protects cells from ferroptosis when cysteine is scarce.
  • Mutations in CDO1 linked to human neurodevelopmental disorders produce proteins that resist LRRC58-mediated degradation, highlighting the axis's role in neural development.
  • Structural insights into the CDO1-LRRC58 interaction were validated through saturation mutagenesis, pinpointing critical residues in LRRC58's C-terminus.