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Leucine aminopeptidase LyLAP enables lysosomal degradation of membrane proteins

Aakriti Jain, Isaac P. Heremans, Gilles Rademaker, Tyler C. Detomasi, Peter J. Rohweder, Dashiell Anderson, Zuopeng Zhang, Grace A. Hernandez, Suprit Gupta, Teresa von Linde, Mike Lange, Martina Spacci, Jiayi Luo, Y. Rose Citron, James A. Olzmann, David W. Dawson, Charles S. Craik, Guido T. Bommer, Rushika M. Perera, Roberto Zoncu

2025Science11 citationsDOIOpen Access PDF

Abstract

Breakdown of every transmembrane protein trafficked to lysosomes requires proteolysis of their hydrophobic helical transmembrane domains. Combining lysosomal proteomics with functional genomic datasets, we identified lysosomal leucine aminopeptidase (LyLAP; formerly phospholipase B domain-containing 1) as the hydrolase most tightly associated with elevated endocytosis. Untargeted metabolomics and biochemical reconstitution demonstrated that LyLAP is a processive monoaminopeptidase with preference for amino-terminal leucine. This activity was necessary and sufficient for the breakdown of hydrophobic transmembrane domains. LyLAP was up-regulated in pancreatic ductal adenocarcinoma (PDA), which relies on macropinocytosis for nutrient uptake. In PDA cells, LyLAP ablation led to the buildup of undigested hydrophobic peptides, lysosomal membrane damage, and growth inhibition. Thus, LyLAP enables lysosomal degradation of membrane proteins and protects lysosomal integrity in highly endocytic cancer cells.

Topics & Concepts

AminopeptidaseTransmembrane proteinPinocytosisEndocytic cycleEndocytosisLeucineProteolysisBiochemistryEndosomeCell biologyTransmembrane domainMembrane proteinLysosomeBiologyChemistryAmino acidMembraneEnzymeIntracellularCellReceptorCellular transport and secretionPeptidase Inhibition and AnalysisEndoplasmic Reticulum Stress and Disease