2015
DOI: 10.1242/bio.013334
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The lysosomal enzyme receptor protein (LERP) is not essential, but is implicated in lysosomal function inDrosophila melanogaster

Abstract: The lysosomal enzyme receptor protein (LERP) of Drosophila melanogaster is the ortholog of the mammalian cation-independent mannose 6-phosphate (Man 6-P) receptor, which mediates trafficking of newly synthesized lysosomal acid hydrolases to lysosomes. However, flies lack the enzymes necessary to make the Man 6-P mark, and the amino acids implicated in Man 6-P binding by the mammalian receptor are not conserved in LERP. Thus, the function of LERP in sorting of lysosomal enzymes to lysosomes in Drosophila is unc… Show more

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Cited by 18 publications
(9 citation statements)
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“…Without recycling of CI-M6PR, lysosomal hydrolases are not efficiently delivered, resulting in lysosomal dysfunction ( McMillan et al, 2017 ). To test whether lysosomal dysfunction results in accumulation of mucin cargo proteins within endosomes, we looked for retention of mucins in loss-of-function Lerp (the fly ortholog of CI-M6PR ) mutant cells ( Hasanagic et al, 2015 ). Importantly, Lerp mutant salivary glands did not contain any mucin cargo proteins at the onset of metamorphosis ( …”
Section: Resultsmentioning
confidence: 99%
“…Without recycling of CI-M6PR, lysosomal hydrolases are not efficiently delivered, resulting in lysosomal dysfunction ( McMillan et al, 2017 ). To test whether lysosomal dysfunction results in accumulation of mucin cargo proteins within endosomes, we looked for retention of mucins in loss-of-function Lerp (the fly ortholog of CI-M6PR ) mutant cells ( Hasanagic et al, 2015 ). Importantly, Lerp mutant salivary glands did not contain any mucin cargo proteins at the onset of metamorphosis ( …”
Section: Resultsmentioning
confidence: 99%
“…Notably, the single MPR orthologue identified in D . melanogaster , named lysosomal enzyme receptor protein (LERP) [ 61 ], lacks the residues critical for mannose 6-phosphate binding and it contributes only moderately to lysosomal enzyme targeting [ 62 ]. This may explain why two of the key lysine residues (K183 and K214), and the tyrosine one, are not conserved in AtAGA and LhAGA.…”
Section: Discussionmentioning
confidence: 99%
“…Without recycling of CI-M6PR, lysosomal hydrolases are not efficiently delivered, resulting in lysosomal dysfunction (McMillan et al, 2017). To test whether lysosomal dysfunction results in accumulation of mucin cargo proteins within endosomes, we looked for retention of mucins in loss-of-function Lerp (the fly ortholog of CI-M6PR) mutant cells (Hasanagic et al, 2015).…”
Section: Mistargeting Of Secretory Cargo Proteins To Endosomes Beginsmentioning
confidence: 99%