Ontology highlight
ABSTRACT:
SUBMITTER: McGrath MJ
PROVIDER: S-EPMC7773396 | biostudies-literature | 2021 Jan
REPOSITORIES: biostudies-literature
McGrath Meagan J MJ Eramo Matthew J MJ Gurung Rajendra R Sriratana Absorn A Gehrig Stefan M SM Lynch Gordon S GS Lourdes Sonia Raveena SR Koentgen Frank F Feeney Sandra J SJ Lazarou Michael M McLean Catriona A CA Mitchell Christina A CA
The Journal of clinical investigation 20210101 1
The regulation of autophagy-dependent lysosome homeostasis in vivo is unclear. We showed that the inositol polyphosphate 5-phosphatase INPP5K regulates autophagic lysosome reformation (ALR), a lysosome recycling pathway, in muscle. INPP5K hydrolyzes phosphatidylinositol-4,5-bisphosphate [PI(4,5)P2] to phosphatidylinositol 4-phosphate [PI(4)P], and INPP5K mutations cause muscular dystrophy by unknown mechanisms. We report that loss of INPP5K in muscle caused severe disease, autophagy inhibition, ...[more]