biostudies-literatureGoto AJapan Agency for Medical Research and DevelopmentJapan Society for the Promotion of Science8576https://www.ebi.ac.uk/biostudies/studies/S-EPMC9368931biostudies-literatureUnknown23(15)Ceramide transport protein (CERT) mediates ceramide transfer from the endoplasmic reticulum to the Golgi for sphingomyelin (SM) biosynthesis. CERT is inactivated by multiple phosphorylation at the serine-repeat motif (SRM), and mutations that impair the SRM phosphorylation are associated with a group of inherited intellectual disorders in humans. It has been suggested that the <i>N</i>-terminal phosphatidylinositol 4-monophosphate [PtdIns(4)P] binding domain and the <i>C</i>-terminal ceramide-transfer domain of CERT physically interfere with each other in the SRM phosphorylated state, thereby repressing the function of CERT; however, it remains unclear which regions in CERT are involved in the SRM phosphorylation-dependent repression of CERT. Here, we identified a previously uncharacterizeInternational journal of molecular sciencesInvolvement of a Cluster of Basic Amino Acids in Phosphorylation-Dependent Functional Repression of the Ceramide Transport Protein CERT.PMC9368931JP20gm0910005j000618K06649Kumagai KEgawa DYamaji TTomishige NGoto AShimasaki KHanada KfalseInvolvement of a Cluster of Basic Amino Acids in Phosphorylation-Dependent Functional Repression of the Ceramide Transport Protein CERT.Ceramide transport protein (CERT) mediates ceramide transfer from the endoplasmic reticulum to the Golgi for sphingomyelin (SM) biosynthesis. CERT is inactivated by multiple phosphorylation at the serine-repeat motif (SRM), and mutations that impair the SRM phosphorylation are associated with a group of inherited intellectual disorders in humans. It has been suggested that the <i>N</i>-terminal phosphatidylinositol 4-monophosphate [PtdIns(4)P] binding domain and the <i>C</i>-terminal ceramide-transfer domain of CERT physically interfere with each other in the SRM phosphorylated state, thereby repressing the function of CERT; however, it remains unclear which regions in CERT are involved in the SRM phosphorylation-dependent repression of CERT. Here, we identified a previously uncharacterize2022-01-01T00:00:00Z2022 Aug2025-04-04T10:44:52.347Z2025-02-19T01:55:43.923ZS-EPMC93689313595571910.3390/ijms23158576