{"doi":"10.1083/jcb.202111095","title":"Vps13-like proteins provide phosphatidylethanolamine for GPI anchor synthesis in the ER","abstract":"Glycosylphosphatidylinositol (GPI) is a glycolipid membrane anchor found on surface proteins in all eukaryotes. It is synthesized in the ER membrane. Each GPI anchor requires three molecules of ethanolamine phosphate (P-Etn), which are derived from phosphatidylethanolamine (PE). We found that efficient GPI anchor synthesis in Saccharomyces cerevisiae requires Csf1; cells lacking Csf1 accumulate GPI precursors lacking P-Etn. Structure predictions suggest Csf1 is a tube-forming lipid transport protein like Vps13. Csf1 is found at contact sites between the ER and other organelles. It interacts with the ER protein Mcd4, an enzyme that adds P-Etn to nascent GPI anchors, suggesting Csf1 channels PE to Mcd4 in the ER at contact sites to support GPI anchor biosynthesis. CSF1 has orthologues in Caenorhabditis elegans (lpd-3) and humans (KIAA1109/TWEEK); mutations in KIAA1109 cause the autosomal recessive neurodevelopmental disorder Alkuraya-Kučinskas syndrome. Knockout of lpd-3 and knockdown of KIAA1109 reduced GPI-anchored proteins on the surface of cells, suggesting Csf1 orthologues in human cells support GPI anchor biosynthesis.","journal":"The Journal of Cell Biology","year":2022,"id":235551,"datarank":0.0,"base_score":0.0,"endowment":0.0,"self_citation_contribution":0.0,"citation_network_contribution":0.0,"self_endowment_contribution":0.0,"citer_contribution":0.0,"corpus_percentile":null,"corpus_rank":null,"citation_count":78,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":0.9528,"is_data_producer":false,"deposit_databanks":null,"is_oa":true,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":"2022-01-01","fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":466369,"name":"Fawn B. Whittle","orcid":null,"position":1,"is_corresponding":false},{"id":759641,"name":"Jerry Yang","orcid":"0009-0009-8581-8314","position":2,"is_corresponding":false},{"id":315046,"name":"Xiaofei Bai","orcid":"0000-0001-8179-8162","position":3,"is_corresponding":false},{"id":855208,"name":"Jessica Diarra","orcid":null,"position":4,"is_corresponding":false},{"id":291109,"name":"Subhrajit Banerjee","orcid":"0000-0003-1474-8864","position":5,"is_corresponding":false},{"id":847844,"name":"Tim P. Levine","orcid":"0000-0002-7231-0775","position":6,"is_corresponding":false},{"id":315049,"name":"Andy Golden","orcid":"0000-0002-8599-2031","position":7,"is_corresponding":false},{"id":444294,"name":"William A. Prinz","orcid":"0000-0002-9053-2398","position":8,"is_corresponding":false},{"id":689011,"name":"Alexandre Toulmay","orcid":"0009-0009-5383-3632","position":0,"is_corresponding":true}],"reference_count":53,"raw_metadata":null,"created_at":"2026-07-19T00:21:53.333008Z","pmid":"35015055","pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}