{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T20:53:23Z","timestamp":1780433603629,"version":"3.54.1"},"reference-count":93,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2023,7,13]],"date-time":"2023-07-13T00:00:00Z","timestamp":1689206400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100001312","name":"Charles Revson Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001312","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Joy McNicoll Award"},{"DOI":"10.13039\/501100001538","name":"Victoria University of Wellington Doctoral Scholarship","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001538","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,8,31]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Niemann\u2013Pick type C (NP-C) disease is a rare lysosomal storage disease caused by mutations in NPC1 (95% cases) or NPC2 (5% cases). These proteins function together in cholesterol egress from the lysosome, whereby upon mutation, cholesterol and other lipids accumulate causing major pathologies. However, it is not fully understood how cholesterol is transported from NPC1 residing at the lysosomal membrane to the endoplasmic reticulum (ER) and plasma membrane. The yeast ortholog of NPC1, Niemann\u2013Pick type C\u2013related protein-1 (Ncr1), functions similarly to NPC1; when transfected into a mammalian cell lacking NPC1, Ncr1 rescues the diagnostic hallmarks of cholesterol and sphingolipid accumulation. Here, we aimed to identify and characterize protein\u2013protein interactions (PPIs) with the yeast Ncr1 protein. A genome-wide split-ubiquitin membrane yeast two-hybrid (MYTH) protein interaction screen identified 11 ER membrane-localized, full-length proteins interacting with Ncr1 at the lysosomal\/vacuolar membrane. These highlight the importance of ER-vacuole membrane interface and include PPIs with the Cyb5\/Cbr1 electron transfer system, the ceramide synthase complex, and the Sec61\/Sbh1 protein translocation complex. These PPIs were not detected in a sterol auxotrophy condition and thus depend on normal sterol metabolism. To provide biological context for the Ncr1-Cyb5 PPI, a yeast strain lacking this PPI (via gene deletions) exhibited altered levels of sterols and sphingolipids including increased levels of glucosylceramide that mimic NP-C disease. Overall, the results herein provide new physical and genetic interaction models to further use the yeast model of NP-C disease to better understand human NP-C disease.<\/jats:p>","DOI":"10.1093\/genetics\/iyad129","type":"journal-article","created":{"date-parts":[[2023,7,13]],"date-time":"2023-07-13T17:14:18Z","timestamp":1689268458000},"source":"Crossref","is-referenced-by-count":5,"title":["Identification and characterization of protein interactions with the major Niemann\u2013Pick type C disease protein in yeast reveals pathways of therapeutic potential"],"prefix":"10.1093","volume":"225","author":[{"given":"Natalie","family":"Hammond","sequence":"first","affiliation":[{"name":"School of Biological Sciences, Victoria University of Wellington , Wellington 6012 , New Zealand"},{"name":"Centre for Biodiscovery, Victoria University of Wellington , Wellington 6012 , New Zealand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jamie","family":"Snider","sequence":"additional","affiliation":[{"name":"Donnelly Centre, University of Toronto , Toronto, Ontario M5S 3E1 , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Igor","family":"Stagljar","sequence":"additional","affiliation":[{"name":"Donnelly Centre, University of Toronto , Toronto, Ontario M5S 3E1 , Canada"},{"name":"Department of Molecular Genetics, University of Toronto , Toronto, Ontario M5S 1A8 , Canada"},{"name":"Department of Biochemistry, University of Toronto , Toronto, Ontario M5S 1A8 , Canada"},{"name":"Mediterranean Institute for Life Sciences , Me\u0161trovi\u0107evo \u0160etali\u0161te 45, HR-21000 Split , Croatia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kevin","family":"Mitchell","sequence":"additional","affiliation":[{"name":"Callaghan Innovation , Lower Hutt 5040 , New Zealand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kirill","family":"Lagutin","sequence":"additional","affiliation":[{"name":"Callaghan Innovation , Lower Hutt 5040 , New Zealand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew","family":"Jessulat","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Research and Innovation Centre, University of Regina , Regina, Saskatchewan S4S 0A2 , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mohan","family":"Babu","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Research and Innovation Centre, University of Regina , Regina, Saskatchewan S4S 0A2 , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul H","family":"Teesdale-Spittle","sequence":"additional","affiliation":[{"name":"School of Biological Sciences, Victoria University of Wellington , Wellington 6012 , New Zealand"},{"name":"Centre for Biodiscovery, Victoria University of Wellington , Wellington 6012 , New Zealand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey P","family":"Sheridan","sequence":"additional","affiliation":[{"name":"School of Biological Sciences, Victoria University of Wellington , Wellington 6012 , New Zealand"},{"name":"Centre for Biodiscovery, Victoria University of Wellington , Wellington 6012 , New Zealand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephen L","family":"Sturley","sequence":"additional","affiliation":[{"name":"Department of Biology, Barnard College-Columbia University , New York, NY 10027 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew B","family":"Munkacsi","sequence":"additional","affiliation":[{"name":"School of Biological Sciences, Victoria University of Wellington , Wellington 6012 , New Zealand"},{"name":"Centre for Biodiscovery, Victoria University of Wellington , Wellington 6012 , New Zealand"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2023,7,13]]},"reference":[{"issue":"1","key":"2024091719474286900_iyad129-B1","doi-asserted-by":"publisher","first-page":"23","DOI":"10.1002\/jcp.1030410103","article-title":"Anaerobic nutrition of Saccharomyces cerevisiae. 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