140619 20240430120009.0 10.1038/s41420-019-0158-6 doi pmid:30886743 pmid pmc:PMC6411869 pmc altmetric:57930719 altmetric DZNE-2020-06941 English 610 Schwintzer, Lukas P:(DE-2719)2810987 0 First author dzne TRIAD3/RNF216 E3 ligase specifically synthesises K63-linked ubiquitin chains and is inactivated by mutations associated with Gordon Holmes syndrome. London 2019 Nature Publishing Group online Crossref Springer Science and Business Media LLC 2019-03-11 print Crossref Springer Science and Business Media LLC 2019-12-01 article DRIVER Output Types/Journal article DataCite Journal Article journal journal PUB:(DE-HGF)16 1714468365_8221 PUB:(DE-HGF) ARTICLE BibTeX JOURNAL_ARTICLE ORCID Journal Article 0 EndNote TRIAD3/RNF216 is a ubiquitin ligase of the RING-in-between-RING family. Recent publications identified TRIAD3 mutations in patients with neurological diseases, including Gordon Holmes syndrome and Huntington-like disorder. To understand the functional relevance of these disease-associated mutations, we have tested the ubiquitin ligase activity of mutated TRIAD3 in vitro. Several of these point mutations completely abrogated TRIAD3's catalytic activity. Using mass spectrometry, we identified new TRIAD3-interacting proteins/substrates from mouse brain lysate, which provide a new link between TRIAD3 and processes involving clathrin-mediated endocytosis. Strikingly, we found that TRIAD3 synthesises specifically lysine-63 (K63)-linked poly-ubiquitin chains in vitro, a chain type that usually plays a role in mediating signalling events rather than triggering proteasomal degradation. Therefore, this finding is of great importance to further understand TRIAD3's cellular role and loss-of-function in disease. 341 - Molecular Signaling (POF3-341) G:(DE-HGF)POF3-341 POF3-341 POF III 0 2019-03-11 Crossref https://creativecommons.org/licenses/by/4.0 Dataset connected to CrossRef, PubMed, Aguado Roca, Eva P:(DE-2719)2810320 1 dzne Broemer, Meike P:(DE-2719)2810307 2 Last author dzne 10.1038/s41420-019-0158-6 : Springer Science and Business Media LLC, 2019-03-11 1 75 journal-article Crossref Cell Death Discovery 5 2019 2058-7716 10.1038/s41420-019-0158-6 Vol. 5, no. 1, p. 75 PERI:(DE-600)2842546-7 1 5:1<75 75 Cell death discovery 5 2019 2058-7716 OpenAccess http://pub.dzne.de/record/140619/files/DZNE-2020-06941.pdf OpenAccess pdfa http://pub.dzne.de/record/140619/files/DZNE-2020-06941.pdf?subformat=pdfa Pubmed Central http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6411869 oai:pub.dzne.de:140619 openaire open_access VDB driver dnbdelivery Deutsches Zentrum für Neurodegenerative Erkrankungen I:(DE-588)1065079516 DZNE 0 P:(DE-2719)2810987 Deutsches Zentrum für Neurodegenerative Erkrankungen I:(DE-588)1065079516 DZNE 1 P:(DE-2719)2810320 Deutsches Zentrum für Neurodegenerative Erkrankungen I:(DE-588)1065079516 DZNE 2 P:(DE-2719)2810307 DE-HGF Gesundheit Erkrankungen des Nervensystems G:(DE-HGF)POF3-340 G:(DE-HGF)POF3-341 G:(DE-HGF)POF3 G:(DE-HGF)POF3-300 G:(DE-HGF)POF Molecular Signaling 0 2019 DBCoverage StatID:(DE-HGF)0200 StatID SCOPUS 2023-03-31 Creative Commons Attribution CC BY (No Version) LIC:(DE-HGF)CCBYNV V:(DE-HGF) DOAJ 2021-02-14T16:19:13Z DBCoverage StatID:(DE-HGF)1050 StatID BIOSIS Previews 2023-03-31 DBCoverage StatID:(DE-HGF)1190 StatID Biological Abstracts 2023-03-31 JCR StatID:(DE-HGF)0100 StatID CELL DEATH DISCOV : 2021 2023-03-31 DBCoverage StatID:(DE-HGF)1030 StatID Current Contents - Life Sciences 2023-03-31 DBCoverage StatID:(DE-HGF)0160 StatID Essential Science Indicators 2023-03-31 DBCoverage StatID:(DE-HGF)0501 StatID DOAJ Seal 2021-02-14T16:19:13Z DBCoverage StatID:(DE-HGF)0500 StatID DOAJ 2021-02-14T16:19:13Z WoS StatID:(DE-HGF)0113 StatID Science Citation Index Expanded 2023-03-31 Fees StatID:(DE-HGF)0700 StatID 2023-03-31 OpenAccess StatID:(DE-HGF)0510 StatID DBCoverage StatID:(DE-HGF)0150 StatID Web of Science Core Collection 2023-03-31 Peer Review StatID:(DE-HGF)0030 StatID DOAJ : Blind peer review 2021-02-14T16:19:13Z Article Processing Charges StatID:(DE-HGF)0561 StatID 2023-03-31 IF >= 5 StatID:(DE-HGF)9905 StatID CELL DEATH DISCOV : 2021 2023-03-31 DBCoverage StatID:(DE-HGF)0300 StatID Medline 2023-03-31 DBCoverage StatID:(DE-HGF)0199 StatID Clarivate Analytics Master Journal List 2023-03-31 I:(DE-2719)5000021 AG Brömer 1 Ubiquitin-Mediated Processes in Neurodegenerative Diseases 0 journal VDB UNRESTRICTED I:(DE-2719)5000021 FullTexts -- missing cx lookup -- 10.1038/ni1066 Crossref 10.1038/ni1066 -- missing cx lookup -- 10.1371/journal.ppat.1000650 Crossref 10.1371/journal.ppat.1000650 -- missing cx lookup -- 10.4161/15548627.2014.981792 Crossref 10.4161/15548627.2014.981792 -- missing cx lookup -- 10.1016/j.neuron.2014.05.016 Crossref 10.1016/j.neuron.2014.05.016 -- missing cx lookup -- 10.1056/NEJMoa1215993 Crossref 10.1056/NEJMoa1215993 -- missing cx lookup -- 10.1111/acel.12551 Crossref 10.1111/acel.12551 -- missing cx lookup -- 10.1212/WNL.0000000000001521 Crossref 10.1212/WNL.0000000000001521 -- missing cx lookup -- 10.1038/cr.2016.39 Crossref 10.1038/cr.2016.39 -- missing cx lookup -- 10.1146/annurev-biochem-060815-014922 Crossref 10.1146/annurev-biochem-060815-014922 -- missing cx lookup -- 10.1038/s41594-018-0063-3 Crossref 10.1038/s41594-018-0063-3 -- missing cx lookup -- 10.1038/nature09966 Crossref 10.1038/nature09966 -- missing cx lookup -- 10.1152/physiolgenomics.00226.2003 Crossref 10.1152/physiolgenomics.00226.2003 -- missing cx lookup -- 10.1038/33416 Crossref 10.1038/33416 -- missing cx lookup -- 10.1038/emboj.2011.204 Crossref 10.1038/emboj.2011.204 -- missing cx lookup -- 10.1083/jcb.201402104 Crossref 10.1083/jcb.201402104 -- missing cx lookup -- 10.1042/BJ20140334 Crossref 10.1042/BJ20140334 -- missing cx lookup -- 10.1038/nature13392 Crossref 10.1038/nature13392 -- missing cx lookup -- 10.1126/science.1237908 Crossref 10.1126/science.1237908 -- missing cx lookup -- 10.1038/sj.emboj.7601360 Crossref 10.1038/sj.emboj.7601360 -- missing cx lookup -- 10.1038/emboj.2012.217 Crossref 10.1038/emboj.2012.217 -- missing cx lookup -- 10.1093/nar/gky1131 Crossref 10.1093/nar/gky1131 -- missing cx lookup -- 10.1016/S0092-8674(02)00735-3 Crossref 10.1016/S0092-8674(02)00735-3 -- missing cx lookup -- 10.1034/j.1600-0854.2002.31006.x Crossref 10.1034/j.1600-0854.2002.31006.x -- missing cx lookup -- 10.1083/jcb.200312034 Crossref 10.1083/jcb.200312034 -- missing cx lookup -- 10.1091/mbc.e11-09-0817 Crossref 10.1091/mbc.e11-09-0817 -- missing cx lookup -- 10.1242/jcs.00723 Crossref 10.1242/jcs.00723 -- missing cx lookup -- 10.1016/S0968-0004(01)01835-7 Crossref 10.1016/S0968-0004(01)01835-7 -- missing cx lookup -- 10.1146/annurev-biochem-051810-094654 Crossref 10.1146/annurev-biochem-051810-094654 -- missing cx lookup -- 10.1091/mbc.e07-07-0678 Crossref 10.1091/mbc.e07-07-0678 -- missing cx lookup -- 10.1038/s41598-017-18513-5 Crossref 10.1038/s41598-017-18513-5 -- missing cx lookup -- 10.1038/nprot.2015.018 Crossref 10.1038/nprot.2015.018 -- missing cx lookup -- 10.1523/JNEUROSCI.20-11-03993.2000 Crossref 10.1523/JNEUROSCI.20-11-03993.2000 -- missing cx lookup -- 10.1016/j.neuron.2006.09.031 Crossref 10.1016/j.neuron.2006.09.031 -- missing cx lookup -- 10.1016/j.neuron.2006.08.034 Crossref 10.1016/j.neuron.2006.08.034 -- missing cx lookup -- 10.1038/nature16511 Crossref 10.1038/nature16511 -- missing cx lookup -- 10.1038/nature12638 Crossref 10.1038/nature12638 -- missing cx lookup -- 10.1146/annurev-biochem-060310-170328 Crossref 10.1146/annurev-biochem-060310-170328 -- missing cx lookup -- 10.1038/s41418-018-0164-x Crossref 10.1038/s41418-018-0164-x -- missing cx lookup -- 10.1006/abio.1995.1010 Crossref 10.1006/abio.1995.1010 -- missing cx lookup -- 10.1016/0003-2697(92)90061-B Crossref 10.1016/0003-2697(92)90061-B -- missing cx lookup -- 10.1021/pr700600n Crossref 10.1021/pr700600n -- missing cx lookup -- 10.1093/bioinformatics/btt703 Crossref 10.1093/bioinformatics/btt703