000136797 001__ 136797 000136797 005__ 20240229105100.0 000136797 0247_ $$2doi$$a10.1016/bs.pmbts.2018.01.002 000136797 0247_ $$2pmid$$apmid:29747811 000136797 0247_ $$2ISSN$$a0079-6603 000136797 0247_ $$2ISSN$$a1877-1173 000136797 0247_ $$2ISSN$$a1878-0814 000136797 0247_ $$2altmetric$$aaltmetric:41151052 000136797 037__ $$aDKFZ-2018-01235 000136797 041__ $$aeng 000136797 082__ $$a570 000136797 1001_ $$0P:(DE-He78)a928ded2085c8911822370cad0b4a728$$aSandhoff, Roger$$b0$$eFirst author$$udkfz 000136797 245__ $$aGanglioside Metabolism in Health and Disease. 000136797 260__ $$aPittsburgh, PA$$bGenetics Soc. of America$$c2018 000136797 3367_ $$2DRIVER$$aarticle 000136797 3367_ $$2DataCite$$aOutput Types/Journal article 000136797 3367_ $$0PUB:(DE-HGF)16$$2PUB:(DE-HGF)$$aJournal Article$$bjournal$$mjournal$$s1536239235_3642$$xReview Article 000136797 3367_ $$2BibTeX$$aARTICLE 000136797 3367_ $$2ORCID$$aJOURNAL_ARTICLE 000136797 3367_ $$00$$2EndNote$$aJournal Article 000136797 520__ $$aGangliosides (GGs) are cell type-specific sialic acid-containing glycosphingolipids (GSLs), which are enriched in mammalian brain. Defects in GSL metabolism cause fatal human diseases. GSLs are composed of a hydrophilic oligosaccharide linked in 1-O-position to a hydrophobic ceramide anchor, which itself is composed of a long-chain amino alcohol, the sphingoid base, and an amide-bound acyl chain. Biosynthesis of mammalian GGs and other GSLs starts with the formation of their hydrophobic ceramide anchor in the endoplasmic reticulum, followed by sequential glycosylation reactions along the secretory pathway, mainly at the luminal surface of Golgi and trans-Golgi network (TGN) membranes. Few membrane-anchored and often promiscuous glycosyltransferases allow the formation of cell type-specific glycolipid patterns in a combinatorial process. Inherited defects of these transferases therefore affect not only single structures but defined glycolipid series. GGs and other GSLs are thereafter transported by an exocytotic membrane flow to the plasma membrane where they are expressed in cell type-specific patterns, which can be modified by metabolic reactions at or near the cellular surface. Endocytosed (glyco)sphingolipids are degraded, together with other membrane lipids in a stepwise fashion by endolysosomal enzymes with the help of small lipid-binding proteins, the sphingolipid activator proteins (SAPs), at the surface of intraluminal lysosomal vesicles. Inherited defects in a sphingolipid-degrading enzyme or SAP cause the accumulation of the corresponding lipid substrates. Endolysosomal GSL degradation is strongly modified by the lipid components of the organelle microenvironments. 000136797 536__ $$0G:(DE-HGF)POF3-317$$a317 - Translational cancer research (POF3-317)$$cPOF3-317$$fPOF III$$x0 000136797 588__ $$aDataset connected to CrossRef Book Series, PubMed, 000136797 7001_ $$aSchulze, Heike$$b1 000136797 7001_ $$aSandhoff, Konrad$$b2 000136797 773__ $$0PERI:(DE-600)2629978-1$$a10.1016/bs.pmbts.2018.01.002$$p1-62$$tG3: Genes, genomes, genetics$$v156$$x2160-1836$$y2018 000136797 909CO $$ooai:inrepo02.dkfz.de:136797$$pVDB 000136797 9101_ $$0I:(DE-588b)2036810-0$$6P:(DE-He78)a928ded2085c8911822370cad0b4a728$$aDeutsches Krebsforschungszentrum$$b0$$kDKFZ 000136797 9131_ $$0G:(DE-HGF)POF3-317$$1G:(DE-HGF)POF3-310$$2G:(DE-HGF)POF3-300$$3G:(DE-HGF)POF3$$4G:(DE-HGF)POF$$aDE-HGF$$bGesundheit$$lKrebsforschung$$vTranslational cancer research$$x0 000136797 9141_ $$y2018 000136797 915__ $$0StatID:(DE-HGF)0100$$2StatID$$aJCR$$bG3-GENES GENOM GENET : 2015 000136797 915__ $$0StatID:(DE-HGF)0200$$2StatID$$aDBCoverage$$bSCOPUS 000136797 915__ $$0StatID:(DE-HGF)0300$$2StatID$$aDBCoverage$$bMedline 000136797 915__ $$0StatID:(DE-HGF)0310$$2StatID$$aDBCoverage$$bNCBI Molecular Biology Database 000136797 915__ $$0StatID:(DE-HGF)0501$$2StatID$$aDBCoverage$$bDOAJ Seal 000136797 915__ $$0StatID:(DE-HGF)0500$$2StatID$$aDBCoverage$$bDOAJ 000136797 915__ $$0LIC:(DE-HGF)CCBYNV$$2V:(DE-HGF)$$aCreative Commons Attribution CC BY (No Version)$$bDOAJ 000136797 915__ $$0StatID:(DE-HGF)0199$$2StatID$$aDBCoverage$$bThomson Reuters Master Journal List 000136797 915__ $$0StatID:(DE-HGF)0111$$2StatID$$aWoS$$bScience Citation Index Expanded 000136797 915__ $$0StatID:(DE-HGF)0150$$2StatID$$aDBCoverage$$bWeb of Science Core Collection 000136797 915__ $$0StatID:(DE-HGF)1050$$2StatID$$aDBCoverage$$bBIOSIS Previews 000136797 915__ $$0StatID:(DE-HGF)9900$$2StatID$$aIF < 5 000136797 9201_ $$0I:(DE-He78)G131-20160331$$kG131$$lAG Lipid-Pathobiochemie$$x0 000136797 980__ $$ajournal 000136797 980__ $$aVDB 000136797 980__ $$aI:(DE-He78)G131-20160331 000136797 980__ $$aUNRESTRICTED