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Unraveling the molecular principles by which ceramides commit cells to death.

Dadsena S, Hassan DG, Holthuis JCM.

Cell Stress. 2019 Jul 16;3(8):280-283. doi: 10.15698/cst2019.08.196.


Ceramides bind VDAC2 to trigger mitochondrial apoptosis.

Dadsena S, Bockelmann S, Mina JGM, Hassan DG, Korneev S, Razzera G, Jahn H, Niekamp P, Müller D, Schneider M, Tafesse FG, Marrink SJ, Melo MN, Holthuis JCM.

Nat Commun. 2019 Apr 23;10(1):1832. doi: 10.1038/s41467-019-09654-4.


Osteoporosis and skeletal dysplasia caused by pathogenic variants in SGMS2.

Pekkinen M, Terhal PA, Botto LD, Henning P, Mäkitie RE, Roschger P, Jain A, Kol M, Kjellberg MA, Paschalis EP, van Gassen K, Murray M, Bayrak-Toydemir P, Magnusson MK, Jans J, Kausar M, Carey JC, Somerharju P, Lerner UH, Olkkonen VM, Klaushofer K, Holthuis JC, Mäkitie O.

JCI Insight. 2019 Apr 4;4(7). pii: 126180. doi: 10.1172/jci.insight.126180. eCollection 2019 Apr 4.


Optical manipulation of sphingolipid biosynthesis using photoswitchable ceramides.

Kol M, Williams B, Toombs-Ruane H, Franquelim HG, Korneev S, Schroeer C, Schwille P, Trauner D, Holthuis JC, Frank JA.

Elife. 2019 Feb 5;8. pii: e43230. doi: 10.7554/eLife.43230.


A search for ceramide binding proteins using bifunctional lipid analogs yields CERT-related protein StarD7.

Bockelmann S, Mina JGM, Korneev S, Hassan DG, Müller D, Hilderink A, Vlieg HC, Raijmakers R, Heck AJR, Haberkant P, Holthuis JCM.

J Lipid Res. 2018 Mar;59(3):515-530. doi: 10.1194/jlr.M082354. Epub 2018 Jan 17.


Ceramide phosphoethanolamine synthase SMSr is a target of caspase-6 during apoptotic cell death.

Cabukusta B, Nettebrock NT, Kol M, Hilderink A, Tafesse FG, Holthuis JCM.

Biosci Rep. 2017 Jul 16;37(4). pii: BSR20170867. doi: 10.1042/BSR20170867. Print 2017 Aug 31.


Membrane contact sites, ancient and central hubs of cellular lipid logistics.

Jain A, Holthuis JCM.

Biochim Biophys Acta Mol Cell Res. 2017 Sep;1864(9):1450-1458. doi: 10.1016/j.bbamcr.2017.05.017. Epub 2017 May 26. Review.


Switching head group selectivity in mammalian sphingolipid biosynthesis by active-site-engineering of sphingomyelin synthases.

Kol M, Panatala R, Nordmann M, Swart L, van Suijlekom L, Cabukusta B, Hilderink A, Grabietz T, Mina JGM, Somerharju P, Korneev S, Tafesse FG, Holthuis JCM.

J Lipid Res. 2017 May;58(5):962-973. doi: 10.1194/jlr.M076133. Epub 2017 Mar 23.


ER residency of the ceramide phosphoethanolamine synthase SMSr relies on homotypic oligomerization mediated by its SAM domain.

Cabukusta B, Kol M, Kneller L, Hilderink A, Bickert A, Mina JG, Korneev S, Holthuis JC.

Sci Rep. 2017 Jan 25;7:41290. doi: 10.1038/srep41290.


Diverting CERT-mediated ceramide transport to mitochondria triggers Bax-dependent apoptosis.

Jain A, Beutel O, Ebell K, Korneev S, Holthuis JC.

J Cell Sci. 2017 Jan 15;130(2):360-371. doi: 10.1242/jcs.194191. Epub 2016 Nov 25.


Monitoring Changes in the Oligomeric State of a Candidate Endoplasmic Reticulum (ER) Ceramide Sensor by Single-molecule Photobleaching.

Cabukusta B, Köhlen JA, Richter CP, You C, Holthuis JC.

J Biol Chem. 2016 Nov 18;291(47):24735-24746. Epub 2016 Oct 10.


Switching head group selectivity in mammalian sphingolipid biosynthesis by active-site engineering of sphingomyelin synthases.

Kol M, Panatala R, Nordmann M, Swart L, van Suijlekom L, Cabukusta B, Hilderink A, Grabietz T, Mina JG, Somerharju P, Korneev S, Tafesse FG, Holthuis JC.

J Lipid Res. 2016 Jul;57(7):1273-85. doi: 10.1194/jlr.M068692. Epub 2016 May 10. Retraction in: J Lipid Res. 2017 Apr;58(4):821.


Inner workings and biological impact of phospholipid flippases.

Panatala R, Hennrich H, Holthuis JC.

J Cell Sci. 2015 Jun 1;128(11):2021-32. doi: 10.1242/jcs.102715. Epub 2015 Apr 27. Review.


Functional characterization of enzymes catalyzing ceramide phosphoethanolamine biosynthesis in mice.

Bickert A, Ginkel C, Kol M, vom Dorp K, Jastrow H, Degen J, Jacobs RL, Vance DE, Winterhager E, Jiang XC, Dörmann P, Somerharju P, Holthuis JC, Willecke K.

J Lipid Res. 2015 Apr;56(4):821-35. doi: 10.1194/jlr.M055269. Epub 2015 Feb 9. Erratum in: J Lipid Res. 2017 Apr;58(4):820.


Lipid landscapes and pipelines in membrane homeostasis.

Holthuis JC, Menon AK.

Nature. 2014 Jun 5;510(7503):48-57. doi: 10.1038/nature13474. Review.


Fat & fabulous: bifunctional lipids in the spotlight.

Haberkant P, Holthuis JC.

Biochim Biophys Acta. 2014 Aug;1841(8):1022-30. doi: 10.1016/j.bbalip.2014.01.003. Epub 2014 Jan 15. Review.


Sphingomyelin synthase-related protein SMSr is a suppressor of ceramide-induced mitochondrial apoptosis.

Tafesse FG, Vacaru AM, Bosma EF, Hermansson M, Jain A, Hilderink A, Somerharju P, Holthuis JC.

J Cell Sci. 2014 Jan 15;127(Pt 2):445-54. doi: 10.1242/jcs.138933. Epub 2013 Nov 20.


In vivo profiling and visualization of cellular protein-lipid interactions using bifunctional fatty acids.

Haberkant P, Raijmakers R, Wildwater M, Sachsenheimer T, Brügger B, Maeda K, Houweling M, Gavin AC, Schultz C, van Meer G, Heck AJ, Holthuis JC.

Angew Chem Int Ed Engl. 2013 Apr 2;52(14):4033-8. doi: 10.1002/anie.201210178. Epub 2013 Feb 28. No abstract available.


Ceramide phosphoethanolamine biosynthesis in Drosophila is mediated by a unique ethanolamine phosphotransferase in the Golgi lumen.

Vacaru AM, van den Dikkenberg J, Ternes P, Holthuis JC.

J Biol Chem. 2013 Apr 19;288(16):11520-30. doi: 10.1074/jbc.M113.460972. Epub 2013 Feb 28.


Cellular microcompartments constitute general suborganellar functional units in cells.

Holthuis JC, Ungermann C.

Biol Chem. 2013 Feb;394(2):151-61. doi: 10.1515/hsz-2012-0265. Review.


Mapping functional interactions in a heterodimeric phospholipid pump.

Puts CF, Panatala R, Hennrich H, Tsareva A, Williamson P, Holthuis JC.

J Biol Chem. 2012 Aug 31;287(36):30529-40. doi: 10.1074/jbc.M112.371088. Epub 2012 Jul 12.


Pumping lipids with P4-ATPases.

López-Marqués RL, Holthuis JC, Pomorski TG.

Biol Chem. 2011 Jan;392(1-2):67-76. doi: 10.1515/BC.2011.015. Review.


CDC50 proteins are critical components of the human class-1 P4-ATPase transport machinery.

Bryde S, Hennrich H, Verhulst PM, Devaux PF, Lenoir G, Holthuis JC.

J Biol Chem. 2010 Dec 24;285(52):40562-72. doi: 10.1074/jbc.M110.139543. Epub 2010 Oct 20.


Tales and mysteries of the enigmatic sphingomyelin synthase family.

Holthuis JC, Luberto C.

Adv Exp Med Biol. 2010;688:72-85. Review.


A flippase-independent function of ATP8B1, the protein affected in familial intrahepatic cholestasis type 1, is required for apical protein expression and microvillus formation in polarized epithelial cells.

Verhulst PM, van der Velden LM, Oorschot V, van Faassen EE, Klumperman J, Houwen RH, Pomorski TG, Holthuis JC, Klomp LW.

Hepatology. 2010 Jun;51(6):2049-60. doi: 10.1002/hep.23586.


Cell biology: A brake on lipid synthesis.

Tafesse FG, Holthuis JC.

Nature. 2010 Feb 25;463(7284):1028-9. doi: 10.1038/4631028a. No abstract available.


A P4-ATPase protein interaction network reveals a link between aminophospholipid transport and phosphoinositide metabolism.

Puts CF, Lenoir G, Krijgsveld J, Williamson P, Holthuis JC.

J Proteome Res. 2010 Feb 5;9(2):833-42. doi: 10.1021/pr900743b.


Folding defects in P-type ATP 8B1 associated with hereditary cholestasis are ameliorated by 4-phenylbutyrate.

van der Velden LM, Stapelbroek JM, Krieger E, van den Berghe PV, Berger R, Verhulst PM, Holthuis JC, Houwen RH, Klomp LW, van de Graaf SF.

Hepatology. 2010 Jan;51(1):286-96. doi: 10.1002/hep.23268.


Membrane contact sites between apicoplast and ER in Toxoplasma gondii revealed by electron tomography.

Tomova C, Humbel BM, Geerts WJ, Entzeroth R, Holthuis JC, Verkleij AJ.

Traffic. 2009 Oct;10(10):1471-80. doi: 10.1111/j.1600-0854.2009.00954.x. Epub 2009 Jun 9.


Sphingomyelin synthase-related protein SMSr controls ceramide homeostasis in the ER.

Vacaru AM, Tafesse FG, Ternes P, Kondylis V, Hermansson M, Brouwers JF, Somerharju P, Rabouille C, Holthuis JC.

J Cell Biol. 2009 Jun 15;185(6):1013-27. doi: 10.1083/jcb.200903152. Epub 2009 Jun 8.


Sphingomyelin synthase SMS2 displays dual activity as ceramide phosphoethanolamine synthase.

Ternes P, Brouwers JF, van den Dikkenberg J, Holthuis JC.

J Lipid Res. 2009 Nov;50(11):2270-7. doi: 10.1194/jlr.M900230-JLR200. Epub 2009 May 19.


Cdc50p plays a vital role in the ATPase reaction cycle of the putative aminophospholipid transporter Drs2p.

Lenoir G, Williamson P, Puts CF, Holthuis JC.

J Biol Chem. 2009 Jul 3;284(27):17956-67. doi: 10.1074/jbc.M109.013722. Epub 2009 May 2.


Mechanism and significance of P4 ATPase-catalyzed lipid transport: lessons from a Na+/K+-pump.

Puts CF, Holthuis JC.

Biochim Biophys Acta. 2009 Jul;1791(7):603-11. doi: 10.1016/j.bbalip.2009.02.005. Epub 2009 Feb 21. Review.


On the origin of lipid asymmetry: the flip side of ion transport.

Lenoir G, Williamson P, Holthuis JC.

Curr Opin Chem Biol. 2007 Dec;11(6):654-61. Epub 2007 Nov 5. Review.


Both sphingomyelin synthases SMS1 and SMS2 are required for sphingomyelin homeostasis and growth in human HeLa cells.

Tafesse FG, Huitema K, Hermansson M, van der Poel S, van den Dikkenberg J, Uphoff A, Somerharju P, Holthuis JC.

J Biol Chem. 2007 Jun 15;282(24):17537-47. Epub 2007 Apr 22.


The multigenic sphingomyelin synthase family.

Tafesse FG, Ternes P, Holthuis JC.

J Biol Chem. 2006 Oct 6;281(40):29421-5. Epub 2006 Aug 11. Review. No abstract available.


Loss of P4 ATPases Drs2p and Dnf3p disrupts aminophospholipid transport and asymmetry in yeast post-Golgi secretory vesicles.

Alder-Baerens N, Lisman Q, Luong L, Pomorski T, Holthuis JC.

Mol Biol Cell. 2006 Apr;17(4):1632-42. Epub 2006 Feb 1.


The CLN9 protein, a regulator of dihydroceramide synthase.

Schulz A, Mousallem T, Venkataramani M, Persaud-Sawin DA, Zucker A, Luberto C, Bielawska A, Bielawski J, Holthuis JC, Jazwinski SM, Kozhaya L, Dbaibo GS, Boustany RM.

J Biol Chem. 2006 Feb 3;281(5):2784-94. Epub 2005 Nov 22.


Lipid traffic: floppy drives and a superhighway.

Holthuis JC, Levine TP.

Nat Rev Mol Cell Biol. 2005 Mar;6(3):209-20. Review.


The elusive flippases.

Lenoir G, Holthuis JC.

Curr Biol. 2004 Nov 9;14(21):R912-3. Review. No abstract available.


HOR7, a multicopy suppressor of the Ca2+-induced growth defect in sphingolipid mannosyltransferase-deficient yeast.

Lisman Q, Urli-Stam D, Holthuis JC.

J Biol Chem. 2004 Aug 27;279(35):36390-6. Epub 2004 Jun 18.


Tracking down lipid flippases and their biological functions.

Pomorski T, Holthuis JC, Herrmann A, van Meer G.

J Cell Sci. 2004 Feb 29;117(Pt 6):805-13. Review.


Identification of a family of animal sphingomyelin synthases.

Huitema K, van den Dikkenberg J, Brouwers JF, Holthuis JC.

EMBO J. 2004 Jan 14;23(1):33-44. Epub 2003 Dec 18.


Sensing membrane curvature.

Holthuis JC, Burger KN.

Dev Cell. 2003 Dec;5(6):821-2. Review. No abstract available.


Protein sorting in the late Golgi of Saccharomyces cerevisiae does not require mannosylated sphingolipids.

Lisman Q, Pomorski T, Vogelzangs C, Urli-Stam D, de Cocq van Delwijnen W, Holthuis JC.

J Biol Chem. 2004 Jan 9;279(2):1020-9. Epub 2003 Oct 28.


Lipid microdomains, lipid translocation and the organization of intracellular membrane transport (Review).

Holthuis JC, van Meer G, Huitema K.

Mol Membr Biol. 2003 Jul-Sep;20(3):231-41. Review.


Drs2p-related P-type ATPases Dnf1p and Dnf2p are required for phospholipid translocation across the yeast plasma membrane and serve a role in endocytosis.

Pomorski T, Lombardi R, Riezman H, Devaux PF, van Meer G, Holthuis JC.

Mol Biol Cell. 2003 Mar;14(3):1240-54.


The organizing potential of sphingolipids in intracellular membrane transport.

Holthuis JC, Pomorski T, Raggers RJ, Sprong H, Van Meer G.

Physiol Rev. 2001 Oct;81(4):1689-723. Review.


Lipid traffic: the ABC of transbilayer movement.

Raggers RJ, Pomorski T, Holthuis JC, Kälin N, van Meer G.

Traffic. 2000 Mar;1(3):226-34. Review.


Inhibition of the vacuolar H+-ATPase perturbs the transport, sorting, processing and release of regulated secretory proteins.

Schoonderwoert VT, Holthuis JC, Tanaka S, Tooze SA, Martens GJ.

Eur J Biochem. 2000 Sep;267(17):5646-54.

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