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Targeted inactivation of copper transporter Atp7b in hepatocytes causes liver steatosis and obesity in mice.

Muchenditsi A, Yang H, Hamilton JP, Koganti L, Housseau F, Aronov L, Fan H, Pierson H, Bhattacharjee A, Murphy R, Sears C, Potter J, Wooton-Kee CR, Lutsenko S.

Am J Physiol Gastrointest Liver Physiol. 2017 Jul 1;313(1):G39-G49. doi: 10.1152/ajpgi.00312.2016. Epub 2017 Apr 20.


Early gestational gene transfer with targeted ATP7B expression in the liver improves phenotype in a murine model of Wilson's disease.

Roybal JL, Endo M, Radu A, Gray L, Todorow CA, Zoltick PW, Lutsenko S, Flake AW.

Gene Ther. 2012 Nov;19(11):1085-94. doi: 10.1038/gt.2011.186. Epub 2011 Dec 8.


Hepatic copper-transporting ATPase ATP7B: function and inactivation at the molecular and cellular level.

Bartee MY, Lutsenko S.

Biometals. 2007 Jun;20(3-4):627-37. Epub 2007 Feb 1. Review.


Wilson disease at a single cell level: intracellular copper trafficking activates compartment-specific responses in hepatocytes.

Ralle M, Huster D, Vogt S, Schirrmeister W, Burkhead JL, Capps TR, Gray L, Lai B, Maryon E, Lutsenko S.

J Biol Chem. 2010 Oct 1;285(40):30875-83. doi: 10.1074/jbc.M110.114447. Epub 2010 Jul 20.


Wilson disease protein ATP7B utilizes lysosomal exocytosis to maintain copper homeostasis.

Polishchuk EV, Concilli M, Iacobacci S, Chesi G, Pastore N, Piccolo P, Paladino S, Baldantoni D, van IJzendoorn SC, Chan J, Chang CJ, Amoresano A, Pane F, Pucci P, Tarallo A, Parenti G, Brunetti-Pierri N, Settembre C, Ballabio A, Polishchuk RS.

Dev Cell. 2014 Jun 23;29(6):686-700. doi: 10.1016/j.devcel.2014.04.033. Epub 2014 Jun 5.


The discrepancy between the absence of copper deposition and the presence of neuronal damage in the brain of Atp7b(-/-) mice.

Dong Y, Shi SS, Chen S, Ni W, Zhu M, Wu ZY.

Metallomics. 2015 Feb;7(2):283-8. doi: 10.1039/c4mt00242c.


Hepatocyte GP73 expression in Wilson disease.

Wright LM, Huster D, Lutsenko S, Wrba F, Ferenci P, Fimmel CJ.

J Hepatol. 2009 Sep;51(3):557-64. doi: 10.1016/j.jhep.2009.05.029. Epub 2009 Jun 25.


Urinary copper elevation in a mouse model of Wilson's disease is a regulated process to specifically decrease the hepatic copper load.

Gray LW, Peng F, Molloy SA, Pendyala VS, Muchenditsi A, Muzik O, Lee J, Kaplan JH, Lutsenko S.

PLoS One. 2012;7(6):e38327. doi: 10.1371/journal.pone.0038327. Epub 2012 Jun 22.


Activation of Autophagy, Observed in Liver Tissues From Patients With Wilson Disease and From ATP7B-Deficient Animals, Protects Hepatocytes From Copper-Induced Apoptosis.

Polishchuk EV, Merolla A, Lichtmannegger J, Romano A, Indrieri A, Ilyechova EY, Concilli M, De Cegli R, Crispino R, Mariniello M, Petruzzelli R, Ranucci G, Iorio R, Pietrocola F, Einer C, Borchard S, Zibert A, Schmidt HH, Di Schiavi E, Puchkova LV, Franco B, Kroemer G, Zischka H, Polishchuk RS.

Gastroenterology. 2019 Mar;156(4):1173-1189.e5. doi: 10.1053/j.gastro.2018.11.032. Epub 2018 Nov 17.


Distinct Wilson's disease mutations in ATP7B are associated with enhanced binding to COMMD1 and reduced stability of ATP7B.

de Bie P, van de Sluis B, Burstein E, van de Berghe PV, Muller P, Berger R, Gitlin JD, Wijmenga C, Klomp LW.

Gastroenterology. 2007 Oct;133(4):1316-26. Epub 2007 Jul 25.


Intracellular targeting of copper-transporting ATPase ATP7A in a normal and Atp7b-/- kidney.

Linz R, Barnes NL, Zimnicka AM, Kaplan JH, Eipper B, Lutsenko S.

Am J Physiol Renal Physiol. 2008 Jan;294(1):F53-61. Epub 2007 Oct 10.


NH2-terminal signals in ATP7B Cu-ATPase mediate its Cu-dependent anterograde traffic in polarized hepatic cells.

Guo Y, Nyasae L, Braiterman LT, Hubbard AL.

Am J Physiol Gastrointest Liver Physiol. 2005 Nov;289(5):G904-16. Epub 2005 Jun 30.


Activation of liver X receptor/retinoid X receptor pathway ameliorates liver disease in Atp7B(-/-) (Wilson disease) mice.

Hamilton JP, Koganti L, Muchenditsi A, Pendyala VS, Huso D, Hankin J, Murphy RC, Huster D, Merle U, Mangels C, Yang N, Potter JJ, Mezey E, Lutsenko S.

Hepatology. 2016 Jun;63(6):1828-41. doi: 10.1002/hep.28406. Epub 2016 Feb 22.


Novel ATPase Cu(2+) transporting beta polypeptide mutations in Chinese families with Wilson's disease.

Gu S, Yang H, Qi Y, Deng X, Zhang L, Guo Y, Huang Q, Li J, Shi X, Song Z, Deng H.

PLoS One. 2013 Jul 2;8(7):e66526. doi: 10.1371/journal.pone.0066526. Print 2013.


Elevated copper impairs hepatic nuclear receptor function in Wilson's disease.

Wooton-Kee CR, Jain AK, Wagner M, Grusak MA, Finegold MJ, Lutsenko S, Moore DD.

J Clin Invest. 2015 Sep;125(9):3449-60. doi: 10.1172/JCI78991. Epub 2015 Aug 4.


The copper-transporting ATPases, menkes and wilson disease proteins, have distinct roles in adult and developing cerebellum.

Barnes N, Tsivkovskii R, Tsivkovskaia N, Lutsenko S.

J Biol Chem. 2005 Mar 11;280(10):9640-5. Epub 2005 Jan 5.


Hepatocyte transplantation in the Long Evans Cinnamon rat model of Wilson's disease.

Park SM, Vo K, Lallier M, Cloutier AS, Brochu P, Alvarez F, Martin SR.

Cell Transplant. 2006;15(1):13-22.


Distinct phenotype of a Wilson disease mutation reveals a novel trafficking determinant in the copper transporter ATP7B.

Braiterman LT, Murthy A, Jayakanthan S, Nyasae L, Tzeng E, Gromadzka G, Woolf TB, Lutsenko S, Hubbard AL.

Proc Natl Acad Sci U S A. 2014 Apr 8;111(14):E1364-73. doi: 10.1073/pnas.1314161111. Epub 2014 Mar 24.


Functional interactions of Cu-ATPase ATP7B with cisplatin and the role of ATP7B in the resistance of cells to the drug.

Leonhardt K, Gebhardt R, Mössner J, Lutsenko S, Huster D.

J Biol Chem. 2009 Mar 20;284(12):7793-802. doi: 10.1074/jbc.M805145200. Epub 2009 Jan 13.


Functional analysis and drug response to zinc and D-penicillamine in stable ATP7B mutant hepatic cell lines.

Chandhok G, Horvath J, Aggarwal A, Bhatt M, Zibert A, Schmidt HH.

World J Gastroenterol. 2016 Apr 28;22(16):4109-19. doi: 10.3748/wjg.v22.i16.4109.

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