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Select item 112240281.

Hepatic oxylipin profiles in mouse models of Wilson disease: New insights into early hepatic manifestations

Tagreed A. Mazi, Noreene M. Shibata, Gaurav V. Sarode, Valentina Medici

Biochim Biophys Acta Mol Cell Biol Lipids. Author manuscript; available in PMC 2024 Jul 5.

Published in final edited form as: Biochim Biophys Acta Mol Cell Biol Lipids. 2024 Mar; 1869(2): 159446. Published online 2023 Dec 9. doi: 10.1016/j.bbalip.2023.159446

PMCID:: PMC11224028

Abstract Article PDF–2.2M

Select item 108969252.

Identification of lncRNA-miRNA-mRNA Networks in the Lenticular Nucleus Region of the Brain Contributes to Hepatolenticular Degeneration Pathogenesis and Therapy

Wenjie Hao, Wenming Yang, Yue Yang, Ting Cheng, Taohua Wei, Lulu Tang, Nannan Qian, Yulong Yang, Xiang Li, Hailin Jiang, Meixia Wang

Mol Neurobiol. 2024; 61(3): 1673–1686. Published online 2023 Sep 27. doi: 10.1007/s12035-023-03631-1

PMCID:: PMC10896925

Abstract Article PDF–1.6M

Select item 107212603.

Genome editing without nucleases confers proliferative advantage to edited hepatocytes and corrects Wilson disease

Agnese Padula, Michele Spinelli, Edoardo Nusco, Xabier Bujanda Cundin, Filomena Capolongo, Severo Campione, Claudia Perna, Amy Bastille, Megan Ericson, Chih-Chieh Wang, Shengwen Zhang, Angela Amoresano, Mariana Nacht, Pasquale Piccolo

JCI Insight. 2023 Nov 8; 8(21): e171281. Published online 2023 Nov 8. doi: 10.1172/jci.insight.171281

PMCID:: PMC10721260

Abstract Article PDF–2.6M

Select item 104972504.

The role of intestine in metabolic dysregulation in murine Wilson disease

Gaurav V. Sarode, Tagreed A. Mazi, Kari Neier, Noreene M. Shibata, Guillaume Jospin, Nathaniel H.O. Harder, Amanda Caceres, Marie C. Heffern, Ashok K. Sharma, Shyam K. More, Maneesh Dave, Shannon M. Schroeder, Li Wang, Janine M. LaSalle, Svetlana Lutsenko, Valentina Medici

Hepatol Commun. 2023 Oct; 7(10): e0247. Published online 2023 Sep 11. doi: 10.1097/HC9.0000000000000247

PMCID:: PMC10497250

Abstract Article PDF–1.9M

Select item 98701415.

Atp7b-dependent choroid plexus dysfunction causes transient copper deficit and metabolic changes in the developing mouse brain

Clorissa L. Washington-Hughes, Shubhrajit Roy, Herana Kamal Seneviratne, Senthilkumar S. Karuppagounder, Yulemni Morel, Jace W. Jones, Alex Zak, Tong Xiao, Tatiana N. Boronina, Robert N. Cole, Namandjé N. Bumpus, Christopher J. Chang, Ted M. Dawson, Svetlana Lutsenko

PLoS Genet. 2023 Jan; 19(1): e1010558. Published online 2023 Jan 10. doi: 10.1371/journal.pgen.1010558

PMCID:: PMC9870141

Abstract Article PDF–3.5M

Select item 87590436.

Hepatic Steatosis in the Mouse Model of Wilson Disease Coincides with a Muted Inflammatory Response

Aline Gottlieb, Som Dev, Lauren DeVine, Kathleen L. Gabrielson, Robert N. Cole, James P. Hamilton, Svetlana Lutsenko

Am J Pathol. 2022 Jan; 192(1): 146–159. doi: 10.1016/j.ajpath.2021.09.010

PMCID:: PMC8759043

Abstract Article PDF–5.0M

Select item 83257787.

Copper Sulfide Facilitates Hepatobiliary Clearance of Gold Nanoparticles through the Copper-Transporting ATPase ATP7B

Xiaodong Wang, Liangran Guo, Sihang Zhang, Yuan Chen, Yi-Tzai Chen, Binbin Zheng, Jingwen Sun, Yuyi Qian, Yixin Chen, Bingfang Yan, Wei Lu

ACS Nano. Author manuscript; available in PMC 2021 Aug 1.

Published in final edited form as: ACS Nano. 2019 May 28; 13(5): 5720–5730. Published online 2019 Apr 18. doi: 10.1021/acsnano.9b01154

PMCID:: PMC8325778

Abstract Article PDF–1.4M

Select item 83132398.

Cell-Type-Specific Gene Expression in Developing Mouse Neocortex: Intermediate Progenitors Implicated in Axon Development

Francesco Bedogni, Robert F. Hevner

Front Mol Neurosci. 2021; 14: 686034. Published online 2021 Jul 12. doi: 10.3389/fnmol.2021.686034

PMCID:: PMC8313239

Abstract Article PDF–1.6M

Select item 80823939.

Comprehensive RNA-Seq Analysis of Potential Therapeutic Targets of Gan–Dou–Fu–Mu Decoction for Treatment of Wilson Disease Using a Toxic Milk Mouse Model

Taohua Wei, Wenjie Hao, Lulu Tang, Huan Wu, Shi Huang, Yue Yang, Nannan Qian, Jie Liu, Wenming Yang, Xianchun Duan

Front Pharmacol. 2021; 12: 622268. Published online 2021 Apr 15. doi: 10.3389/fphar.2021.622268

PMCID:: PMC8082393

Abstract Article PDF–6.0M

Select item 795258010.

Systemic deletion of Atp7b modifies the hepatocytes’ response to copper overload in the mouse models of Wilson disease

Abigael Muchenditsi, C. Conover Talbot, Jr., Aline Gottlieb, Haojun Yang, Byunghak Kang, Tatiana Boronina, Robert Cole, Li Wang, Som Dev, James P. Hamilton, Svetlana Lutsenko

Sci Rep. 2021; 11: 5659. Published online 2021 Mar 11. doi: 10.1038/s41598-021-84894-3

PMCID:: PMC7952580

Abstract Article PDF–10M

Select item 793877011.

BraInMap elucidates the macromolecular connectivity landscape of mammalian brain

Reza Pourhaghighi, Peter E. A. Ash, Sadhna Phanse, Florian Goebels, Lucas Z. M. Hu, Siwei Chen, Yingying Zhang, Shayne D. Wierbowski, Samantha Boudeau, Mohamed T. Moutaoufik, Ramy H. Malty, Edyta Malolepsza, Kalliopi Tsafou, Aparna Nathan, Graham Cromar, Hongbo Guo, Ali Al Abdullatif, Daniel J. Apicco, Lindsay A. Becker, Aaron D. Gitler, Stefan M. Pulst, Ahmed Youssef, Ryan Hekman, Pierre C. Havugimana, Carl A. White, Benjamin C. Blum, Antonia Ratti, Camron D. Bryant, John Parkinson, Kasper Lage, Mohan Babu, Haiyuan Yu, Gary D. Bader, Benjamin Wolozin, Andrew Emili

Cell Syst. Author manuscript; available in PMC 2021 Apr 22.

Published in final edited form as: Cell Syst. 2020 Apr 22; 10(4): 333–350.e14. doi: 10.1016/j.cels.2020.03.003

Correction in:: Cell Syst. 2020 Aug 26; 11(2): 208.

PMCID:: PMC7938770

Abstract Article PDF–2.0M

Select item 781385112.

Inhibiting NLRP3 inflammasome activation prevents copper-induced neuropathology in a murine model of Wilson’s disease

Jianjian Dong, Xun Wang, Chenchen Xu, Manli Gao, Shijing Wang, Jin Zhang, Haiyang Tong, Lulu Wang, Yongzhu Han, Nan Cheng, Yongsheng Han

Cell Death Dis. 2021 Jan; 12(1): 87. Published online 2021 Jan 18. doi: 10.1038/s41419-021-03397-1

PMCID:: PMC7813851

Abstract Article PDF–3.7M

Select item 699499013.

Metabolic dysregulation in the Atp7b^−/− Wilson’s disease mouse model

Clavia Ruth Wooton-Kee, Matthew Robertson, Ying Zhou, Bingning Dong, Zhen Sun, Kang Ho Kim, Hailan Liu, Yong Xu, Nagireddy Putluri, Pradip Saha, Cristian Coarfa, David D. Moore, Alli M. Nuotio-Antar

Proc Natl Acad Sci U S A. 2020 Jan 28; 117(4): 2076–2083. Published online 2020 Jan 10. doi: 10.1073/pnas.1914267117

PMCID:: PMC6994990

Abstract Article PDF–1.1M

Select item 645274414.

ATP7A delivers copper to the lysyl oxidase family of enzymes and promotes tumorigenesis and metastasis

Vinit Shanbhag, Kimberly Jasmer-McDonald, Sha Zhu, Adam L. Martin, Nikita Gudekar, Aslam Khan, Erik Ladomersky, Kamlendra Singh, Gary A. Weisman, Michael J. Petris

Proc Natl Acad Sci U S A. 2019 Apr 2; 116(14): 6836–6841. Published online 2019 Mar 19. doi: 10.1073/pnas.1817473116

PMCID:: PMC6452744

Abstract Article PDF–1.1M

Select item 631149815.

ATP7A and ATP7B copper transporters have distinct functions in the regulation of neuronal dopamine-β-hydroxylase

Katharina Schmidt, Martina Ralle, Thomas Schaffer, Samuel Jayakanthan, Bilal Bari, Abigael Muchenditsi, Svetlana Lutsenko

J Biol Chem. 2018 Dec 28; 293(52): 20085–20098. Published online 2018 Oct 19. doi: 10.1074/jbc.RA118.004889

PMCID:: PMC6311498

Abstract Article PDF–3.7M

Select item 631003116.

Altered zinc balance in the Atp7b^−/− mouse model reveals a mechanism of copper toxicity in Wilson Disease

Kelsey A. Meacham, María Paz Cortés, Eve M. Wiggins, Alejandro Maass, Mauricio Latorre, Martina Ralle, Jason L. Burkhead

Metallomics. Author manuscript; available in PMC 2019 Nov 14.

Published in final edited form as: Metallomics. 2018 Nov 14; 10(11): 1595–1606. doi: 10.1039/c8mt00199e

PMCID:: PMC6310031

Abstract Article PDF–752K

Select item 621621117.

Epigenetic changes of the thioredoxin system in the tx-j mouse model and in patients with Wilson disease

Charles E Mordaunt, Noreene M Shibata, Dorothy A Kieffer, Anna Członkowska, Tomasz Litwin, Karl Heinz Weiss, Daniel N Gotthardt, Kristin Olson, Dongguang Wei, Stewart Cooper, Yu-Jui Yvonne Wan, Mohamed R Ali, Janine M LaSalle, Valentina Medici

Hum Mol Genet. 2018 Nov 15; 27(22): 3854–3869. Published online 2018 Jul 16. doi: 10.1093/hmg/ddy262

PMCID:: PMC6216211

Abstract Article PDF–32M

Select item 607503118.

DPM-1001 decreased copper levels and ameliorated deficits in a mouse model of Wilson's disease

Navasona Krishnan, Christy Felice, Keith Rivera, Darryl J. Pappin, Nicholas K. Tonks

Genes Dev. 2018 Jul 1; 32(13-14): 944–952. doi: 10.1101/gad.314658.118

PMCID:: PMC6075031

Abstract Article PDF–2.7M

Select item 553883619.

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

Abigael Muchenditsi, Haojun Yang, James P. Hamilton, Lahari Koganti, Franck Housseau, Lisa Aronov, Hongni Fan, Hannah Pierson, Ashima Bhattacharjee, Robert Murphy, Cynthia Sears, James Potter, Clavia R. Wooton-Kee, Svetlana Lutsenko

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

PMCID:: PMC5538836

Abstract Article

Select item 458828520.

Elevated copper impairs hepatic nuclear receptor function in Wilson’s disease

Clavia Ruth Wooton-Kee, Ajay K. Jain, Martin Wagner, Michael A. Grusak, Milton J. Finegold, Svetlana Lutsenko, David D. Moore

J Clin Invest. 2015 Sep 1; 125(9): 3449–3460. Published online 2015 Aug 4. doi: 10.1172/JCI78991

PMCID:: PMC4588285

Abstract Article PDF–1.3M

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