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Select item 110313801.

CRISPR editing of anti-anemia drug target rescues independent preclinical models of retinitis pigmentosa

Nicholas D. Nolan, Xuan Cui, Brian M. Robbings, Aykut Demirkol, Kriti Pandey, Wen-Hsuan Wu, Hannah F. Hu, Laura A. Jenny, Chyuan-Sheng Lin, Daniel T. Hass, Jianhai Du, James B. Hurley, Stephen H. Tsang

Cell Rep Med. 2024 Apr 16; 5(4): 101459. Published online 2024 Mar 21. doi: 10.1016/j.xcrm.2024.101459

PMCID:: PMC11031380

Abstract Article PDF–6.3M

Select item 95783862.

PKM2 ablation enhanced retinal function and survival in a preclinical model of retinitis pigmentosa

Ethan Zhang, Joseph Ryu, Sarah R. Levi, Jin Kyun Oh, Chun Wei Hsu, Xuan Cui, Ting-Ting Lee, Nan-Kai Wang, Jose Ronaldo Lima de Carvalho, Stephen H. Tsang

Mamm Genome. Author manuscript; available in PMC 2022 Oct 18.

Published in final edited form as: Mamm Genome. 2020 Apr; 31(3-4): 77–85. Published online 2020 Apr 27. doi: 10.1007/s00335-020-09837-1

PMCID:: PMC9578386

Abstract Article PDF–998K

Select item 95093283.

The Musashi proteins direct post-transcriptional control of protein expression and alternate exon splicing in vertebrate photoreceptors

Fatimah Matalkah, Bohye Jeong, Macie Sheridan, Eric Horstick, Visvanathan Ramamurthy, Peter Stoilov

Commun Biol. 2022; 5: 1011. Published online 2022 Sep 24. doi: 10.1038/s42003-022-03990-w

PMCID:: PMC9509328

Abstract Article PDF–8.0M

Select item 78384184.

Absence of S100A4 in the mouse lens induces an aberrant retina-specific differentiation program and cataract

Rupalatha Maddala, Junyuan Gao, Richard T. Mathias, Tylor R. Lewis, Vadim Y. Arshavsky, Adriana Levine, Jonathan M. Backer, Anne R. Bresnick, Ponugoti V. Rao

Sci Rep. 2021; 11: 2203. Published online 2021 Jan 26. doi: 10.1038/s41598-021-81611-y

PMCID:: PMC7838418

Abstract Article PDF–6.5M

Select item 74934295.

CAPN5 genetic inactivation phenotype supports therapeutic inhibition trials

Katherine J. Wert, Susanne F. Koch, Gabriel Velez, Chun-Wei Hsu, MaryAnn Mahajan, Alexander G. Bassuk, Stephen H. Tsang, Vinit B. Mahajan

Hum Mutat. Author manuscript; available in PMC 2020 Sep 16.

Published in final edited form as: Hum Mutat. 2019 Dec; 40(12): 2377–2392. Published online 2019 Aug 26. doi: 10.1002/humu.23894

PMCID:: PMC7493429

Abstract Article PDF–2.0M

Select item 72270286.

Mouse Models of Inherited Retinal Degeneration with Photoreceptor Cell Loss

Gayle B. Collin, Navdeep Gogna, Bo Chang, Nattaya Damkham, Jai Pinkney, Lillian F. Hyde, Lisa Stone, Jürgen K. Naggert, Patsy M. Nishina, Mark P. Krebs

Cells. 2020 Apr; 9(4): 931. Published online 2020 Apr 10. doi: 10.3390/cells9040931

PMCID:: PMC7227028

Abstract Article PDF–5.4M

Select item 68160937.

Interaction of the tetratricopeptide repeat domain of aryl hydrocarbon receptor–interacting protein–like 1 with the regulatory Pγ subunit of phosphodiesterase 6

Ravi P. Yadav, Kimberly Boyd, Liping Yu, Nikolai O. Artemyev

J Biol Chem. 2019 Oct 25; 294(43): 15795–15807. Published online 2019 Sep 5. doi: 10.1074/jbc.RA119.010666

PMCID:: PMC6816093

Abstract Article PDF–2.9M

Select item 63812538.

ARL13B, a Joubert Syndrome-Associated Protein, Is Critical for Retinogenesis and Elaboration of Mouse Photoreceptor Outer Segments

Tanya L. Dilan, Abigail R. Moye, Ezequiel M. Salido, Thamaraiselvi Saravanan, Saravanan Kolandaivelu, Andrew F.X. Goldberg, Visvanathan Ramamurthy

J Neurosci. 2019 Feb 20; 39(8): 1347–1364. doi: 10.1523/JNEUROSCI.1761-18.2018

PMCID:: PMC6381253

Abstract Article PDF–8.3M

Select item 60944869.

Genetic Rescue Reverses Microglial Activation in Preclinical Models of Retinitis Pigmentosa

Lijuan Zhang, Xuan Cui, Ruben Jauregui, Karen Sophia Park, Sally Justus, Yi-Ting Tsai, Jimmy K. Duong, Chun-Wei Hsu, Wen-Hsuan Wu, Christine L. Xu, Chyuan-Sheng Lin, Stephen H. Tsang

Mol Ther. 2018 Aug 1; 26(8): 1953–1964. Published online 2018 Jun 21. doi: 10.1016/j.ymthe.2018.06.014

PMCID:: PMC6094486

Abstract Article PDF–4.8M

Select item 607558110.

Two pathways of rod photoreceptor cell death induced by elevated cGMP

Tian Wang, Stephen H. Tsang, Jeannie Chen

Hum Mol Genet. 2017 Jun 15; 26(12): 2299–2306. Published online 2017 Apr 3. doi: 10.1093/hmg/ddx121

PMCID:: PMC6075581

Abstract Article PDF–571K

Select item 529119811.

Reprogramming towards anabolism impedes degeneration in a preclinical model of retinitis pigmentosa

Lijuan Zhang, Sally Justus, Yu Xu, Tamara Pluchenik, Chun-Wei Hsu, Jin Yang, Jimmy K. Duong, Chyuan-Sheng Lin, Yading Jia, Alexander G. Bassuk, Vinit B. Mahajan, Stephen H. Tsang

Hum Mol Genet. 2016 Oct 1; 25(19): 4244–4255. Published online 2016 Aug 11. doi: 10.1093/hmg/ddw256

PMCID:: PMC5291198

Abstract Article PDF–1.8M

Select item 512768412.

Reprogramming metabolism by targeting sirtuin 6 attenuates retinal degeneration

Lijuan Zhang, Jianhai Du, Sally Justus, Chun-Wei Hsu, Luis Bonet-Ponce, Wen-Hsuan Wu, Yi-Ting Tsai, Wei-Pu Wu, Yading Jia, Jimmy K. Duong, Vinit B. Mahajan, Chyuan-Sheng Lin, Shuang Wang, James B. Hurley, Stephen H. Tsang

J Clin Invest. 2016 Dec 1; 126(12): 4659–4673. Published online 2016 Nov 14. doi: 10.1172/JCI86905

PMCID:: PMC5127684

Abstract Article PDF–3.0M

Select item 496557613.

Aryl Hydrocarbon Receptor-interacting Protein-like 1 Is an Obligate Chaperone of Phosphodiesterase 6 and Is Assisted by the γ-Subunit of Its Client

Kota N. Gopalakrishna, Kimberly Boyd, Ravi P. Yadav, Nikolai O. Artemyev

J Biol Chem. 2016 Jul 29; 291(31): 16282–16291. Published online 2016 Jun 7. doi: 10.1074/jbc.M116.737593

PMCID:: PMC4965576

Abstract Article PDF–1.8M

Select item 458829914.

Halting progressive neurodegeneration in advanced retinitis pigmentosa

Susanne F. Koch, Yi-Ting Tsai, Jimmy K. Duong, Wen-Hsuan Wu, Chun-Wei Hsu, Wei-Pu Wu, Luis Bonet-Ponce, Chyuan-Sheng Lin, Stephen H. Tsang

J Clin Invest. 2015 Sep 1; 125(9): 3704–3713. Published online 2015 Aug 24. doi: 10.1172/JCI82462

PMCID:: PMC4588299

Abstract Article PDF–3.2M

Select item 440033915.

Targeted Ablation of the Pde6h Gene in Mice Reveals Cross-species Differences in Cone and Rod Phototransduction Protein Isoform Inventory

Christina Brennenstuhl, Naoyuki Tanimoto, Markus Burkard, Rebecca Wagner, Sylvia Bolz, Dragana Trifunovic, Clement Kabagema-Bilan, Francois Paquet-Durand, Susanne C. Beck, Gesine Huber, Mathias W. Seeliger, Peter Ruth, Bernd Wissinger, Robert Lukowski

J Biol Chem. 2015 Apr 17; 290(16): 10242–10255. Published online 2015 Mar 4. doi: 10.1074/jbc.M114.611921

PMCID:: PMC4400339

Abstract Article PDF–2.5M

Select item 357241016.

A conditional knockout resource for the genome–wide study of mouse gene function

William C. Skarnes, Barry Rosen, Anthony P. West, Manousos Koutsourakis, Wendy Bushell, Vivek Iyer, Alejandro O. Mujica, Mark Thomas, Jennifer Harrow, Tony Cox, David Jackson, Jessica Severin, Patrick Biggs, Jun Fu, Michael Nefedov, Pieter J. de Jong, A. Francis Stewart, Allan Bradley

Nature. Author manuscript; available in PMC 2013 Feb 14.

Published in final edited form as: Nature. 2011 Jun 15; 474(7351): 337–342. Published online 2011 Jun 15. doi: 10.1038/nature10163

PMCID:: PMC3572410

Abstract Article PDF–1.1M

Select item 333976417.

Stimulation of the insulin/mTOR pathway delays cone death in a mouse model of Retinitis Pigmentosa

Claudio Punzo, Karl Kornacker, Constance L. Cepko

Nat Neurosci. Author manuscript; available in PMC 2012 Apr 30.

Published in final edited form as: Nat Neurosci. 2009 Jan; 12(1): 44–52. Published online 2008 Dec 7. doi: 10.1038/nn.2234

PMCID:: PMC3339764

Abstract Article PDF–4.2M

Select item 320519818.

Effect of the ILE86TER mutation in the γ subunit of cGMP phosphodiesterase (PDE6) on rod photoreceptor signalling

Stephen H. Tsang, Michael L. Woodruff, Chyuan-Sheng Lin, Barry D. Jacobson, Matthew C. Naumann, Chun Wei Hsu, Richard J. Davis, Marianne C. Cilluffo, Jeannie Chen, Gordon L. Fain

Cell Signal. Author manuscript; available in PMC 2013 Jan 1.

Published in final edited form as: Cell Signal. 2012 Jan; 24(1): 181–188. Published online 2011 Sep 8. doi: 10.1016/j.cellsig.2011.08.021