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Select item 58862441.

Rescue of peripheral vestibular function in Usher syndrome mice using a splice-switching antisense oligonucleotide

Sarath Vijayakumar, Frederic F. Depreux, Francine M. Jodelka, Jennifer J. Lentz, Frank Rigo, Timothy A. Jones, Michelle L. Hastings

Hum Mol Genet. 2017 Sep 15; 26(18): 3482–3494. Published online 2017 Jun 19. doi: 10.1093/hmg/ddx234

PMCID:: PMC5886244

Abstract Article PDF–424K

Select item 56838982.

Antisense oligonucleotide therapy rescues disruptions in organization of exploratory movements associated with Usher syndrome type 1C in mice

Tia N. Donaldson, Kelsey T. Jennings, Lucia A. Cherep, Adam M. McNeela, Frederic F. Depreux, Francine M. Jodelka, Michelle L. Hastings, Douglas G. Wallace

Behav Brain Res. Author manuscript; available in PMC 2019 Feb 15.

Published in final edited form as: Behav Brain Res. 2018 Feb 15; 338: 76–87. Published online 2017 Oct 14. doi: 10.1016/j.bbr.2017.10.012

PMCID:: PMC5683898

Abstract Article PDF–4.8M

Select item 28932983.

Harmonin in the Murine Retina and the Retinal Phenotypes of Ush1c-Mutant Mice and Human USH1C

David S. Williams, Tomas S. Aleman, Concepción Lillo, Vanda S. Lopes, Louise C. Hughes, Edwin M. Stone, Samuel G. Jacobson

Invest Ophthalmol Vis Sci. Author manuscript; available in PMC 2010 Jun 29.

Published in final edited form as: Invest Ophthalmol Vis Sci. 2009 Aug; 50(8): 3881–3889. Published online 2009 Mar 25. doi: 10.1167/iovs.08-3358

PMCID:: PMC2893298

Abstract Article PDF–1.0M

Select item 27672394.

Harmonin-b, an actin-binding scaffold protein, is involved in the adaptation of mechanoelectrical transduction by sensory hair cells

Nicolas Michalski, Vincent Michel, Elisa Caberlotto, Gaelle M. Lefèvre, Alexander F. J. van Aken, Jean-Yves Tinevez, Emilie Bizard, Christophe Houbron, Dominique Weil, Jean-Pierre Hardelin, Guy P. Richardson, Corné J. Kros, Pascal Martin, Christine Petit

Pflugers Arch. 2009; 459(1): 115–130. Published online 2009 Sep 11. doi: 10.1007/s00424-009-0711-x

PMCID:: PMC2767239

Abstract Article PDF–844K

Select item 26913935.

Harmonin mutations cause mechanotransduction defects in cochlear hair cells

Nicolas Grillet, Wei Xiong, Anna Reynolds, Piotr Kazmierczak, Takashi Sato, Concepcion Lillo, Rachel A. Dumont, Edith Hintermann, Anna Sczaniecka, Martin Schwander, David Williams, Bechara Kachar, Peter G. Gillespie, Ulrich Müller

Neuron. Author manuscript; available in PMC 2010 May 14.

Published in final edited form as: Neuron. 2009 May 14; 62(3): 375–387. doi: 10.1016/j.neuron.2009.04.006

PMCID:: PMC2691393

Abstract Article PDF–4.1M

Select item 72298506.

Fetal antisense oligonucleotide therapy for congenital deafness and vestibular dysfunction

Lingyan Wang, J Beth Kempton, Han Jiang, Francine M Jodelka, Alev M Brigande, Rachel A Dumont, Frank Rigo, Jennifer J Lentz, Michelle L Hastings, John V Brigande

Nucleic Acids Res. 2020 May 21; 48(9): 5065–5080. Published online 2020 Apr 6. doi: 10.1093/nar/gkaa194

PMCID:: PMC7229850

Abstract Article PDF–8.7M

Select item 57924407.

Cone degeneration is triggered by the absence of USH1 proteins but prevented by antioxidant treatments

Alix Trouillet, Elisabeth Dubus, Julie Dégardin, Amrit Estivalet, Ivana Ivkovic, David Godefroy, Diego García-Ayuso, Manuel Simonutti, Iman Sahly, José A. Sahel, Aziz El-Amraoui, Christine Petit, Serge Picaud

Sci Rep. 2018; 8: 1968. Published online 2018 Jan 31. doi: 10.1038/s41598-018-20171-0

PMCID:: PMC5792440

Abstract Article PDF–3.9M

Select item 37172268.

Harmonin enhances voltage-dependent facilitation of Ca_v1.3 channels and synchronous exocytosis in mouse inner hair cells

Frederick D Gregory, Tina Pangršič, Irina E Calin-Jageman, Tobias Moser, Amy Lee

J Physiol. 2013 Jul 1; 591(Pt 13): 3253–3269. Published online 2013 Apr 22. doi: 10.1113/jphysiol.2013.254367

PMCID:: PMC3717226

Abstract Article PDF–1.4M

Select item 36577449.

Rescue of hearing and vestibular function in a mouse model of human deafness

Jennifer J. Lentz, Francine M. Jodelka, Anthony J. Hinrich, Kate E. McCaffrey, Hamilton E. Farris, Mathew J. Spalitta, Nicolas G. Bazan, Dominik M. Duelli, Frank Rigo, Michelle L. Hastings

Nat Med. Author manuscript; available in PMC 2013 Sep 1.

Published in final edited form as: Nat Med. 2013 Mar; 19(3): 345–350. Published online 2013 Feb 4. doi: 10.1038/nm.3106

PMCID:: PMC3657744

Abstract Article PDF–1.1M

Select item 316492010.

Harmonin inhibits presynaptic Ca_v1.3 Ca²⁺ channels in mouse inner hair cells

Frederick D. Gregory, Keith E. Bryan, Tina Pangršic, Irina E. Calin-Jageman, Tobias Moser, Amy Lee

Nat Neurosci. Author manuscript; available in PMC 2012 Mar 1.

Published in final edited form as: Nat Neurosci. 2011 Sep; 14(9): 1109–1111. Published online 2011 Aug 7. doi: 10.1038/nn.2895

PMCID:: PMC3164920

Abstract Article PDF–491K

Select item 290766311.

Ush1c gene expression levels in the ear and eye suggest different roles for Ush1c in neurosensory organs in a new Ush1c knockout mouse

Cong Tian, Xue Z. Liu, Fengchan Han, Heping Yu, Chantal Longo-Guess, Bin Yang, Changjun Lu, Denise Yan, Qing Y. Zheng

Brain Res. Author manuscript; available in PMC 2011 Apr 30.

Published in final edited form as: Brain Res. 2010 Apr 30; 1328: 57–70. Published online 2010 Mar 6. doi: 10.1016/j.brainres.2010.02.079

PMCID:: PMC2907663

Abstract Article PDF–5.6M

Select item 292812812.

Cadherin-23, myosin VIIa and harmonin, encoded by Usher syndrome type I genes, form a ternary complex and interact with membrane phospholipids

Amel Bahloul, Vincent Michel, Jean-Pierre Hardelin, Sylvie Nouaille, Sylviane Hoos, Anne Houdusse, Patrick England, Christine Petit

Hum Mol Genet. 2010 Sep 15; 19(18): 3557–3565. Published online 2010 Jul 16. doi: 10.1093/hmg/ddq271

PMCID:: PMC2928128

Abstract Article PDF–416K

Select item 292525013.

Deafness and Retinal Degeneration in A Novel USH1C Knock-In Mouse Model

Jennifer J. Lentz, William C. Gordon, Hamilton E. Farris, Glen H. MacDonald, Dale E. Cunningham, Carol A. Robbins, Bruce L. Tempel, Nicolas G. Bazan, Edwin W. Rubel, Elizabeth C. Oesterle, Bronya J. Keats

Dev Neurobiol. Author manuscript; available in PMC 2011 Mar 1.

Published in final edited form as: Dev Neurobiol. 2010 Mar; 70(4): 253–267. doi: 10.1002/dneu.20771

PMCID:: PMC2925250

Abstract Article PDF–1.2M

Select item 616731814.

Mechanotransduction is required for establishing and maintaining mature inner hair cells and regulating efferent innervation

Laura F. Corns, Stuart L. Johnson, Terri Roberts, Kishani M. Ranatunga, Aenea Hendry, Federico Ceriani, Saaid Safieddine, Karen P. Steel, Andy Forge, Christine Petit, David N. Furness, Corné J. Kros, Walter Marcotti

Nat Commun. 2018; 9: 4015. Published online 2018 Oct 1. doi: 10.1038/s41467-018-06307-w

PMCID:: PMC6167318

Abstract Article PDF–3.9M

Select item 333814615.

Stereocilin-deficient mice reveal the origin of cochlear waveform distortions

Elisabeth Verpy, Dominique Weil, Michel Leibovici, Richard J. Goodyear, Ghislaine Hamard, Carine Houdon, Gaelle M. Lefèvre, Jean-Pierre Hardelin, Guy P. Richardson, Paul Avan, Christine Petit

Nature. Author manuscript; available in PMC 2012 Apr 27.

Published in final edited form as: Nature. 2008 Nov 13; 456(7219): 255–258. Published online 2008 Oct 8. doi: 10.1038/nature07380