Literature DB >> 25125600

Transgenic mice expressing mutated Tyr437His human myocilin develop progressive loss of retinal ganglion cell electrical responsiveness and axonopathy with normal iop.

Tsung-Han Chou1, Stanislav Tomarev2, Vittorio Porciatti1.   

Abstract

PURPOSE: To characterize age-related changes of retinal ganglion cell (RGC) function, IOP, and anatomical markers of axon/glia integrity in a transgenic mouse expressing Tyr437His mutant of human myocilin protein.
METHODS: Retinal ganglion cell electrical responsiveness was tested with pattern electroretinogram (PERG) in 11 transgenic mice expressing mutated myocilin at different ages over 18 months under ketamine/xylazine anesthesia. Twelve age-matched C57BL/6J mice also were tested as controls. Intraocular pressure was measured with a Tonolab tonometer. Immunohistochemistry for GFAP and neurofilament was performed on dissected optic nerve heads.
RESULTS: In transgenic mice expressing mutated myocilin, the PERG amplitude progressively decreased with increasing age by approximately 50%, whereas the PERG peak latency increased by approximately 40 ms (ANOVA, P < 0.05). In contrast, PERGs of young and old control mice had similar amplitudes and peak latencies. In transgenic mice, GFAP staining was more intense and extended than in control mice, and increased with increasing age; neurofilament staining showed swollen and partially degenerated axons in old transgenic mice. The IOP of young transgenic mice was similar to that of control mice and did not significantly change with increasing age.
CONCLUSIONS: Transgenic mice expressing mutated human myocilin display progressive age-related changes in RGC electrical responsiveness that are not associated with IOP elevation but are associated with marked astrogliosis and axonopathy. Our results support the view that MYOC expression in the optic nerve may impact structural, metabolic, or neurotrophic support to RGC axons, thereby influencing their susceptibility to glaucomatous damage independently of IOP. Copyright 2014 The Association for Research in Vision and Ophthalmology, Inc.

Entities:  

Keywords:  intraocular pressure; mouse; myocylin; optic nerve; pattern electroretinogram; retinal ganglion cell

Mesh:

Substances:

Year:  2014        PMID: 25125600      PMCID: PMC4160076          DOI: 10.1167/iovs.14-14793

Source DB:  PubMed          Journal:  Invest Ophthalmol Vis Sci        ISSN: 0146-0404            Impact factor:   4.799


  57 in total

1.  Noninvasive determination of intraocular pressure (IOP) in nonsedated mice of 5 different inbred strains.

Authors:  Nicholas Nissirios; David Goldblum; Kaspar Rohrer; Thomas Mittag; John Danias
Journal:  J Glaucoma       Date:  2007-01       Impact factor: 2.503

2.  The pattern electroretinogram as a tool to monitor progressive retinal ganglion cell dysfunction in the DBA/2J mouse model of glaucoma.

Authors:  Vittorio Porciatti; Maher Saleh; Mahesh Nagaraju
Journal:  Invest Ophthalmol Vis Sci       Date:  2007-02       Impact factor: 4.799

3.  Inherited glaucoma in DBA/2J mice: pertinent disease features for studying the neurodegeneration.

Authors:  Richard T Libby; Michael G Anderson; Iok-Hou Pang; Zachary H Robinson; Olga V Savinova; I Mihai Cosma; Amy Snow; Lawriston A Wilson; Richard S Smith; Abbot F Clark; Simon W M John
Journal:  Vis Neurosci       Date:  2005 Sep-Oct       Impact factor: 3.241

4.  Expression of mutated mouse myocilin induces open-angle glaucoma in transgenic mice.

Authors:  Vladimir Senatorov; Irina Malyukova; Robert Fariss; Eric F Wawrousek; Srividya Swaminathan; Shyam K Sharan; Stanislav Tomarev
Journal:  J Neurosci       Date:  2006-11-15       Impact factor: 6.167

5.  Glaucoma-causing myocilin mutants require the Peroxisomal targeting signal-1 receptor (PTS1R) to elevate intraocular pressure.

Authors:  Allan R Shepard; Nasreen Jacobson; J Cameron Millar; Iok-Hou Pang; H Thomas Steely; Charles C Searby; Val C Sheffield; Edwin M Stone; Abbot F Clark
Journal:  Hum Mol Genet       Date:  2007-02-22       Impact factor: 6.150

6.  Longitudinal evaluation of retinal ganglion cell function and IOP in the DBA/2J mouse model of glaucoma.

Authors:  Maher Saleh; Mahesh Nagaraju; Vittorio Porciatti
Journal:  Invest Ophthalmol Vis Sci       Date:  2007-10       Impact factor: 4.799

7.  IOP-dependent retinal ganglion cell dysfunction in glaucomatous DBA/2J mice.

Authors:  Mahesh Nagaraju; Maher Saleh; Vittorio Porciatti
Journal:  Invest Ophthalmol Vis Sci       Date:  2007-10       Impact factor: 4.799

8.  Retinal ganglion cells downregulate gene expression and lose their axons within the optic nerve head in a mouse glaucoma model.

Authors:  Ileana Soto; Ericka Oglesby; Brian P Buckingham; Janice L Son; Elisha D O Roberson; Michael R Steele; Denise M Inman; Monica L Vetter; Philip J Horner; Nicholas Marsh-Armstrong
Journal:  J Neurosci       Date:  2008-01-09       Impact factor: 6.167

9.  Genetic context determines susceptibility to intraocular pressure elevation in a mouse pigmentary glaucoma.

Authors:  Michael G Anderson; Richard T Libby; Mao Mao; Ioan M Cosma; Larry A Wilson; Richard S Smith; Simon W M John
Journal:  BMC Biol       Date:  2006-07-07       Impact factor: 7.431

10.  Axons of retinal ganglion cells are insulted in the optic nerve early in DBA/2J glaucoma.

Authors:  Gareth R Howell; Richard T Libby; Tatjana C Jakobs; Richard S Smith; F Campbell Phalan; Joseph W Barter; Jessica M Barbay; Jeffrey K Marchant; Nagaraju Mahesh; Vittorio Porciatti; Alan V Whitmore; Richard H Masland; Simon W M John
Journal:  J Cell Biol       Date:  2007-12-24       Impact factor: 10.539

View more
  5 in total

Review 1.  Electrophysiological assessment of retinal ganglion cell function.

Authors:  Vittorio Porciatti
Journal:  Exp Eye Res       Date:  2015-05-18       Impact factor: 3.467

Review 2.  Major review: Molecular genetics of primary open-angle glaucoma.

Authors:  Yutao Liu; R Rand Allingham
Journal:  Exp Eye Res       Date:  2017-05-10       Impact factor: 3.467

Review 3.  Evaluating retinal ganglion cell loss and dysfunction.

Authors:  Ben Mead; Stanislav Tomarev
Journal:  Exp Eye Res       Date:  2016-08-12       Impact factor: 3.467

4.  Binding of a glaucoma-associated myocilin variant to the αB-crystallin chaperone impedes protein clearance in trabecular meshwork cells.

Authors:  Jeffrey M Lynch; Bing Li; Parvaneh Katoli; Chuanxi Xiang; Barrett Leehy; Nalini Rangaswamy; Veronica Saenz-Vash; Y Karen Wang; Hong Lei; Thomas B Nicholson; Erik Meredith; Dennis S Rice; Ganesh Prasanna; Amy Chen
Journal:  J Biol Chem       Date:  2018-11-02       Impact factor: 5.157

Review 5.  Common and rare myocilin variants: Predicting glaucoma pathogenicity based on genetics, clinical, and laboratory misfolding data.

Authors:  Hailee F Scelsi; Brett M Barlow; Emily G Saccuzzo; Raquel L Lieberman
Journal:  Hum Mutat       Date:  2021-06-24       Impact factor: 4.700

  5 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.