Deletion of myosin VI causes slow retinal optic neuropathy and age-related macular degeneration (AMD)-relevant retinal phenotype

Publication type:
Journal article
Metadata:
Autoren
  • Timm Schubert
  • Corinna Gleiser
  • Peter Heiduschka
  • Christoph Franz
  • Kerstin Nagel-Wolfrum
  • Ayse Sahaboglu
  • Nicole Weisschuh
  • Gordon Eske
  • Karin Rohbock
  • Norman Rieger
  • Francois Paquet-Durand
  • Bernd Wissinger
  • Uwe Wolfrum
  • Bernhard Hirt
  • Wibke Singer
  • Lukas Ruettiger
  • Ulrike Zimmermann
  • Marlies Knipper
Autoren-URL
https://www.webofscience.com/api/gateway?GWVersion=2&SrcApp=fis-test-1&SrcAuth=WosAPI&KeyUT=WOS:000361566700010&DestLinkType=FullRecord&DestApp=WOS_CPL
DOI
10.1007/s00018-015-1913-3
eISSN
1420-9071
Externe Identifier
  • Clarivate Analytics Document Solution ID: CR7XZ
  • PubMed Identifier: 25939269
ISSN
1420-682X
Ausgabe der Veröffentlichung
20
Zeitschrift
CELLULAR AND MOLECULAR LIFE SCIENCES
Schlüsselwörter
  • Bipolar cell
  • Inner retina
  • Outer retina
  • Synapse
  • Stereocilia
  • Translocator protein TSPO
  • Choriocapillaris
  • Mouse model
Paginierung
3953 - 3969
Datum der Veröffentlichung
2015
Status
Published
Titel
Deletion of myosin VI causes slow retinal optic neuropathy and age-related macular degeneration (AMD)-relevant retinal phenotype
Sub types
  • Article
Ausgabe der Zeitschrift
72

Data source: Web of Science (Lite)

Other metadata sources:
Autoren
  • Timm Schubert
  • Corinna Gleiser
  • Peter Heiduschka
  • Christoph Franz
  • Kerstin Nagel-Wolfrum
  • Ayse Sahaboglu
  • Nicole Weisschuh
  • Gordon Eske
  • Karin Rohbock
  • Norman Rieger
  • François Paquet-Durand
  • Bernd Wissinger
  • Uwe Wolfrum
  • Bernhard Hirt
  • Wibke Singer
  • Lukas Rüttiger
  • Ulrike Zimmermann
  • Marlies Knipper
DOI
10.1007/s00018-015-1913-3
eISSN
1420-9071
ISSN
1420-682X
Ausgabe der Veröffentlichung
20
Zeitschrift
Cellular and Molecular Life Sciences
Sprache
en
Online publication date
2015
Paginierung
3953 - 3969
Datum der Veröffentlichung
2015
Status
Published
Herausgeber
Springer Science and Business Media LLC
Herausgeber URL
http://dx.doi.org/10.1007/s00018-015-1913-3
Datum der Datenerfassung
2019
Titel
Deletion of myosin VI causes slow retinal optic neuropathy and age-related macular degeneration (AMD)-relevant retinal phenotype
Ausgabe der Zeitschrift
72

Data source: Crossref

Abstract
The unconventional myosin VI, a member of the actin-based motor protein family of myosins, is expressed in the retina. Its deletion was previously shown to reduce amplitudes of the a- and b-waves of the electroretinogram. Analyzing wild-type and myosin VI-deficient Snell's Waltzer mice in more detail, the expression pattern of myosin VI in retinal pigment epithelium, outer limiting membrane, and outer plexiform layer could be linked with differential progressing ocular deficits. These encompassed reduced a-waves and b-waves and disturbed oscillatory potentials in the electroretinogram, photoreceptor cell death, retinal microglia infiltration, and formation of basal laminar deposits. A phenotype comprising features of glaucoma (neurodegeneration) and age-related macular degeneration could thus be uncovered that suggests dysfunction of myosin VI and its variable cargo adaptor proteins for membrane sorting and autophagy, as possible candidate mediators for both disease forms.
Addresses
  • Werner Reichardt Centre for Integrative Neuroscience (CIN)/Institute for Ophthalmic Research, University of Tübingen, Otfried-Müller-Str. 25, 72076, Tübingen, Germany. timm.schubert@uni-tuebingen.de.
Autoren
  • Timm Schubert
  • Corinna Gleiser
  • Peter Heiduschka
  • Christoph Franz
  • Kerstin Nagel-Wolfrum
  • Ayse Sahaboglu
  • Nicole Weisschuh
  • Gordon Eske
  • Karin Rohbock
  • Norman Rieger
  • François Paquet-Durand
  • Bernd Wissinger
  • Uwe Wolfrum
  • Bernhard Hirt
  • Wibke Singer
  • Lukas Rüttiger
  • Ulrike Zimmermann
  • Marlies Knipper
DOI
10.1007/s00018-015-1913-3
eISSN
1420-9071
Externe Identifier
  • PubMed Identifier: 25939269
  • PubMed Central ID: PMC4575690
Funding acknowledgements
  • The European Community: FP7/2009/241955 (SYSCILIA)</
  • The ProRetina:
  • E-Rare-2, The ERA-Net for Research on Rare Diseases No 58: EUR-USH
  • Werner Reichardt Centre for Neuroscience, CIN: EXC 307
  • RNID: G54
  • The Deutsche Forschungsgemeinschaft, DFG: KN 316-10-1
  • The Kerstan Foundation Tübingen:
  • The Deutsche Forschungsgemeinschaft, DFG: FOR 2060
  • Rhe Royal National Institute for Deaf People, RNID: G54
  • The FAUN Stiftung Nuremberg:
  • The Deutsche Forschungsgemeinschaft, DFG: GRK 1044
  • The Foundation Fighting Blindness, FFP: TA-NMT-0611-0538-JGU
  • The European Community: FP7/2009/242013 (TREATRUSH)
  • The Bundesministerium für Bildung und Forschung, BMBF: 0314106 (HOPE2)
Open access
true
ISSN
1420-682X
Ausgabe der Veröffentlichung
20
Zeitschrift
Cellular and molecular life sciences : CMLS
Schlüsselwörter
  • Microglia
  • Retina
  • Animals
  • Mice, Inbred C57BL
  • Mice
  • Optic Nerve Diseases
  • Macular Degeneration
  • Myosin Heavy Chains
  • Gene Deletion
  • Genotype
  • Photoreceptor Cells, Vertebrate
Sprache
eng
Medium
Print-Electronic
Online publication date
2015
Open access status
Open Access
Paginierung
3953 - 3969
Datum der Veröffentlichung
2015
Status
Published
Publisher licence
CC BY
Datum der Datenerfassung
2015
Titel
Deletion of myosin VI causes slow retinal optic neuropathy and age-related macular degeneration (AMD)-relevant retinal phenotype.
Sub types
  • Research Support, Non-U.S. Gov't
  • research-article
  • Journal Article
Ausgabe der Zeitschrift
72

Files

https://link.springer.com/content/pdf/10.1007/s00018-015-1913-3.pdf https://europepmc.org/articles/PMC4575690?pdf=render

Data source: Europe PubMed Central

Abstract
The unconventional myosin VI, a member of the actin-based motor protein family of myosins, is expressed in the retina. Its deletion was previously shown to reduce amplitudes of the a- and b-waves of the electroretinogram. Analyzing wild-type and myosin VI-deficient Snell's Waltzer mice in more detail, the expression pattern of myosin VI in retinal pigment epithelium, outer limiting membrane, and outer plexiform layer could be linked with differential progressing ocular deficits. These encompassed reduced a-waves and b-waves and disturbed oscillatory potentials in the electroretinogram, photoreceptor cell death, retinal microglia infiltration, and formation of basal laminar deposits. A phenotype comprising features of glaucoma (neurodegeneration) and age-related macular degeneration could thus be uncovered that suggests dysfunction of myosin VI and its variable cargo adaptor proteins for membrane sorting and autophagy, as possible candidate mediators for both disease forms.
Date of acceptance
2015
Autoren
  • Timm Schubert
  • Corinna Gleiser
  • Peter Heiduschka
  • Christoph Franz
  • Kerstin Nagel-Wolfrum
  • Ayse Sahaboglu
  • Nicole Weisschuh
  • Gordon Eske
  • Karin Rohbock
  • Norman Rieger
  • François Paquet-Durand
  • Bernd Wissinger
  • Uwe Wolfrum
  • Bernhard Hirt
  • Wibke Singer
  • Lukas Rüttiger
  • Ulrike Zimmermann
  • Marlies Knipper
Autoren-URL
https://www.ncbi.nlm.nih.gov/pubmed/25939269
DOI
10.1007/s00018-015-1913-3
eISSN
1420-9071
Externe Identifier
  • PubMed Central ID: PMC4575690
Funding acknowledgements
  • RNID: G54
Ausgabe der Veröffentlichung
20
Zeitschrift
Cell Mol Life Sci
Schlüsselwörter
  • Bipolar cell
  • Choriocapillaris
  • Inner retina
  • Mouse model
  • Outer retina
  • Stereocilia
  • Synapse
  • Translocator protein TSPO
  • Animals
  • Gene Deletion
  • Genotype
  • Macular Degeneration
  • Mice
  • Mice, Inbred C57BL
  • Microglia
  • Myosin Heavy Chains
  • Optic Nerve Diseases
  • Photoreceptor Cells, Vertebrate
  • Retina
Sprache
eng
Country
Switzerland
Paginierung
3953 - 3969
PII
10.1007/s00018-015-1913-3
Datum der Veröffentlichung
2015
Status
Published
Datum, an dem der Datensatz öffentlich gemacht wurde
2015
Titel
Deletion of myosin VI causes slow retinal optic neuropathy and age-related macular degeneration (AMD)-relevant retinal phenotype.
Sub types
  • Journal Article
  • Research Support, Non-U.S. Gov't
Ausgabe der Zeitschrift
72

Data source: PubMed

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