The giant spectrin V couples the molecular motors to phototransduction and Usher syndrome type I proteins along their trafficking route
- Publikationstyp:
- Zeitschriftenaufsatz
- Metadaten:
-
- Autoren
- Samantha Papal
- Matteo Cortese
- Kirian Legendre
- Nasrin Sorusch
- Joseph Dragavon
- Iman Sahly
- Spencer Shorte
- Uwe Wolfrum
- Christine Petit
- Aziz El-Amraoui
- Autoren-URL
- https://www.webofscience.com/api/gateway?GWVersion=2&SrcApp=fis-test-1&SrcAuth=WosAPI&KeyUT=WOS:000323582000013&DestLinkType=FullRecord&DestApp=WOS_CPL
- DOI
- 10.1093/hmg/ddt228
- eISSN
- 1460-2083
- Externe Identifier
- Clarivate Analytics Document Solution ID: 207FD
- PubMed Identifier: 23704327
- ISSN
- 0964-6906
- Ausgabe der Veröffentlichung
- 18
- Zeitschrift
- HUMAN MOLECULAR GENETICS
- Paginierung
- 3773 - 3788
- Datum der Veröffentlichung
- 2013
- Status
- Published
- Titel
- The giant spectrin V couples the molecular motors to phototransduction and Usher syndrome type I proteins along their trafficking route
- Sub types
- Article
- Ausgabe der Zeitschrift
- 22
Datenquelle: Web of Science (Lite)
- Andere Metadatenquellen:
-
- Autoren
- Samantha Papal
- Matteo Cortese
- Kirian Legendre
- Nasrin Sorusch
- Joseph Dragavon
- Iman Sahly
- Spencer Shorte
- Uwe Wolfrum
- Christine Petit
- Aziz El-Amraoui
- DOI
- 10.1093/hmg/ddt228
- eISSN
- 1460-2083
- ISSN
- 0964-6906
- Ausgabe der Veröffentlichung
- 18
- Zeitschrift
- Human Molecular Genetics
- Sprache
- en
- Online publication date
- 2013
- Paginierung
- 3773 - 3788
- Datum der Veröffentlichung
- 2013
- Status
- Published
- Herausgeber
- Oxford University Press (OUP)
- Herausgeber URL
- http://dx.doi.org/10.1093/hmg/ddt228
- Datum der Datenerfassung
- 2022
- Titel
- The giant spectrin βV couples the molecular motors to phototransduction and Usher syndrome type I proteins along their trafficking route
- Ausgabe der Zeitschrift
- 22
Datenquelle: Crossref
- Abstract
- Mutations in the myosin VIIa gene cause Usher syndrome type IB (USH1B), characterized by deaf-blindness. A delay of opsin trafficking has been observed in the retinal photoreceptor cells of myosin VIIa-deficient mice. We identified spectrin βV, the mammalian β-heavy spectrin, as a myosin VIIa- and rhodopsin-interacting partner in photoreceptor cells. Spectrin βV displays a polarized distribution from the Golgi apparatus to the base of the outer segment, which, unlike that of other β spectrins, matches the trafficking route of opsin and other phototransduction proteins. Formation of spectrin βV-rhodopsin complex could be detected in the differentiating photoreceptors as soon as their outer segment emerges. A failure of the spectrin βV-mediated coupling between myosin VIIa and opsin molecules thus probably accounts for the opsin transport delay in myosin VIIa-deficient mice. We showed that spectrin βV also associates with two USH1 proteins, sans (USH1G) and harmonin (USH1C). Spectrins are supposed to function as heteromers of α and β subunits, but fluorescence resonance energy transfer and in vitro binding experiments indicated that spectrin βV can also form homodimers, which likely supports its αII-independent βV functions. Finally, consistent with its distribution along the connecting cilia axonemes, spectrin βV binds to several subunits of the microtubule-based motor proteins, kinesin II and the dynein complex. We therefore suggest that spectrin βV homomers couple some USH1 proteins, opsin and other phototransduction proteins to both actin- and microtubule-based motors, thereby contributing to their transport towards the photoreceptor outer disks.
- Addresses
- Institut Pasteur, Unité de génétique et physiologie de l'audition, Paris, France.
- Autoren
- Samantha Papal
- Matteo Cortese
- Kirian Legendre
- Nasrin Sorusch
- Joseph Dragavon
- Iman Sahly
- Spencer Shorte
- Uwe Wolfrum
- Christine Petit
- Aziz El-Amraoui
- DOI
- 10.1093/hmg/ddt228
- eISSN
- 1460-2083
- Externe Identifier
- PubMed Identifier: 23704327
- Open access
- false
- ISSN
- 0964-6906
- Ausgabe der Veröffentlichung
- 18
- Zeitschrift
- Human molecular genetics
- Schlüsselwörter
- Retina
- Hela Cells
- Animals
- Humans
- Mice
- Microtubule Proteins
- Myosins
- Rhodopsin
- Adaptor Proteins, Signal Transducing
- Spectrin
- Cell Cycle Proteins
- Cytoskeletal Proteins
- Usher Syndromes
- Photoreceptor Cells, Vertebrate
- Light Signal Transduction
- Myosin VIIa
- Sprache
- eng
- Medium
- Print-Electronic
- Online publication date
- 2013
- Paginierung
- 3773 - 3788
- Datum der Veröffentlichung
- 2013
- Status
- Published
- Datum der Datenerfassung
- 2013
- Titel
- The giant spectrin βV couples the molecular motors to phototransduction and Usher syndrome type I proteins along their trafficking route.
- Sub types
- Research Support, Non-U.S. Gov't
- Journal Article
- Ausgabe der Zeitschrift
- 22
Datenquelle: Europe PubMed Central
- Abstract
- Mutations in the myosin VIIa gene cause Usher syndrome type IB (USH1B), characterized by deaf-blindness. A delay of opsin trafficking has been observed in the retinal photoreceptor cells of myosin VIIa-deficient mice. We identified spectrin βV, the mammalian β-heavy spectrin, as a myosin VIIa- and rhodopsin-interacting partner in photoreceptor cells. Spectrin βV displays a polarized distribution from the Golgi apparatus to the base of the outer segment, which, unlike that of other β spectrins, matches the trafficking route of opsin and other phototransduction proteins. Formation of spectrin βV-rhodopsin complex could be detected in the differentiating photoreceptors as soon as their outer segment emerges. A failure of the spectrin βV-mediated coupling between myosin VIIa and opsin molecules thus probably accounts for the opsin transport delay in myosin VIIa-deficient mice. We showed that spectrin βV also associates with two USH1 proteins, sans (USH1G) and harmonin (USH1C). Spectrins are supposed to function as heteromers of α and β subunits, but fluorescence resonance energy transfer and in vitro binding experiments indicated that spectrin βV can also form homodimers, which likely supports its αII-independent βV functions. Finally, consistent with its distribution along the connecting cilia axonemes, spectrin βV binds to several subunits of the microtubule-based motor proteins, kinesin II and the dynein complex. We therefore suggest that spectrin βV homomers couple some USH1 proteins, opsin and other phototransduction proteins to both actin- and microtubule-based motors, thereby contributing to their transport towards the photoreceptor outer disks.
- Autoren
- Samantha Papal
- Matteo Cortese
- Kirian Legendre
- Nasrin Sorusch
- Joseph Dragavon
- Iman Sahly
- Spencer Shorte
- Uwe Wolfrum
- Christine Petit
- Aziz El-Amraoui
- Autoren-URL
- https://www.ncbi.nlm.nih.gov/pubmed/23704327
- DOI
- 10.1093/hmg/ddt228
- eISSN
- 1460-2083
- Ausgabe der Veröffentlichung
- 18
- Zeitschrift
- Hum Mol Genet
- Schlüsselwörter
- Adaptor Proteins, Signal Transducing
- Animals
- Cell Cycle Proteins
- Cytoskeletal Proteins
- HeLa Cells
- Humans
- Light Signal Transduction
- Mice
- Microtubule Proteins
- Myosin VIIa
- Myosins
- Photoreceptor Cells, Vertebrate
- Retina
- Rhodopsin
- Spectrin
- Usher Syndromes
- Sprache
- eng
- Country
- England
- Paginierung
- 3773 - 3788
- PII
- ddt228
- Datum der Veröffentlichung
- 2013
- Status
- Published
- Datum, an dem der Datensatz öffentlich gemacht wurde
- 2014
- Titel
- The giant spectrin βV couples the molecular motors to phototransduction and Usher syndrome type I proteins along their trafficking route.
- Sub types
- Journal Article
- Research Support, Non-U.S. Gov't
- Ausgabe der Zeitschrift
- 22
Datenquelle: PubMed
- Beziehungen:
- Eigentum von