Congenital Nystagmus Gene FRMD7 Is Necessary for Establishing a Neuronal Circuit Asymmetry for Direction Selectivity.

Details

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State: Public
Version: Final published version
Serval ID
serval:BIB_891621F3D894
Type
Article: article from journal or magazin.
Collection
Publications
Title
Congenital Nystagmus Gene FRMD7 Is Necessary for Establishing a Neuronal Circuit Asymmetry for Direction Selectivity.
Journal
Neuron
Author(s)
Yonehara K., Fiscella M., Drinnenberg A., Esposti F., Trenholm S., Krol J., Franke F., Scherf B.G., Kusnyerik A., Müller J., Szabo A., Jüttner J., Cordoba F., Reddy A.P., Németh J., Nagy Z.Z., Munier F., Hierlemann A., Roska B.
ISSN
1097-4199 (Electronic)
ISSN-L
0896-6273
Publication state
Published
Issued date
01/2016
Peer-reviewed
Oui
Volume
89
Number
1
Pages
177-193
Language
english
Notes
Publication types: Journal Article Publication Status: ppublish
Abstract
Neuronal circuit asymmetries are important components of brain circuits, but the molecular pathways leading to their establishment remain unknown. Here we found that the mutation of FRMD7, a gene that is defective in human congenital nystagmus, leads to the selective loss of the horizontal optokinetic reflex in mice, as it does in humans. This is accompanied by the selective loss of horizontal direction selectivity in retinal ganglion cells and the transition from asymmetric to symmetric inhibitory input to horizontal direction-selective ganglion cells. In wild-type retinas, we found FRMD7 specifically expressed in starburst amacrine cells, the interneuron type that provides asymmetric inhibition to direction-selective retinal ganglion cells. This work identifies FRMD7 as a key regulator in establishing a neuronal circuit asymmetry, and it suggests the involvement of a specific inhibitory neuron type in the pathophysiology of a neurological disease. VIDEO ABSTRACT.
Pubmed
Web of science
Open Access
Yes
Create date
29/01/2016 8:37
Last modification date
20/08/2019 14:48
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