title: Nyctalopin is essential for synaptic transmission in the cone dominated zebrafish retina.
abstract: The first synapse in the vertebrate visual system is the photoreceptor synapse between rod and cone photoreceptors and the second-order bipolar cells. Although mutations in the nyctalopin gene (NYX) in humans lead to congenital stationary night blindness (CSNB1), affecting synaptic transmission between both types of photoreceptors and ON-bipolar cells, the function of nyctalopin in cone-dominant animal models has not been studied. Because the larval zebrafish retina is cone-dominant, we isolated the zebrafish nyx ortholog and raised a polyclonal antibody against the protein. Nyctalopin is expressed postsynaptically in both synaptic layers of the retina. Functional disruption via morpholino antisense injection leads to characteristic defects in the electroretinogram and defects in visual contrast sensitivity. We therefore demonstrated that nyctalopin plays a similar role in retinal synapse function in the cone pathway as in the rod pathway, thereby creating a genetic model for CSNB1 and its effects on cone vision.
authors.affiliation: Swiss Federal Institute of Technology (ETH) Zurich, Department of Biology, at the University Zurich, Zurich, Switzerland.
authors.forename: Ronja
authors.initials: R
authors.lastname: Bahadori
authors.forename: Oliver
authors.initials: O
authors.lastname: Biehlmaier
authors.forename: Christina
authors.initials: C
authors.lastname: Zeitz
authors.forename: Thomas
authors.initials: T
authors.lastname: Labhart
authors.forename: Yuri V
authors.initials: YV
authors.lastname: Makhankov
authors.forename: Ursula
authors.initials: U
authors.lastname: Forster
authors.forename: Matthias
authors.initials: M
authors.lastname: Gesemann
authors.forename: Wolfgang
authors.initials: W
authors.lastname: Berger
authors.forename: Stephan C F
authors.initials: SC
authors.lastname: Neuhauss
mesh_terms: D000818:Animals; D003001:Cloning, Molecular / Q000379:methods; D004596:Electroretinography / Q000379:methods; D006801:Humans; D007150:Immunohistochemistry / Q000379:methods; D017403:In Situ Hybridization / Q000379:methods; D047908:Intracellular Signaling Peptides and Proteins / Q000378:metabolism; D008562:Membrane Glycoproteins / Q000235:genetics / Q000378:metabolism; D009025:Morpholines / Q000037:antagonists & inhibitors / Q000737:chemistry; D017120:Nystagmus, Optokinetic / Q000187:drug effects / Q000502:physiology; D020319:Oligodeoxyribonucleotides, Antisense / Q000494:pharmacology; D011509:Proteoglycans / Q000235:genetics / Q000378:metabolism*; D012160:Retina / Q000166:cytology*; D017949:Retinal Cone Photoreceptor Cells / Q000187:drug effects / Q000378:metabolism*; D020539:Sequence Analysis, Protein; D009435:Synaptic Transmission / Q000502:physiology*; D014162:Transfection / Q000379:methods; D015027:Zebrafish; D029961:Zebrafish Proteins / Q000235:genetics / Q000378:metabolism
chemical_list: D047908:Intracellular Signaling Peptides and Proteins; D008562:Membrane Glycoproteins; D009025:Morpholines; C416505:NYX protein, human; C515870:NYX protein, zebrafish; D020319:Oligodeoxyribonucleotides, Antisense; D011509:Proteoglycans; D029961:Zebrafish Proteins; C037574:morpholine