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Publisher Correction: Moderate Nucleoporin 133 deficiency leads to glomerular damage in zebrafish.

Chiara Cianciolo Cosentino, Alessandro Berto, Stéphane Pelletier, Michelle Hari, Johannes Loffing, Stephan C F Neuhauss, Valérie Doye,

An amendment to this paper has been published and can be accessed via a link at the top of the paper. ... Read more >>

Sci Rep (Scientific reports)
[2020, 10(1):3326]

Cited: 0 times

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Author Correction: Moderate Nucleoporin 133 deficiency leads to glomerular damage in zebrafish.

Chiara Cianciolo Cosentino, Alessandro Berto, Stéphane Pelletier, Michelle Hari, Johannes Loffing, Stephan C F Neuhauss, Valérie Doye,

An amendment to this paper has been published and can be accessed via a link at the top of the paper. ... Read more >>

Sci Rep (Scientific reports)
[2020, 10(1):756]

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Photoreceptor Degeneration Accompanies Vascular Changes in a Zebrafish Model of Diabetic Retinopathy.

Zaheer Ali, Jingjing Zang, Neil Lagali, Nicole Schmitner, Willi Salvenmoser, Anthony Mukwaya, Stephan C F Neuhauss, Lasse D Jensen, Robin A Kimmel,

Purpose:Diabetic retinopathy (DR) is a leading cause of vision impairment and blindness worldwide in the working-age population, and the incidence is rising. Until now it has been difficult to define initiating events and disease progression at the molecular level, as available diabetic rodent models do not present the full spectrum of ... Read more >>

Invest. Ophthalmol. Vis. Sci. (Investigative ophthalmology & visual science)
[2020, 61(2):43]

Cited: 0 times

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Transgenic zebrafish modeling low-molecular-weight proteinuria and lysosomal storage diseases.

Zhiyong Chen, Alessandro Luciani, José María Mateos, Gery Barmettler, Rachel H Giles, Stephan C F Neuhauss, Olivier Devuyst,

Epithelial cells lining the proximal tubule of the kidney reabsorb and metabolize most of the filtered low-molecular-weight proteins through receptor-mediated endocytosis and lysosomal processing. Congenital and acquired dysfunctions of the proximal tubule are consistently reflected by the inappropriate loss of solutes including low-molecular-weight proteins in the urine. The zebrafish pronephros ... Read more >>

Kidney Int. (Kidney International)
[2020, 97(6):1150-1163]

Cited: 0 times

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A New Zebrafish Model for CACNA2D4-Dysfunction.

Domino K Schlegel, Stella M K Glasauer, José M Mateos, Gery Barmettler, Urs Ziegler, Stephan C F Neuhauss,

Purpose:Mutations in CACNA2D4, encoding the α2δ4 subunit of retinal voltage-gated calcium channels (Cav), cause a rare type of retinal dysfunction in human, mainly affecting cone vision. Here, we investigate the role of CACNA2D4 in targeting of Cav, its influence on cone-mediated signal transmission, and the cellular and subcellular changes upon ... Read more >>

Invest. Ophthalmol. Vis. Sci. (Investigative ophthalmology & visual science)
[2019, 60(15):5124-5135]

Cited: 0 times

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Moderate Nucleoporin 133 deficiency leads to glomerular damage in zebrafish.

Chiara Cianciolo Cosentino, Alessandro Berto, Stéphane Pelletier, Michelle Hari, Johannes Loffing, Stephan C F Neuhauss, Valérie Doye,

Although structural nuclear pore proteins (nucleoporins) are seemingly required in every cell type to assemble a functional nuclear transport machinery, mutations or deregulation of a subset of them have been associated with specific human hereditary diseases. In particular, previous genetic studies of patients with nephrotic syndrome identified mutations in Nup107 ... Read more >>

Sci Rep (Scientific reports)
[2019, 9(1):4750]

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Differential expression of PKCα and -β in the zebrafish retina.

Marion F Haug, Manuela Berger, Matthias Gesemann, Stephan C F Neuhauss,

The retina is a complex neural circuit, which processes and transmits visual information from light perceiving photoreceptors to projecting retinal ganglion cells. Much of the computational power of the retina rests on signal integrating interneurons, such as bipolar cells. Commercially available antibodies against bovine and human conventional protein kinase C ... Read more >>

Histochem. Cell Biol. (Histochemistry and cell biology)
[2019, 151(6):521-530]

Cited: 0 times

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The Binding Properties and Physiological Functions of Recoverin.

Jingjing Zang, Stephan C F Neuhauss,

Recoverin (Rcv) is a low molecular-weight, neuronal calcium sensor (NCS) primarily located in photoreceptor outer segments of the vertebrate retina. Calcium ions (Ca2+)-bound Rcv has been proposed to inhibit G-protein-coupled receptor kinase (GRKs) in darkness. During the light response, the Ca2+-free Rcv releases GRK, which in turn phosphorylates visual pigment, ... Read more >>

Front Mol Neurosci (Frontiers in molecular neuroscience)
[2018, 11:473]

Cited: 2 times

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Publisher Correction: The ciliopathy protein TALPID3/KIAA0586 acts upstream of Rab8 activation in zebrafish photoreceptor outer segment formation and maintenance.

Irene Ojeda Naharros, Flavia B Cristian, Jingjing Zang, Matthias Gesemann, Philip W Ingham, Stephan C F Neuhauss, Ruxandra Bachmann-Gagescu,

A correction to this article has been published and is linked from the HTML and PDF versions of this paper. The error has been fixed in the paper. ... Read more >>

Sci Rep (Scientific reports)
[2018, 8(1):12534]

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Sensory Biology: How to Structure a Tailor-Made Retina.

Stephan C F Neuhauss,

A new study of the zebrafish retina using sophisticated imaging has revealed how anisotropic properties of the retina are closely matched to the statistics of the natural visual world that the fish experiences. ... Read more >>

Curr. Biol. (Current biology : CB)
[2018, 28(13):R737-R739]

Cited: 0 times

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The ciliopathy protein TALPID3/KIAA0586 acts upstream of Rab8 activation in zebrafish photoreceptor outer segment formation and maintenance.

Irene Ojeda Naharros, Flavia B Cristian, Jingjing Zang, Matthias Gesemann, Philip W Ingham, Stephan C F Neuhauss, Ruxandra Bachmann-Gagescu,

Ciliopathies are human disorders caused by dysfunction of primary cilia, ubiquitous microtubule-based organelles involved in signal transduction. Cilia are anchored inside the cell through basal bodies (BBs), modified centrioles also acting as microtubule-organization centers. Photoreceptors (PRs) are sensory neurons, whose primary cilium forms a highly specialized compartment called the outer ... Read more >>

Sci Rep (Scientific reports)
[2018, 8(1):2211]

Cited: 1 time

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Phylogeny and distribution of protein kinase C variants in the zebrafish.

Marion F Haug, Matthias Gesemann, Manuela Berger, Stephan C F Neuhauss,

Conventional protein kinases-consisting of α, β, and γ family members-play key roles in numerous signal transduction events. Phylogenetic analysis demonstrated the existence of five prkcs (the genes representing PKCs) in zebrafish, two paralogous forms of prkca and prkcb and one prkcg variant. mRNA expression analysis showed distinct, mainly nervous system ... Read more >>

J. Comp. Neurol. (The Journal of comparative neurology)
[2018, 526(7):1097-1109]

Cited: 1 time

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Guidelines for morpholino use in zebrafish.

Didier Y R Stainier, Erez Raz, Nathan D Lawson, Stephen C Ekker, Rebecca D Burdine, Judith S Eisen, Philip W Ingham, Stefan Schulte-Merker, Deborah Yelon, Brant M Weinstein, Mary C Mullins, Stephen W Wilson, Lalita Ramakrishnan, Sharon L Amacher, Stephan C F Neuhauss, Anming Meng, Naoki Mochizuki, Pertti Panula, Cecilia B Moens,

PLoS Genet. (PLoS genetics)
[2017, 13(10):e1007000]

Cited: 54 times

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Correlative Super-resolution and Electron Microscopy to Resolve Protein Localization in Zebrafish Retina.

José M Mateos, Gery Barmettler, Jana Doehner, Irene Ojeda Naharros, Bruno Guhl, Stephan C F Neuhauss, Andres Kaech, Ruxandra Bachmann-Gagescu, Urs Ziegler,

We present a method to investigate the subcellular protein localization in the larval zebrafish retina by combining super-resolution light microscopy and scanning electron microscopy. The sub-diffraction limit resolution capabilities of super-resolution light microscopes allow improving the accuracy of the correlated data. Briefly, 110 nanometer thick cryo-sections are transferred to a ... Read more >>

J Vis Exp (Journal of visualized experiments : JoVE)
[2017, (129):]

Cited: 1 time

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Comparative transcriptomic analysis identifies evolutionarily conserved gene products in the vertebrate renal distal convoluted tubule.

Yuya Sugano, Chiara Cianciolo Cosentino, Dominique Loffing-Cueni, Stephan C F Neuhauss, Johannes Loffing,

Understanding the molecular basis of the complex regulatory networks controlling renal ion transports is of major physiological and clinical importance. In this study, we aimed to identify evolutionarily conserved critical players in the function of the renal distal convoluted tubule (DCT) by a comparative transcriptomic approach. We generated a transgenic ... Read more >>

Pflugers Arch. (Pflugers Archiv : European journal of physiology)
[2017, 469(7-8):859-867]

Cited: 2 times

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Mutations in ARMC9, which Encodes a Basal Body Protein, Cause Joubert Syndrome in Humans and Ciliopathy Phenotypes in Zebrafish.

Julie C Van De Weghe, Tamara D S Rusterholz, Brooke Latour, Megan E Grout, Kimberly A Aldinger, Ranad Shaheen, Jennifer C Dempsey, Sateesh Maddirevula, Yong-Han H Cheng, Ian G Phelps, Matthias Gesemann, Himanshu Goel, Ohad S Birk, Talal Alanzi, Rifaat Rawashdeh, Arif O Khan, , Michael J Bamshad, Deborah A Nickerson, Stephan C F Neuhauss, William B Dobyns, Fowzan S Alkuraya, Ronald Roepman, Ruxandra Bachmann-Gagescu, Dan Doherty,

Joubert syndrome (JS) is a recessive neurodevelopmental disorder characterized by hypotonia, ataxia, abnormal eye movements, and variable cognitive impairment. It is defined by a distinctive brain malformation known as the "molar tooth sign" on axial MRI. Subsets of affected individuals have malformations such as coloboma, polydactyly, and encephalocele, as well ... Read more >>

Am. J. Hum. Genet. (American journal of human genetics)
[2017, 101(1):23-36]

Cited: 12 times

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Genetic approaches to retinal research in zebrafish.

Stephanie Niklaus, Stephan C F Neuhauss,

The zebrafish (Danio rerio) possesses a vertebrate-type retina that is extraordinarily conserved in evolution. This well-organized and anatomically easily accessible part of the central nervous system has been widely investigated in zebrafish, promoting general understanding of retinal development, morphology, function and associated diseases. Over the recent years, genome and protein ... Read more >>

J. Neurogenet. (Journal of neurogenetics)
[2017, 31(3):70-87]

Cited: 2 times

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Shaping of Signal Transmission at the Photoreceptor Synapse by EAAT2 Glutamate Transporters.

Stephanie Niklaus, Lucia Cadetti, Colette M Vom Berg-Maurer, André Lehnherr, Adriana L Hotz, Ian C Forster, Matthias Gesemann, Stephan C F Neuhauss,

Photoreceptor ribbon synapses tonically release glutamate. To ensure efficient signal transmission and prevent glutamate toxicity, a highly efficient glutamate removal system provided by members of the SLC1 gene family is required. By using a combination of biophysical and in vivo studies, we elucidate the role of excitatory amino acid transporter ... Read more >>

eNeuro (eNeuro)
[2017, 4(3):]

Cited: 3 times

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Olfaction: How Fish Catch a Whiff.

Stephan C F Neuhauss,

An elegant new study shows that multiciliated cells in the noses of aquatic vertebrates generate flow fields that help odor detection and processing. ... Read more >>

Curr. Biol. (Current biology : CB)
[2017, 27(2):R57-R58]

Cited: 0 times

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Important role of endocannabinoid signaling in the development of functional vision and locomotion in zebrafish.

Andrea Martella, Rosa M Sepe, Cristoforo Silvestri, Jingjing Zang, Giulia Fasano, Oliana Carnevali, Paolo De Girolamo, Stephan C F Neuhauss, Paolo Sordino, Vincenzo Di Marzo,

The developmental role of the endocannabinoid system still remains to be fully understood. Here, we report the presence of a complete endocannabinoid system during zebrafish development and show that the genes that code for enzymes that catalyze the anabolism and catabolism (mgll and dagla) of the endocannabinoid, 2-AG (2-arachidonoylglycerol), as ... Read more >>

FASEB J. (FASEB journal : official publication of the Federation of American Societies for Experimental Biology)
[2016, 30(12):4275-4288]

Cited: 5 times

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Linking Genomo- and Pathotype: Exploiting the Zebrafish Embryo Model to Investigate the Divergent Virulence Potential among Cronobacter spp.

Athmanya K Eshwar, Ben D Tall, Jayanthi Gangiredla, Gopal R Gopinath, Isha R Patel, Stephan C F Neuhauss, Roger Stephan, Angelika Lehner,

Bacteria belonging to the genus Cronobacter have been recognized as causative agents of life-threatening systemic infections primarily in premature, low-birth weight and immune-compromised neonates. Apparently not all Cronobacter species are linked to infantile infections and it has been proposed that virulence varies among strains. Whole genome comparisons and in silico ... Read more >>

PLoS ONE (PloS one)
[2016, 11(6):e0158428]

Cited: 4 times

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mglur6b:EGFP Transgenic zebrafish suggest novel functions of metabotropic glutamate signaling in retina and other brain regions.

Stella M K Glasauer, Robert Wäger, Matthias Gesemann, Stephan C F Neuhauss,

Metabotropic glutamate receptors (mGluRs) are mainly known for regulating excitability of neurons. However, mGluR6 at the photoreceptor-ON bipolar cell synapse mediates sign inversion through glutamatergic inhibition. Although this is currently the only confirmed function of mGluR6, other functions have been suggested. Here we present Tg(mglur6b:EGFP)zh1, a new transgenic zebrafish line ... Read more >>

J. Comp. Neurol. (The Journal of comparative neurology)
[2016, 524(12):2363-2378]

Cited: 3 times

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The Authors Reply.

Stephan Segerer, Yuya Sugano, Maja T Lindenmeyer, Ines Auberger, Urs Ziegler, Clemens D Cohen, Stephan C F Neuhauss, Johannes Loffing,

Kidney Int. (Kidney international)
[2016, 89(5):1161-1162]

Cited: 0 times

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The Rho-GTPase binding protein IQGAP2 is required for the glomerular filtration barrier.

Yuya Sugano, Maja T Lindenmeyer, Ines Auberger, Urs Ziegler, Stephan Segerer, Clemens D Cohen, Stephan C F Neuhauss, Johannes Loffing,

Podocyte dysfunction impairs the size selectivity of the glomerular filter, leading to proteinuria, hypoalbuminuria, and edema, clinically defined as nephrotic syndrome. Hereditary forms of nephrotic syndrome are linked to mutations in podocyte-specific genes. To identify genes contributing to podocyte dysfunction in acquired nephrotic syndrome, we studied human glomerular gene expression ... Read more >>

Kidney Int. (Kidney international)
[2015, 88(5):1047-1056]

Cited: 4 times

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Thyroid disruption in zebrafish (Danio rerio) larvae: Different molecular response patterns lead to impaired eye development and visual functions.

Lisa Baumann, Albert Ros, Kristina Rehberger, Stephan C F Neuhauss, Helmut Segner,

The vertebrate thyroid system is important for multiple developmental processes, including eye development. Thus, its environmentally induced disruption may impact important fitness-related parameters like visual capacities and behaviour. The present study investigated the relation between molecular effects of thyroid disruption and morphological and physiological changes of eye development in zebrafish ... Read more >>

Aquat. Toxicol. (Aquatic toxicology (Amsterdam, Netherlands))
[2016, 172:44-55]

Cited: 8 times

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