The BH3-Only SNARE BNip1 Mediates Photoreceptor Apoptosis in Response to Vesicular Fusion Defects

Nishiwaki, Yuko; Yoshizawa, Asuka; Kojima, Yutaka; Oguri, Eri; Nakamura, Shohei; Suzuki, Shingo; Yuasa-Kawada, Junichi; Kinoshita-Kawada, Mariko; Mochizuki, Toshiaki; Masai, Ichiro

doi:10.1016/j.devcel.2013.04.015

Cited by 26 publications

(28 citation statements)

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“…Indeed, our recent studies demonstrate a marked fragmentation of the Golgi and decreased expression of Golgi-resident proteins such as GBF1 in ␣SNAP-depleted epithelial cells (34 -36). Similarly, a loss of function mutation of the brain-enriched ␤SNAP isoform was shown to trigger Golgi dispersion in zebrafish photoreceptors leading to photoreceptor degeneration (84). Importantly, we observed that Golgi fragmentation by either pharmacologic agents or GBF1 depletion phenocopied all effects of ␣SNAP knockdown on ECM adhesion including impaired processing and mislocalization of ␤1 integrin, decreased expression and phosphorylation of paxillin, and cell detachment (Fig.…”

Section: Discussionsupporting

confidence: 57%

N-Ethylmaleimide-sensitive Factor Attachment Protein α (αSNAP) Regulates Matrix Adhesion and Integrin Processing in Human Epithelial Cells

Naydenov

Feygin

Wang

et al. 2014

Journal of Biological Chemistry

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Background: Vesicle trafficking plays important roles in regulating cell adhesion and motility. Results: Depletion of the membrane fusion protein, N-ethylmaleimide-sensitive factor attachment protein ␣ (␣SNAP), induced cell detachment, whereas overexpression of this vesicle regulator increased cell-matrix adhesion. Conclusion: ␣SNAP promotes cell-matrix adhesion by controlling integrin trafficking and assembly of focal adhesions. Significance: The ␣SNAP level may serve as a molecular rheostat controlling matrix adhesion and cell motility under normal conditions and in diseases.

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Section: Discussionsupporting

confidence: 57%

N-Ethylmaleimide-sensitive Factor Attachment Protein α (αSNAP) Regulates Matrix Adhesion and Integrin Processing in Human Epithelial Cells

Naydenov

Feygin

Wang

et al. 2014

Journal of Biological Chemistry

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“…Protein purification and SDS-PAGE were conducted as previously described41. Anti-Pde6c (Abcam; ab5660), zGc325, and histone H3 (Abcam; ab1791) antibodies were used at dilutions of 1:2000, 1:1000 and 1:50000, respectively.…”

Section: Methodsmentioning

confidence: 99%

Aipl1 is required for cone photoreceptor function and survival through the stability of Pde6c and Gc3 in zebrafish

Iribarne

Nishiwaki

Nakamura

et al. 2017

Sci Rep

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Genetic mutations in aryl hydrocarbon receptor interacting protein-like 1 (AIPL1) cause photoreceptor degeneration associated with Leber congenital amaurosis 4 (LCA4) in human patients. Here we report retinal phenotypes of a zebrafish aipl1 mutant, gold rush (gosh). In zebrafish, there are two aipl1 genes, aipl1a and aipl1b, which are expressed mainly in rods and cones, respectively. The gosh mutant gene encodes cone-specific aipl1, aipl1b. Cone photoreceptors undergo progressive degeneration in the gosh mutant, indicating that aipl1b is required for cone survival. Furthermore, the cone-specific subunit of cGMP phosphodiesterase 6 (Pde6c) is markedly decreased in the gosh mutant, and the gosh mutation genetically interacts with zebrafish pde6c mutation eclipse (els). These data suggest that Aipl1 is required for Pde6c stability and function. In addition to Pde6c, we found that zebrafish cone-specific guanylate cyclase, zGc3, is also decreased in the gosh and els mutants. Furthermore, zGc3 knockdown embryos showed a marked reduction in Pde6c. These observations illustrate the interdependence of cGMP metabolism regulators between Aipl1, Pde6c, and Gc3 in photoreceptors.

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“…β-SNAP −/− presents photoreceptors degeneration, in which photoreceptors undergo apoptosis in a BH3-only, protein BNip1-dependent mechanism due to failure to disassemble the SNARE. β-SNAP mutant was the first zebrafish mutant to link photoreceptor degeneration to vesicular transport defects [37].…”

Section: Discerning Specific Signaling Pathways Between Cone or Rod Pmentioning

confidence: 99%

Zebrafish Photoreceptor Degeneration and Regeneration Research to Understand Hereditary Human Blindness

Iribarne¹

2020

Visual Impairment and Blindness - What We Know and What We Have to Know

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Humans with mutations in photoreceptor-related genes develop forms of retinal degeneration, such as retinitis pigmentosa, cone dystrophy, or Leber congenital amaurosis. Similarly, numerous photoreceptor mutant animal models present phenotypes that resemble retinal degeneration. Zebrafish retina manifests anatomical organization and development remarkably conserved in humans, making these fish a good model to study photoreceptor development and disease. Zebrafish are ideal for forward genetic screens to isolate mutants with visual defects. More recently, CRISPR/Cas system-mediated genome editing has enabled establishment of specific zebrafish photoreceptor mutants. Here, I review zebrafish models of inherited retinal diseases, focusing on rod versus cone photoreceptor mutants. Because zebrafish possess robust regeneration capacity to replace the lost photoreceptors, here I review the current understanding of molecular mechanisms underlying this response.Visual Impairment and Blindness -What We Know and What We Have to Know 2 2.1 Photoreceptors in the zebrafish retina Zebrafish neural retina, like those of other vertebrates, comprises three nuclear layers, separated by two synaptic layers (Figure 1). The outer nuclear layer (ONL) comprises rod and cone photoreceptors, the light-sensing neurons. The inner nuclear layer (INL) consists of bipolar, horizontal, and amacrine neurons, the second-order neurons. The ganglion cell layer is formed by ganglion cells, axons of which exit the retina, forming the optic nerve, which connects with the tectum. These neurons interconnect by synapses in the plexiform layers. The neural retina is located adjacent to the retinal pigment epithelium (RPE), which supports general homeostasis of photoreceptors, such as recycling 11-cis retinal for visual pigment regeneration [2,3].Photoreceptors are polarized neurons with characteristic morphology. They display very specialized cell regions, including outer segments (OSs), connecting cilia, cell bodies, and terminal synapses. OS structure is important for phototransduction. The cell bodies possess the machinery to support all cell functions, and their synaptic termini transduce signals to bipolar neurons. OSs are formed by hundreds of cell membrane discs stacked horizontally and associated with a high concentration of proteins for phototransduction. These proteins are synthetized in cell bodies, and then are transported to the OS through connecting cilia.Photoreceptors are sensory neurons that produce electrical responses when stimulated by light. In the OS, photons are captured by photopigment molecules to initiate phototransduction cascades [4]. Phototransduction is a complex signaling process that results in closing of voltage-gated ion channels, producing a change in membrane potential. Then, this electrical signal is amplified by other cell types in the inner retina and conducted to the brain. Although this signaling pathway is common to both rods and cones, signaling proteins are mostly encoded by distinct sets of rod-and cone-specifi...

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The BH3-Only SNARE BNip1 Mediates Photoreceptor Apoptosis in Response to Vesicular Fusion Defects

Cited by 26 publications

References 48 publications

N-Ethylmaleimide-sensitive Factor Attachment Protein α (αSNAP) Regulates Matrix Adhesion and Integrin Processing in Human Epithelial Cells

N-Ethylmaleimide-sensitive Factor Attachment Protein α (αSNAP) Regulates Matrix Adhesion and Integrin Processing in Human Epithelial Cells

Aipl1 is required for cone photoreceptor function and survival through the stability of Pde6c and Gc3 in zebrafish

Zebrafish Photoreceptor Degeneration and Regeneration Research to Understand Hereditary Human Blindness

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