eys; lrp5 Zebrafish Reveals Lrp5 Can Be the Receptor of Retinol in the Visual Cycle

Takita, Shimpei; Seko, Yuko

doi:10.1016/j.isci.2020.101762

Cited by 4 publications

(13 citation statements)

References 42 publications

(43 reference statements)

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“…LRP5 and 6, which play an important role in Wnt signalling during many developmental processes, including neural development (Angers and Moon, 2009; Gray et al, 2013; Jeong et al, 2014), can also bind RAP, albeit with lower affinity than other members of the LRP family (Li et al, 2005). As LRP5 and LRP6 have been reported to be present at the apical surface of other epithelial cell types, they are also potentially present at the apical membrane of neuronal precursors and hence may play a role in RAP uptake at the neuroepithelium (Takita and Seko, 2020; Yamamoto et al, 2017). Further studies are necessary to identify the RAP receptor at the apical membrane of the zebrafish neuroepithelium.…”

Section: Discussionmentioning

confidence: 99%

Assessment of endocytic traffic and Ocrl function in the developing zebrafish neuroepithelium

Williams

Güngördü

Jackson-Crawford

et al. 2022

Preprint

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Endocytosis is a vital process, required during development and for maintenance of tissue homeostasis, that allows cells to internalize a wide range of molecules from their environment as well maintain their plasma membrane composition. The ability to visualise endocytosis in vivo requires suitable assays to monitor the process. Here, we describe imaging-based assays to visualize endocytosis in the neuroepithelium of living zebrafish embryos. These assays rely on injection of fluorescent tracers into the brain ventricles followed by live imaging and can be used to study fluid-phase or receptor-mediated endocytosis, for which we use receptor-associated protein (RAP) as a ligand for LDL receptor-related protein (LRP) receptors expressed at the neuroepithelium. Using dual colour imaging combined with transient or stable expression of endocytic markers, it is possible to track the progression of endocytosed tracers and to monitor trafficking dynamics. Using these assays, we reveal a role for the Lowe syndrome protein Ocrl in endocytic trafficking within the neuroepithelium. We also find that the RAP binding receptor Lrp2 appears to only partially contribute to neuroepithelial RAP endocytosis. Altogether, our results provide a basis to track endocytosis within the neuroepithelium in vivo, and support a role for Ocrl in this process.

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Section: Discussionmentioning

confidence: 99%

Assessment of endocytic traffic and Ocrl function in the developing zebrafish neuroepithelium

Williams

Güngördü

Jackson-Crawford

et al. 2022

Preprint

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show abstract

“…The basal process of neuroepithelial cells contacts the basal lamina adjacent to the brain wall, whilst the apical process forms junctions with neighbouring cells to integrate into an epithelial sheet that collectively makes contact with the cerebrospinal fluid (CSF) of the brain ventricles (Fig. 1A,B) (Fame et al, 2020;Gotz and Huttner, 2005;Norden, 2017;Taverna et al, 2014). Within these cells, the arrangement of endocytic compartments mirrors that seen in mammalian epithelial cell types.…”

Section: Zebrafish Neuroepithelial Tissue Endocytoses Fluid-phase Tra...mentioning

confidence: 98%

“…By 24 hpf, the overall architecture of the fore, mid and hindbrain regions of the brain is well defined with tissue in these regions lining the fore, mid and hindbrain ventricles. At this stage of development, a significant proportion of brain tissue consists of neuroepithelial cells, a key progenitor cell type from which new-born neurons and further neuroepithelial cells are derived (Gotz and Huttner, 2005;Taverna et Journal of Cell Science • Accepted manuscript al., 2014). Viewed from both coronal (Fig.…”

Section: Zebrafish Neuroepithelial Tissue Endocytoses Fluid-phase Tra...mentioning

confidence: 99%

“…The identity of this receptor is currently unclear. RAP can bind to other members of the LRP receptor family and several of these (LRP1, ApoER2, VLDLR) are expressed in radial glia cells (Auderset et al, 2016a;Bres et al, 2020;Luque et al, 2003), a related neuroepithelial cell type that also have an apical membrane exposed to the ventricle (Gotz and Huttner, 2005;Taverna et al, 2014). However, these receptors appear to be localised to the basolateral membrane of the radial glia cells (Auderset et al, 2016a;Auderset et al, 2016b;Boggild et al, 2018;Luque et al, 2003), and are unlikely to be the receptor for injected RAP.…”

Section: Journal Of Cell Science • Accepted Manuscriptmentioning

confidence: 99%

“…Neuroepithelial cells and related cell types such as radial glia are polarised progenitor cells that make up the bulk of brain tissue at the early stages of brain development (Gotz and Huttner, 2005;Taverna et al, 2014). These cell types respond to multiple developmental cues that guide both tissue patterning and the post-mitotic fate neuronal progenitors commit to in order to establish a functional organ (Gotz and Huttner, 2005;Taverna et al, 2014). Multiple studies have demonstrated the importance of post-Golgi endocytic trafficking in the regulation of neuronal progenitor cell function and fate.…”

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Assessment of endocytic traffic and Ocrl function in the developing zebrafish neuroepithelium

Williams

Güngördü

Jackson-Crawford

et al. 2022

Journal of Cell Science

View full text Add to dashboard Cite

Endocytosis allows cells to internalize a wide range of molecules from their environment and to maintain their plasma membrane composition. It is vital during development and for maintenance of tissue homeostasis. The ability to visualise endocytosis in vivo requires suitable assays to monitor the process. Here, we describe imaging-based assays to visualize endocytosis in the neuroepithelium of living zebrafish embryos. Injection of fluorescent tracers into the brain ventricles followed by live imaging was used to study fluid-phase or receptor-mediated endocytosis, for which we use receptor-associated protein (RAP) as a ligand for LDL receptor-related protein (LRP) receptors. Using dual-colour imaging combined with expression of endocytic markers, it is possible to track the progression of endocytosed tracers and to monitor trafficking dynamics. Using these assays, we reveal a role for the Lowe syndrome protein Ocrl in endocytic trafficking within the neuroepithelium. We also find that the RAP binding receptor Lrp2 appears to only partially contribute to neuroepithelial RAP endocytosis. Altogether, our results provide a basis to track endocytosis within the neuroepithelium in vivo, and support a role for Ocrl in this process.

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Rhodopsin mislocalization drives ciliary dysregulation in a novel autosomal dominant retinitis pigmentosa knock‐in mouse model

Takita,

Jahan,

S. Imanishi

et al. 2024

The FASEB Journal

Self Cite

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Rhodopsin mislocalization encompasses various blind conditions. Rhodopsin mislocalization is the primary factor leading to rod photoreceptor dysfunction and degeneration in autosomal dominant retinitis pigmentosa (adRP) caused by class I mutations. In this study, we report a new knock‐in mouse model that harbors a class I Q344X mutation in the endogenous rhodopsin gene, which causes rod photoreceptor degeneration in an autosomal dominant pattern. In RhoQ344X/+ mice, mRNA transcripts from the wild‐type (Rho) and RhoQ344X mutant rhodopsin alleles are expressed at equal levels. However, the amount of RHOQ344X mutant protein is 2.7 times lower than that of wild‐type rhodopsin, a finding consistent with the rapid degradation of the mutant protein. Immunofluorescence microscopy indicates that RHOQ344X is mislocalized to the inner segment and outer nuclear layers of rod photoreceptors in both RhoQ344X/+ and RhoQ344X/Q344X mice, confirming the essential role of the C‐terminal VxPx motif in promoting OS delivery of rhodopsin. The mislocalization of RHOQ344X is associated with the concurrent mislocalization of wild‐type rhodopsin in RhoQ344X/+ mice. To understand the global changes in proteostasis, we conducted quantitative proteomics analysis and found attenuated expression of rod‐specific OS membrane proteins accompanying reduced expression of ciliopathy causative gene products, including constituents of BBSome and axonemal dynein subunit. Those studies unveil a novel negative feedback regulation involving ciliopathy‐associated proteins. In this process, a defect in the trafficking signal leads to a reduced quantity of the trafficking apparatus, culminating in a widespread reduction in the transport of ciliary proteins.

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eys; lrp5 Zebrafish Reveals Lrp5 Can Be the Receptor of Retinol in the Visual Cycle

Cited by 4 publications

References 42 publications

Assessment of endocytic traffic and Ocrl function in the developing zebrafish neuroepithelium

Assessment of endocytic traffic and Ocrl function in the developing zebrafish neuroepithelium

Assessment of endocytic traffic and Ocrl function in the developing zebrafish neuroepithelium

Rhodopsin mislocalization drives ciliary dysregulation in a novel autosomal dominant retinitis pigmentosa knock‐in mouse model

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