ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2011
PALS1 is essential for retinal pigment epithelium structure and neural retina stratification.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 29 papers.
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Who cites it
29 citing papers in PubMed, 52 citations in OpenAlex.
- Characterization of a spontaneous microphthalmia-like mutant mouse with isolated ocular defects.PloS one · 2026Article
- Development of Retinal Astroglia.Annual review of vision science · 2025Review
- Super-resolution imaging uncovers the nanoscopic segregation of polarity proteins in epithelia.eLife · 2022Article
- Identification and characterization of Crumbs polarity complex proteins in Caenorhabditis elegans.The Journal of biological chemistry · 2022Article
- Scribble basal polarity acquisition in RPE cells and its mislocalization in a pathological AMD-like model.Frontiers in neuroanatomy · 2022Article
- Observational
- Key Role for CRB2 in the Maintenance of Apicobasal Polarity in Retinal Pigment Epithelial Cells.Frontiers in cell and developmental biology · 2021Article
- De novo variants in MPP5 cause global developmental delay and behavioral changes.Human molecular genetics · 2020Article
- Cytoglobin deficiency potentiates Crb1-mediated retinal degeneration in rd8 mice.Developmental biology · 2020Article
- Research Models and Gene Augmentation Therapy forFrontiers in neuroscience · 2020Review
- Review
- Sphingosine Kinase-1 Is Essential for Maintaining External/Outer Limiting Membrane and Associated Adherens Junctions in the Aging Retina.Molecular neurobiology · 2019Article
- Article
- Regulation of spermatid polarity by the actin- and microtubule (MT)-based cytoskeletons.Seminars in cell & developmental biology · 2018Review
- Genetic and Molecular Approaches to Study Neuronal Migration in the Developing Cerebral Cortex.Brain sciences · 2017Review
- Immunohistochemical study of the membrane skeletal protein, membrane protein palmitoylated 6 (MPP6), in the mouse small intestine.Histochemistry and cell biology · 2016Article
- CRB2 completes a fully expressed Crumbs complex in the Retinal Pigment Epithelium.Scientific reports · 2015Article
- The Polarity Protein Pals1 Regulates Radial Sorting of Axons.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2015Article
- Polarity transitions during neurogenesis and germinal zone exit in the developing central nervous system.Frontiers in cellular neuroscience · 2015Review
- Common and distinctive localization patterns of Crumbs polarity complex proteins in the mammalian eye.Gene expression patterns : GEP · 2015Article
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Authors and funding
14 authors at 5 institutions in 4 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
The membrane-associated palmitoylated protein 5 (MPP5 or PALS1) is thought to organize intracellular PALS1-CRB-MUPP1 protein scaffolds in the retina that are involved in maintenance of photoreceptor-Müller glia cell adhesion. In humans, the Crumbs homolog 1 (CRB1) gene is mutated in progressive types of autosomal recessive retinitis pigmentosa and Leber congenital amaurosis. However, there is no clear genotype-phenotype correlation for CRB1 mutations, which suggests that other components of the CRB complex may influence the severity of retinal disease. Therefore, to understand the physiological role of the Crumbs complex proteins, especially PALS1, we generated and analyzed conditional knockdown mice for Pals1. Small irregularly shaped spots were detected throughout the PALS1 deficient retina by confocal scanning laser ophthalmoscopy and spectral domain optical coherence tomography. The electroretinography a- and b-wave was severely attenuated in the aged mutant retinas, suggesting progressive degeneration of photoreceptors. The histological analysis showed abnormal retinal pigment epithelium structure, ectopic photoreceptor nuclei in the subretinal space, an irregular outer limiting membrane, half rosettes of photoreceptors in the outer plexiform layer, and a thinner photoreceptor synaptic layer suggesting improper photoreceptor cell layering during retinal development. The PALS1 deficient retinas showed reduced levels of Crumbs complex proteins adjacent to adherens junctions, upregulation of glial fibrillary acidic protein indicative of gliosis, and persisting programmed cell death after retinal maturation. The phenotype suggests important functions of PALS1 in the retinal pigment epithelium in addition to the neural retina.
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