Expression and localization of the polarity protein CRB2 in adult mouse brain: a comparison with the CRB1 rd8 mutant mouse model.
Jorge F. Dolón,Antonio E. Paniagua,Antonio E. Paniagua,Vicente Valle,Alicia Segurado,Rosario Arévalo,Almudena Velasco,Concepción Lillo +7 more
TL;DR: The presence of CRB2 in adult mice brain is characterized, where it is particularly enriched in cortex, hippocampus, hypothalamus and cerebellum, and double immunofluorescence analysis confirmed thatCRB2 is a neuron-specific protein, present in both soma and projections where colocalizes with certain populations of exocytic and endocytic vesicles and with other members of the Crumbs complex.
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Abstract: Acquisition of cell polarization is essential for the performance of crucial functions, like a successful secretion and appropriate cell signaling in many tissues, and it depends on the correct functioning of polarity proteins, including the Crumbs complex. The CRB proteins, CRB1, CRB2 and CRB3, identified in mammals, are expressed in epithelial-derived tissues like brain, kidney and retina. CRB2 has a ubiquitous expression and has been detected in embryonic brain tissue; but currently there is no data regarding its localization in the adult brain. In our study, we characterized the presence of CRB2 in adult mice brain, where it is particularly enriched in cortex, hippocampus, hypothalamus and cerebellum. Double immunofluorescence analysis confirmed that CRB2 is a neuron-specific protein, present in both soma and projections where colocalizes with certain populations of exocytic and endocytic vesicles and with other members of the Crumbs complex. Finally, in the cortex of CRB1rd8 mutant mice that contain a mutation in the Crb1 gene generating a truncated CRB1 protein, there is an abnormal increase in the expression levels of the CRB2 protein which suggests a possible compensatory mechanism for the malfunction of CRB1 in this mutant background.
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Citations
Development and maintenance of vision's first synapse.
TL;DR: In this paper, the authors review outer retina neuron synapse architecture, neural migration modes, and the cellular and molecular pathways that play key roles in the development and maintenance of these connections.
31
Crumbs2 Is an Essential Slit Diaphragm Protein of the Renal Filtration Barrier.
Annika Möller-Kerutt,Juan E. Rodriguez-Gatica,Karin Wacker,Rohan Bhatia,Jan Peter Siebrasse,Nanda Boon,Veerle Van Marck,Peter Boor,Ulrich Kubitscheck,Jan Wijnholds,Hermann Pavenstädt,Thomas Weide +11 more
TL;DR: In this article, the function of Crumbs2 at the renal filtration barrier was studied in mice lacking Crb2 exclusively in podocytes, and high-resolution expansion microscopy was used to investigate the distribution of crumbs2 distribution in murine glomeruli.
22
AAV-CRB2 protects against vision loss in an inducible CRB1 retinitis pigmentosa mouse model
TL;DR: In this paper, a Muller cell-specific Crb1KOCrb2LowMGC retinitis pigmentosa mouse model was presented, which showed a substantial decrease in inner/outer photoreceptor segment length and optokinetic head-tracking response.
18
Diverse functions and pathogenetic role of Crumbs in retinopathy
Xinru Zhou,Lingling Zhao,Chenguang Wang,Wei Sun,Bo Jia,Dan Li,Jing Fu +6 more
TL;DR: Crumbs protein family plays a crucial role in maintaining the integrity of embryonic epithelia and retinal development. Mutations in human CRB genes lead to retinal dystrophies. Animal and human-derived models provide insights into the pathophysiology of CRB-associated retinopathies.
Targeted deletion of Crb1/Crb2 in the optic vesicle models key features of leber congenital amaurosis 8.
TL;DR: The results suggest that human LCA8 can be modeled in the mouse by simultaneously ablating Crb1/2 from the beginning of eye development and indicate that L CA8 is caused by malfunction of retinal progenitor cells during early ocular development rather than by defective photoreceptor-Muller glial interaction, a mechanism proposed for RP12.
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