scholarly journals Erratum: Corrigendum: Endocytic recycling protein EHD1 regulates primary cilia morphogenesis and SHH signaling during neural tube development

2017 ◽  
Vol 7 (1) ◽  
Author(s):  
Sohinee Bhattacharyya ◽  
Mark A. Rainey ◽  
Priyanka Arya ◽  
Bhopal C. Mohapatra ◽  
Insha Mushtaq ◽  
...  
Keyword(s):  
Neural Tube ◽  
Primary Cilia ◽  
Endocytic Recycling ◽  
Shh Signaling ◽  
Neural Tube Development

scholarly journals Endocytic recycling protein EHD1 regulates primary cilia morphogenesis and SHH signaling during neural tube development

2016 ◽  
Vol 6 (1) ◽  
Author(s):  
Sohinee Bhattacharyya ◽  
Mark A Rainey ◽  
Priyanka Arya ◽  
Bhopal C. Mohapatra ◽  
Insha Mushtaq ◽  
...  
Keyword(s):  
Neural Tube ◽  
Hedgehog Signaling ◽  
Primary Cilia ◽  
Gene Knockout ◽  
Neural Tube Closure ◽  
Endocytic Recycling ◽  
Shh Signaling ◽  
Neuronal Markers ◽  
Surface Receptors ◽  
Direct Binding

Abstract Members of the four-member C-terminal EPS15-Homology Domain-containing (EHD) protein family play crucial roles in endocytic recycling of cell surface receptors from endosomes to the plasma membrane. In this study, we show that Ehd1 gene knockout in mice on a predominantly B6 background is embryonic lethal. Ehd1-null embryos die at mid-gestation with a failure to complete key developmental processes including neural tube closure, axial turning and patterning of the neural tube. We found that Ehd1-null embryos display short and stubby cilia on the developing neuroepithelium at embryonic day 9.5 (E9.5). Loss of EHD1 also deregulates the ciliary SHH signaling with Ehd1-null embryos displaying features indicative of increased SHH signaling, including a significant downregulation in the formation of the GLI3 repressor and increase in the ventral neuronal markers specified by SHH. Using Ehd1-null MEFS we found that EHD1 protein co-localizes with the SHH receptor Smoothened in the primary cilia upon ligand stimulation. Under the same conditions, EHD1 was shown to co-traffic with Smoothened into the developing primary cilia and we identify EHD1 as a direct binding partner of Smoothened. Overall, our studies identify the endocytic recycling regulator EHD1 as a novel regulator of the primary cilium-associated trafficking of Smoothened and Hedgehog signaling.

scholarly journals The role of primary cilia in the pathophysiology of neural tube defects

2012 ◽  
Vol 33 (4) ◽  
pp. E2 ◽  
Author(s):  
Timothy W. Vogel ◽  
Calvin S. Carter ◽  
Kingsley Abode-Iyamah ◽  
Qihong Zhang ◽  
Shenandoah Robinson
Keyword(s):  
Neural Tube ◽  
Neural Tube Defects ◽  
Neural Development ◽  
Planar Cell Polarity ◽  
Primary Cilia ◽  
Integration Site ◽  
Genetics Research ◽  
Neural Tube Development ◽  
Insight Into

Neural tube defects (NTDs) are a set of disorders that occur from perturbation of normal neural development. They occur in open or closed forms anywhere along the craniospinal axis and often result from a complex interaction between environmental and genetic factors. One burgeoning area of genetics research is the effect of cilia signaling on the developing neural tube and how the disruption of primary cilia leads to the development of NTDs. Recent progress has implicated the hedgehog (Hh), wingless-type integration site family (Wnt), and planar cell polarity (PCP) pathways in primary cilia as involved in normal neural tube patterning. A set of disorders involving cilia function, known as ciliopathies, offers insight into abnormal neural development. In this article, the authors discuss the common ciliopathies, such as Meckel-Gruber and Joubert syndromes, that are associated with NTDs, and review cilia-related signaling cascades responsible for mammalian neural tube development. Understanding the contribution of cilia in the formation of NTDs may provide greater insight into this common set of pediatric neurological disorders.

scholarly journals The Mechanism of Fos and Leptin Receptor in Neural Tube Development of Offspring Under The Regulation of Energy Balance

2021 ◽  
Author(s):  
Ting Li ◽  
Quan Huo ◽  
Zhi Guo Lu ◽  
Xin Ran Xing ◽  
Lu Ding ◽  
...  
Keyword(s):  
Energy Balance ◽  
Animal Models ◽  
Neural Tube ◽  
High Fat Diet ◽  
Leptin Receptor ◽  
High Expression ◽  
Control Group ◽  
High Fat ◽  
Neural Tube Development ◽  
Energy States

Abstract Background The occurrence of neural tube defects is a complex process in which genes, internal and external environment and other factors jointly influence and occur interactively. In this experiment, animal models of different energy balance states are constructed. To explore the mechanism of fos and leptin-leptin receptor during neural tube development of offspring under different energy states and its effect on neural tube development of offspringMethods Using gene identification technology to obtain Mex3c+/- negative energy balance mice and high-fat diet to obtain positive energy balance mice, and obtain E10.5d, E12.5d, E14.5d embryos. We will verify the expression of fos, leptin, LEPR, nestin, PAX3, and H3K27me3 proteins in the neural tube of the offspring through relevant experimental methods.Results We have successfully constructed animal models, Control group (18.82g±1.54g), Mex3c group (18.84g±1.08g), HFD group (22.61g±1.10g). Neural tube HE staining showen that compared with the Control group, the neuronal maturity of the Mex3c group and the HFD group was reduced. Immunohistochemical staining showed that both fos and leptin were expressed on the nucleus, and LEPR was expressed on the cell membrane. Western blot experiments showed that compared with the Control group, the Mex3c group and the HFD group had low expression of fos protein (P＜0.01), the Mex3c group had high expression of LEPR protein (P＜0.01) and the HFD group had high expression of LEPR protein (P＜0.01). Immunostaining experiments showed that nestin was expressed in nerve fibers, and PAX3 and H3K27me3 were both expressed in the nucleus. Western blooting experiment showed that compared with the Control group, the Mex3c group had high expression of nestin protein (P＜0.01), PAX3 protein (P＜0.01), H3K27me3 (P＜0.01), and the HFD group had high expression of nestin protein (P＜0.01). ) And PAX3 protein (P<0.01), H3K27me3 (P<0.01).ConclusionsMex3c regulates leptin and LEPR by enhancing the expression of fos mRNA to participate in the neural tube development process of offspring. The neural tube nestin, PAX3, and H3K27me3 of the offspring of Mex3c+/- mice and high-fat diet mice continue to be highly expressed. Mex3c+/- mice express low leptin, and high-fat diet mice highly express leptin; preliminary reveals the regulation of different energy states Leptin-LEPR is involved in the process of neurodevelopment. Mex3c mutant mice and mice on a high-fat diet lead to decreased neurodevelopmental maturity.

The effect of magnetic resonance imaging on neural tube development in an early chicken embryo model

2018 ◽  
Vol 34 (5) ◽  
pp. 933-938 ◽  
Author(s):  
Emrah Kantarcioglu ◽  
Gokmen Kahilogullari ◽  
Murat Zaimoglu ◽  
Esin Ozlem Atmis ◽  
Elif Peker ◽  
...  
Keyword(s):  
Magnetic Resonance Imaging ◽  
Magnetic Resonance ◽  
Neural Tube ◽  
Chicken Embryo ◽  
Resonance Imaging ◽  
Neural Tube Development
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