scholarly journals Noninvasive harmonics optical microscopy for long-term observation of embryonic nervous system development in vivo

2006 ◽  
Vol 11 (5) ◽  
pp. 054022 ◽  
Author(s):  
Szu-Yu Chen ◽  
Cho-Shuen Hsieh ◽  
Shi-Wei Chu ◽  
Cheng-Yung Lin ◽  
Ching-Yi Ko ◽  
...  
Keyword(s):  
Nervous System ◽  
Optical Microscopy ◽  
System Development ◽  
Nervous System Development ◽  
Embryonic Nervous System ◽  
Long Term Observation

scholarly journals Drosophila Stathmin: A Microtubule-destabilizing Factor Involved in Nervous System Formation

2002 ◽  
Vol 13 (2) ◽  
pp. 698-710 ◽  
Author(s):  
Sylvie Ozon ◽  
Antoine Guichet ◽  
Olivier Gavet ◽  
Siegfried Roth ◽  
André Sobel
Keyword(s):  
Nervous System ◽  
System Development ◽  
Nervous System Development ◽  
Microtubule Assembly ◽  
Nervous Systems ◽  
Germ Cell Migration ◽  
Numerous Cell ◽  
First Time

Stathmin is a ubiquitous regulatory phosphoprotein, the generic element of a family of neural phosphoproteins in vertebrates that possess the capacity to bind tubulin and interfere with microtubule dynamics. Although stathmin and the other proteins of the family have been associated with numerous cell regulations, their biological roles remain elusive, as in particular inactivation of the stathmin gene in the mouse resulted in no clear deleterious phenotype. We identified stathmin phosphoproteins inDrosophila, encoded by a unique gene sharing the intron/exon structure of the vertebrate stathmin andstathmin family genes. They interfere with microtubule assembly in vitro, and in vivo when expressed in HeLa cells. Drosophila stathmin expression is regulated during embryogenesis: it is high in the migrating germ cells and in the central and peripheral nervous systems, a pattern resembling that of mammalian stathmin. Furthermore, RNA interference inactivation ofDrosophila stathmin expression resulted in germ cell migration arrest at stage 14. It also induced important anomalies in nervous system development, such as loss of commissures and longitudinal connectives in the ventral cord, or abnormal chordotonal neuron organization. In conclusion, a single Drosophilagene encodes phosphoproteins homologous to the entire vertebrate stathmin family. We demonstrate for the first time their direct involvement in major biological processes such as development of the reproductive and nervous systems.

scholarly journals RNA interference screen to identify genes required for Drosophila embryonic nervous system development

2007 ◽  
Vol 104 (13) ◽  
pp. 5626-5631 ◽  
Author(s):  
K. Koizumi ◽  
H. Higashida ◽  
S. Yoo ◽  
M. S. Islam ◽  
A. I. Ivanov ◽  
...  
Keyword(s):  
Nervous System ◽  
Rna Interference ◽  
System Development ◽  
Nervous System Development ◽  
Embryonic Nervous System

Integrin alpha 6 expression is required for early nervous system development in Xenopus laevis

Development ◽  
1996 ◽  
Vol 122 (8) ◽  
pp. 2539-2554 ◽  
Author(s):  
T.E. Lallier ◽  
C.A. Whittaker ◽  
D.W. DeSimone
Keyword(s):  
Nervous System ◽  
Early Development ◽  
System Development ◽  
Cranial Nerves ◽  
Nervous System Development ◽  
Neural Plate ◽  
Pronephric Duct ◽  
Axial Defects ◽  
Integrin Alpha 6

The integrin alpha 6 subunit pairs with both the beta 1 and beta 4 subunits to form a subfamily of laminin receptors. Here we report the cDNA cloning and primary sequence for the Xenopus homologue of the mammalian integrin alpha 6 subunit. We present data demonstrating the spatial and temporal expression of alpha 6 mRNA and protein during early development. Initially, alpha 6 transcripts are expressed in the dorsal ectoderm and future neural plate at the end of gastrulation. Later in development, alpha 6 mRNAs are expressed in a variety of neural derivatives, including the developing sensory placodes (otic and olfactory) and commissural neurons within the neural tube. Integrin alpha 6 is also expressed in the elongating pronephric duct as well as a subset of the rhombencephalic neural crest, which will form the Schwann cells lining several cranial nerves (VII, VIII and X). In vivo expression of an alpha 6 antisense transcript in the animal hemisphere leads to a reduction in alpha 6 protein expression, a loss of adhesion to laminin, and severe defects in normal development. In 35% of cases, reduced levels of alpha 6 expression result in embryos that complete gastrulation normally but arrest at neurulation prior to the formation of the neural plate. In an additional 22% of cases, embryos develop with severe axial defects, including complete loss of head or tail structures. In contrast, overexpression of the alpha 6 subunit by injection of full-length mRNA has no apparent effect on embryonic development. Co-injection of antisense and sense plasmid constructs results in a partial rescue of the antisense-generated phenotypes. These data indicate that the integrin alpha 6 subunit is critical for the early development of the nervous system in amphibians.

scholarly journals Extracellular Matrix-Associated Protein Sc1 Is Not Essential for Mouse Development

2000 ◽  
Vol 20 (2) ◽  
pp. 656-660 ◽  
Author(s):  
Peter J. McKinnon ◽  
Susan K. McLaughlin ◽  
Manuela Kapsetaki ◽  
Robert F. Margolskee
Keyword(s):  
Extracellular Matrix ◽  
Nervous System ◽  
Gene Targeting ◽  
System Development ◽  
Nervous System Development ◽  
Mouse Development ◽  
Term Survival ◽  
Insight Into ◽  
Developing Nervous System

ABSTRACT Sc1 is an extracellular matrix-associated protein whose function is unknown. During early embryonic development, Sc1 is widely expressed, and from embryonic day 12 (E12), Sc1 is expressed primarily in the developing nervous system. This switch in Sc1 expression at E12 suggests an importance for nervous-system development. To gain insight into Sc1 function, we used gene targeting to inactivate mouse Sc1. The Sc1-null mice showed no obvious deficits in any organs. These mice were born at the expected ratios, were fertile, and had no obvious histological abnormalities, and their long-term survival did not differ from littermate controls. Therefore, the function of Sc1 during development is not critical or, in its absence, is subserved by another protein.

scholarly journals Delayed Development of Nervous System in Mice Homozygous for Disrupted Microtubule-associated Protein 1B (MAP1B) Gene

1997 ◽  
Vol 137 (7) ◽  
pp. 1615-1626 ◽  
Author(s):  
Yosuke Takei ◽  
Satoru Kondo ◽  
Akihiro Harada ◽  
Satomi Inomata ◽  
Tetsuo Noda ◽  
...  
Keyword(s):  
Nervous System ◽  
Gene Targeting ◽  
Time Course ◽  
System Development ◽  
Nervous System Development ◽  
Dominant Negative ◽  
Gene Encoding ◽  
Neuronal Morphogenesis ◽  
Associated Proteins

Microtubule-associated protein 1B (MAP1B), one of the microtubule-associated proteins (MAPs), is a major component of the neuronal cytoskeleton. It is expressed at high levels in immature neurons during growth of their axons, which indicates that it plays a crucial role in neuronal morphogenesis and neurite extension. To better define the role of MAP1B in vivo, we have used gene targeting to disrupt the murine MAP1B gene. Heterozygotes of our MAP1B disruption exhibit no overt abnormalities in their development and behavior, while homozygotes showed a slightly decreased brain weight and delayed nervous system development. Our data indicate that while MAP1B is not essential for survival, it is essential for normal time course development of the murine nervous system. These conclusions are very different from those of a previous MAP1B gene–targeting study (Edelmann, W., M. Zervas, P. Costello, L. Roback, I. Fischer, A. Hammarback, N. Cowan, P. Davis, B. Wainer, and R. Kucherlapati. 1996. Proc. Natl. Acad. Sci. USA. 93: 1270–1275). In this previous effort, homozygotes died before reaching 8-d embryos, while heterozygotes showed severely abnormal phenotypes in their nervous systems. Because the gene targeting event in these mice produced a gene encoding a 571–amino acid truncated product of MAP1B, it seems likely that the phenotypes seen arise from the truncated MAP1B product acting in a dominant-negative fashion, rather than a loss of MAP1B function.

scholarly journals Size matters: Large copy number losses in Hirschsprung disease patients reveal genes involved in enteric nervous system development

PLoS Genetics ◽  
2021 ◽  
Vol 17 (8) ◽  
pp. e1009698
Author(s):  
Laura E. Kuil ◽  
Katherine C. MacKenzie ◽  
Clara S. Tang ◽  
Jonathan D. Windster ◽  
Thuy Linh Le ◽  
...  
Keyword(s):  
Nervous System ◽  
Enteric Nervous System ◽  
Copy Number ◽  
System Development ◽  
Hirschsprung Disease ◽  
Nervous System Development ◽  
Gene Enrichment ◽  
Coding Variants ◽  
Enteric Nervous System Development

Hirschsprung disease (HSCR) is a complex genetic disease characterized by absence of ganglia in the intestine. HSCR etiology can be explained by a unique combination of genetic alterations: rare coding variants, predisposing haplotypes and Copy Number Variation (CNV). Approximately 18% of patients have additional anatomical malformations or neurological symptoms (HSCR-AAM). Pinpointing the responsible culprits within a CNV is challenging as often many genes are affected. Therefore, we selected candidate genes based on gene enrichment strategies using mouse enteric nervous system transcriptomes and constraint metrics. Next, we used a zebrafish model to investigate whether loss of these genes affects enteric neuron development in vivo. This study included three groups of patients, two groups without coding variants in disease associated genes: HSCR-AAM and HSCR patients without associated anomalies (HSCR-isolated). The third group consisted of all HSCR patients in which a confirmed pathogenic rare coding variant was identified. We compared these patient groups to unaffected controls. Predisposing haplotypes were determined, confirming that every HSCR subgroup had increased contributions of predisposing haplotypes, but their contribution was highest in isolated HSCR patients without RET coding variants. CNV profiling proved that specifically HSCR-AAM patients had larger Copy Number (CN) losses. Gene enrichment strategies using mouse enteric nervous system transcriptomes and constraint metrics were used to determine plausible candidate genes located within CN losses. Validation in zebrafish using CRISPR/Cas9 targeting confirmed the contribution of UFD1L, TBX2, SLC8A1, and MAPK8 to ENS development. In addition, we revealed epistasis between reduced Ret and Gnl1 expression and between reduced Ret and Tubb5 expression in vivo. Rare large CN losses—often de novo—contribute to HSCR in HSCR-AAM patients. We proved the involvement of six genes in enteric nervous system development and Hirschsprung disease.

Genetic analysis of vein function in the Drosophila embryonic nervous system

Genome ◽  
2000 ◽  
Vol 43 (3) ◽  
pp. 564-573 ◽  
Author(s):  
Bradley R Lanoue ◽  
Michael D Gordon ◽  
Robin Battye ◽  
J Roger Jacobs
Keyword(s):  
Nervous System ◽  
Egf Receptor ◽  
System Development ◽  
Cell Lineage ◽  
Growth Factor Receptor ◽  
Neuronal Cell ◽  
Cell Number ◽  
Nervous System Development ◽  
Egfr Signaling ◽  
Embryonic Nervous System

The Drosophila epidermal growth factor receptor (EGFR) may be activated by two ligands expressed in the embryonic nervous system, Spitz and Vein. Previous studies have established Spitz as an essential activator of EGFR signaling in nervous system development. Here, we report the pattern of expression of vein mRNA in the nervous system and characterize the contribution of vein to cell lineage and axonogenesis. The number of midline glia (MG) precursors is reduced in vein mutants before the onset of embryonic apoptosis. In contrast to spitz, mis-expression of vein does not suppress apoptosis in the MG. These data indicate that early midline EGFR signaling, requiring vein and spitz, establishes MG precursor number, whereas later EGFR signals, requiring spitz, suppress apoptosis in the MG. vein mutants show early irregularities during axon tract establishment, which resolve later to variable defasciculation and thinner intersegmental axon tracts. vein and spitz phenotypes act additively in the regulation of MG cell number, but show synergism in a midline neuronal cell number phenotype and in axon tract architecture. vein appears to act downstream of spitz to briefly amplify local EGFR activation.Key words: Drosophila, vein, midline, axonogenesis, EGF receptor, lineage, neuregulin, spitz, CNS.

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