scholarly journals Proper expression of the Gcn5 histone acetyltransferase is required for neural tube closure in mouse embryos

2008 ◽  
Vol 237 (4) ◽  
pp. 928-940 ◽  
Author(s):  
Wenchu Lin ◽  
Zhijing Zhang ◽  
Geraldine Srajer ◽  
Yi Chun Chen ◽  
Maosheng Huang ◽  
...  
Keyword(s):  
Neural Tube ◽  
Histone Acetyltransferase ◽  
Mouse Embryos ◽  
Neural Tube Closure ◽  
Tube Closure

scholarly journals Biomechanical coupling facilitates spinal neural tube closure in mouse embryos

2017 ◽  
pp. 201700934 ◽  
Author(s):  
Gabriel L. Galea ◽  
Young-June Cho ◽  
Gauden Galea ◽  
Matteo A. Molè ◽  
Ana Rolo ◽  
...  
Keyword(s):  
Neural Tube ◽  
Mouse Embryos ◽  
Neural Tube Closure ◽  
Tube Closure

scholarly journals Valproic acid disrupts the biomechanics of late spinal neural tube closure in mouse embryos

2018 ◽  
Vol 149 ◽  
pp. 20-26 ◽  
Author(s):  
Amy Hughes ◽  
Nicholas D.E. Greene ◽  
Andrew J. Copp ◽  
Gabriel L. Galea
Keyword(s):  
Valproic Acid ◽  
Neural Tube ◽  
Mouse Embryos ◽  
Neural Tube Closure ◽  
Tube Closure

Curvature of the caudal region is responsible for failure of neural tube closure in the curly tail (ct) mouse embryo

Development ◽  
1991 ◽  
Vol 113 (2) ◽  
pp. 671-678 ◽  
Author(s):  
F.A. Brook ◽  
A.S. Shum ◽  
H.W. Van Straaten ◽  
A.J. Copp
Keyword(s):  
Neural Tube ◽  
Mouse Embryo ◽  
Mouse Embryos ◽  
The Body ◽  
Neural Tube Closure ◽  
Posterior Neuropore ◽  
Curly Tail ◽  
Delayed Closure ◽  
Tube Closure

Delayed closure of the posterior neuropore (PNP) occurs to a variable extent in homozygous mutant curly tail (ct) mouse embryos, and results in the development of spinal neural tube defects (NTD) in 60% of embryos. Previous studies have suggested that curvature of the body axis may delay neural tube closure in the cranial region of the mouse embryo. In order to investigate the relationship between curvature and delayed PNP closure, we measured the extent of ventral curvature of the neuropore region in ct/ct embryos with normal or delayed PNP closure. The results show significantly greater curvature in ct/ct embryos with delayed PNP closure in vivo than in their normal littermates. Reopening of the posterior neuropore in non-mutant mouse embryos, to delay neuropore closure experimentally, did not increase ventral curvature, suggesting that increased curvature in ct/ct embryos is not likely to be a secondary effect of delayed PNP closure. Experimental prevention of ventral curvature in ct/ct embryos, brought about by implantation of an eyelash tip longitudinally into the hindgut lumen, ameliorated the delay in PNP closure. We propose, therefore, that increased ventral curvature of the neuropore region of ct/ct embryos imposes a mechanical stress, which opposes neurulation and thus delays closure of the PNP. Increased ventral curvature may arise as a result of a cell proliferation imbalance, which we demonstrated previously in affected ct/ct embryos.

Spatio-temporal curvature pattern of the caudal body axis for non-mutant and curly tail mouse embryos during the period of caudal neural tube closure

1997 ◽  
Vol 195 (3) ◽  
pp. 259-266 ◽  
Author(s):  
M. C. E. Peeters ◽  
Johan W. M. Hekking ◽  
Tryfon Vainas ◽  
J. Drukker ◽  
Henny W. M. van Straaten
Keyword(s):  
Neural Tube ◽  
Mouse Embryos ◽  
Body Axis ◽  
Neural Tube Closure ◽  
Curly Tail ◽  
Spatio Temporal ◽  
Tube Closure

scholarly journals Excess methionine suppresses the methylation cycle and inhibits neural tube closure in mouse embryos

FEBS Letters ◽  
2006 ◽  
Vol 580 (11) ◽  
pp. 2803-2807 ◽  
Author(s):  
Louisa P.E. Dunlevy ◽  
Katie A. Burren ◽  
Lyn S. Chitty ◽  
Andrew J. Copp ◽  
Nicholas D.E. Greene
Keyword(s):  
Neural Tube ◽  
Mouse Embryos ◽  
Neural Tube Closure ◽  
Tube Closure

scholarly journals Methylmercury induced toxicogenomic response in C57 and SWV mouse embryos undergoing neural tube closure

2010 ◽  
Vol 30 (2) ◽  
pp. 284-291 ◽  
Author(s):  
Joshua F. Robinson ◽  
William C. Griffith ◽  
Xiaozhong Yu ◽  
Sungwoo Hong ◽  
Euvin Kim ◽  
...  
Keyword(s):  
Neural Tube ◽  
Mouse Embryos ◽  
Neural Tube Closure ◽  
Tube Closure

scholarly journals FKBP8 variants are risk factors for spina bifida

2020 ◽  
Vol 29 (18) ◽  
pp. 3132-3144
Author(s):  
Tian Tian ◽  
Xuanye Cao ◽  
Sung-Eun Kim ◽  
Ying Linda Lin ◽  
John W Steele ◽  
...  
Keyword(s):  
Risk Factors ◽  
Spina Bifida ◽  
Neural Tube ◽  
Early Stage ◽  
Mouse Embryos ◽  
Neural Tube Closure ◽  
Abnormal Expression ◽  
Functional Variants ◽  
Cellular Apoptosis ◽  
Tube Closure

Abstract Neural tube defects (NTDs) are a group of severe congenital malformations caused by a failure of neural tube closure during early embryonic development. Although extensively investigated, the genetic etiology of NTDs remains poorly understood. FKBP8 is critical for proper mammalian neural tube closure. Fkbp8−/− mouse embryos showed posterior NTDs consistent with a diagnosis of spina bifida (SB). To date, no publication has reported any association between FKBP8 and human NTDs. Using Sanger sequencing on genomic DNA samples from 472 SB and 565 control samples, we identified five rare (MAF ≤ 0.001) deleterious variants in SB patients, while no rare deleterious variant was identified in the controls (P = 0.0191). p.Glu140* affected FKBP8 localization to the mitochondria and created a truncated form of the FKBP8 protein, thus impairing its interaction with BCL2 and ultimately leading to an increase in cellular apoptosis. p.Ser3Leu, p.Lys315Asn and p.Ala292Ser variants decreased FKBP8 protein level. p.Lys315Asn further increased the cellular apoptosis. RNA sequencing on anterior and posterior tissues isolated from Fkbp8−/− and wildtype mice at E9.5 and E10.5 showed that Fkbp8−/− embryos have an abnormal expression profile within tissues harvested at posterior sites, thus leading to a posterior NTD. Moreover, we found that Fkbp8 knockout mouse embryos have abnormal expression of Wnt3a and Nkx2.9 during the early stage of neural tube development, perhaps also contributing to caudal specific NTDs. These findings provide evidence that functional variants of FKBP8 are risk factors for SB, which may involve a novel mechanism by which Fkbp8 mutations specifically cause SB in mice.

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