scholarly journals Generation ofShox2-Creallele for tissue specific manipulation of genes in the developing heart, palate, and limb

genesis ◽  
2013 ◽  
Vol 51 (7) ◽  
pp. 515-522 ◽  
Author(s):  
Cheng Sun ◽  
Tao Zhang ◽  
Chao Liu ◽  
Shuping Gu ◽  
YiPing Chen
Open Biology ◽  
2014 ◽  
Vol 4 (4) ◽  
pp. 140014 ◽  
Author(s):  
Aina Pi-Roig ◽  
Enrique Martin-Blanco ◽  
Carolina Minguillon

The transcription factor Tbx5 is expressed in the developing heart, eyes and anterior appendages. Mutations in human TBX5 cause Holt–Oram syndrome, a condition characterized by heart and upper limb malformations. Tbx5 -knockout mouse embryos have severely impaired forelimb and heart morphogenesis from the earliest stages of their development. However, zebrafish embryos with compromised tbx5 function show a complete absence of pectoral fins, while heart development is disturbed at significantly later developmental stages and eye development remains to be thoroughly analysed. We identified a novel tbx5 gene in zebrafish— tbx5b— that is co-expressed with its paralogue, tbx5a , in the developing eye and heart and hypothesized that functional redundancy could be occurring in these organs in embryos with impaired tbx5a function. We have now investigated the consequences of tbx5a and/or tbx5b downregulation in zebrafish to reveal that tbx5 genes have essential roles in the establishment of cardiac laterality, dorsoventral retina axis organization and pectoral fin development. Our data show that distinct relationships between tbx5 paralogues are required in a tissue-specific manner to ensure the proper morphogenesis of the three organs in which they are expressed. Furthermore, we uncover a novel role for tbx5 genes in the establishment of correct heart asymmetry in zebrafish embryos.


Author(s):  
Adam R. Prickett ◽  
Bertille Montibus ◽  
Nikolaos Barkas ◽  
Samuele M. Amante ◽  
Maurício M. Franco ◽  
...  

Dopa decarboxylase (DDC) synthesizes serotonin in the developing mouse heart where it is encoded by Ddc_exon1a, a tissue-specific paternally expressed imprinted gene. Ddc_exon1a shares an imprinting control region (ICR) with the imprinted, maternally expressed (outside of the central nervous system) Grb10 gene on mouse chromosome 11, but little else is known about the tissue-specific imprinted expression of Ddc_exon1a. Fluorescent immunostaining localizes DDC to the developing myocardium in the pre-natal mouse heart, in a region susceptible to abnormal development and implicated in congenital heart defects in human. Ddc_exon1a and Grb10 are not co-expressed in heart nor in brain where Grb10 is also paternally expressed, despite sharing an ICR, indicating they are mechanistically linked by their shared ICR but not by Grb10 gene expression. Evidence from a Ddc_exon1a gene knockout mouse model suggests that it mediates the growth of the developing myocardium and a thinning of the myocardium is observed in a small number of mutant mice examined, with changes in gene expression detected by microarray analysis. Comparative studies in the human developing heart reveal a paternal expression bias with polymorphic imprinting patterns between individual human hearts at DDC_EXON1a, a finding consistent with other imprinted genes in human.


1997 ◽  
Vol 99 (2) ◽  
pp. 342-347 ◽  
Author(s):  
Silvina A. Felitti ◽  
Raquel L. Chan ◽  
Gabriela Gago ◽  
Estela M. Valle ◽  
Daniel H. Gonzalez
Keyword(s):  

Sign in / Sign up

Export Citation Format

Share Document