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Objective: Several mouse models of cardiac neural crest cell (NCC)-associated conotruncal heart defects exist, but the specific cellular and molecular defects within cardiac NCC morphogenesis remain largely unknown. Our objective was to investigate the underlying mechanisms resulting in outflow tract defects and why insufficient cardiac NCC reach the heart of the Splotch (Sp 2H ) mouse...
Objective: The splotch (Sp 2H mutation disrupts the Pax3 gene and is lethal in homozygotes. The aim of the present study was to investigate the cause of lethality. Methods and results: Using the splotch (Sp 2H ) mouse mutant, we demonstrated that approximately 60% of Sp 2H homozygotes die in utero at 13.5-14.5 days of gestation. All these embryos have cardiac...
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