genetic analysis of hedgehog signaling in ventral body wall development and the onset of omphalocele formation遗传分析刺猬信号在腹侧体壁开发和脐突出形成的开始.pdfVIP

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2017-09-01 发布于上海
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genetic analysis of hedgehog signaling in ventral body wall development and the onset of omphalocele formation遗传分析刺猬信号在腹侧体壁开发和脐突出形成的开始.pdf

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genetic analysis of hedgehog signaling in ventral body wall development and the onset of omphalocele formation遗传分析刺猬信号在腹侧体壁开发和脐突出形成的开始

Genetic Analysis of Hedgehog Signaling in Ventral Body Wall Development and the Onset of Omphalocele Formation 1 1¤a 1¤b 2 3 Daisuke Matsumaru , Ryuma Haraguchi , Shinichi Miyagawa , Jun Motoyama , Naomi Nakagata , 4 1 Frits Meijlink , Gen Yamada * 1 Global COE Cell Fate Regulation Research and Education Unit, Department of Organ Formation, Institute of Molecular Embryology and Genetics (IMEG), Kumamoto University, Kumamoto, Japan, 2 Department of Medical Life Systems, Doshisha University, Kyoto, Japan, 3 Center for Animal Resources and Development (CARD), Kumamoto University, Kumamoto, Japan, 4 Hubrecht Institute, KNAW and University Medical Center, Utrecht, The Netherlands Abstract Background: An omphalocele is one of the major ventral body wall malformations and is characterized by abnormally herniated viscera from the body trunk. It has been frequently found to be associated with other structural malformations, such as genitourinary malformations and digit abnormalities. In spite of its clinical importance, the etiology of omphalocele formation is still controversial. Hedgehog (Hh) signaling is one of the essential growth factor signaling pathways involved in the formation of the limbs and urogenital system. However, the relationship between Hh signaling and ventral body wall formation remains unclear. Methodology/Principal Findings: To gain insight into the roles of Hh signaling in ventral body wall formation and its malformation, we analyzed phenotypes of mouse mutants of Sonic hedgehog (Shh), GLI-Kruppel family member 3 (Gli3) and Aristaless-like homeobox 4 (Alx4). Introduction of additional Alx4Lst muta