δ-sarcoglycan is necessary for early heart and muscle development in zebrafish

被引:32
作者
Cheng, Lu
Guo, Xiao-fang
Yang, Xue-yan
Chong, Mei
Cheng, Jing
Li, Ge
Gui, Yong-hao
Lu, Da-ru [1 ]
机构
[1] Fudan Univ, Sch Life Sci, Inst Genet, State Key Lab Genet Engn, Shanghai 200433, Peoples R China
[2] Fudan Univ, Childrens Hosp, Shanghai 200433, Peoples R China
关键词
sarcoglycan; heart; muscle; development; zebrafish;
D O I
10.1016/j.bbrc.2006.03.234
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
delta-sarcoglycan, one member of the sarcoglycan complex, is a very conservative muscle-specific protein exclusively expressed in the skeletal and cardiac muscles of vertebrates. Mutations in sarcoglycans are known to be involved in limb-girdle muscular dystrophy (LGMD) and dilated cardiomyopathy (DCM) in humans. To address the role of delta-sarcoglycan gene in zebrafish development, we have studied expression pattern of delta-sarcoglyean in zebrafish embryos and examined the role of delta-sarcoglyean in zebrafish embryonic development by morpholino. Strong expression of delta-sarcoglycan was observed in various muscles including those of the segment, heart, eye, jaw, pectoral fin, branchial arches, and swim bladder in zebrafish embryo. delta-sarcoglycan was also expressed in midbrain and retina. Knockdown of delta-sarcoglyean resulted in severe abnormality in both the cardiac and skeletal muscles. Some severe ones displayed serious morphological abnormality such as hypoplastic head, linear heart, very weak heartbeats, and runtish trunk, all dead within 5 dpf. Whole-mount in situ hybridization analysis showed that adaxial cells and muscle pioneers were affected in delta-sarcoglycan knockdown embryos. In addition, absence of delta-sarcoglycan protein severely delayed the cardiac development and influenced the differentiation of cardiac muscle, and the cardiac left-right asymmetry was dramatically changed in morpholino-treated embryos. These data together suggest that delta-sarcoglycan plays an important role in early heart and muscle development. (c) 2006 Elsevier Inc. All rights reserved.
引用
收藏
页码:1290 / 1299
页数:10
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