Abstract:
:Joint and skeletal development is highly regulated by extracellular matrix (ECM) proteoglycans, of which chondroitin sulfate proteoglycans (CSPGs) are a major class. Despite the requirement of joint CSPGs for skeletal flexibility and structure, relatively little is understood regarding their role in establishing joint positioning or in modulating signaling and cell behavior during joint formation. Chondroitin sulfate synthase 1 (Chsy1) is one of a family of enzymes that catalyze the extension of chondroitin and dermatan sulfate glycosaminoglycans. Recently, human syndromic brachydactylies have been described to have loss-of-function mutations at the CHSY1 locus. In concordance with these observations, we demonstrate that mice lacking Chsy1, though viable, display chondrodysplasia and decreased bone density. Notably, Chsy1(-/-) mice show a profound limb patterning defect in which orthogonally shifted ectopic joints form in the distal digits. Associated with the digit-patterning defect is a shift in cell orientation and an imbalance in chondroitin sulfation. Our results place Chsy1 as an essential regulator of joint patterning and provide a mouse model of human brachydactylies caused by mutations in CHSY1.
journal_name
Dev Bioljournal_title
Developmental biologyauthors
Wilson DG,Phamluong K,Lin WY,Barck K,Carano RA,Diehl L,Peterson AS,Martin F,Solloway MJdoi
10.1016/j.ydbio.2012.01.005subject
Has Abstractpub_date
2012-03-15 00:00:00pages
413-25issue
2eissn
0012-1606issn
1095-564Xpii
S0012-1606(12)00021-8journal_volume
363pub_type
杂志文章abstract::Monoclonal antibodies have been generated to determine the fate of sperm-specific surface components of the sea urchin, Arbacia punctulata, subsequent to gamete fusion. Monoclonal antibody-7 (MAB-7) reacted with two polypeptide bands having apparent molecular masses of 33 and 35 kDa derived from the sperm plasma membr...
journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/s0012-1606(89)80036-3
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abstract::Heparan sulfate proteoglycans (HSPGs) regulate a number of major developmental processes, but their roles in synovial joint formation remain unknown. Here we created conditional mouse embryo mutants lacking Ext1 in developing joints by mating Ext1(f/f) and Gdf5-Cre mice. Ext1 encodes a subunit of the Ext1/Ext2 Golgi-a...
journal_title:Developmental biology
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doi:10.1016/j.ydbio.2014.08.016
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journal_title:Developmental biology
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journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/j.ydbio.2005.07.033
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journal_title:Developmental biology
pub_type: 杂志文章
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journal_title:Developmental biology
pub_type: 杂志文章
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journal_title:Developmental biology
pub_type: 杂志文章
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pub_type: 杂志文章
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doi:10.1016/j.ydbio.2007.05.037
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journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/j.ydbio.2007.08.018
更新日期:2007-11-15 00:00:00
abstract::This paper reports a preliminary in silico analysis of the sea urchin kinome. The predicted protein kinases in the sea urchin genome were identified, annotated and classified, according to both function and kinase domain taxonomy. The results show that the sea urchin kinome, consisting of 353 protein kinases, is close...
journal_title:Developmental biology
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journal_title:Developmental biology
pub_type: 杂志文章
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journal_title:Developmental biology
pub_type: 杂志文章
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