نتایج جستجو برای: oculodentodigital dysplasia

تعداد نتایج: 28629  

2013
Ying Hu I-Ping Chen Salome de Almeida Valdenize Tiziani Cassio M. Raposo Do Amaral Kalpana Gowrishankar Maria Rita Passos-Bueno Ernst J. Reichenberger

Craniometaphyseal dysplasia (CMD) is a rare sclerosing skeletal disorder with progressive hyperostosis of craniofacial bones. CMD can be inherited in an autosomal dominant (AD) trait or occur after de novo mutations in the pyrophosphate transporter ANKH. Although the autosomal recessive (AR) form of CMD had been mapped to 6q21-22 the mutation has been elusive. In this study, we performed whole-...

Journal: :Cardiovascular research 2008
Janet L Manias Isabelle Plante Xiang-Qun Gong Qing Shao Jared Churko Donglin Bai Dale W Laird

AIMS More than 40 mutations in the GJA1 gene encoding connexin43 (Cx43) have been linked to oculodentodigital dysplasia (ODDD), a pleiotropic, autosomal dominant disorder. We hypothesized that even with a significant reduction in the levels of Cx43 in a mutant mouse model of ODDD (Gja1(Jrt/+)) harbouring a G60S mutation (Cx43(G60S)), cardiomyocyte function may only be moderately compromised giv...

2012
Qing Shao Qin Liu Robert Lorentz Xiang-Qun Gong Donglin Bai Gary S. Shaw Dale W. Laird

Mutations in the gene encoding connexin-43 (Cx43) cause the human development disorder known as oculodentodigital dysplasia (ODDD). In this study, ODDD-linked Cx43 N-terminal mutants formed nonfunctional gap junction-like plaques and exhibited dominant-negative effects on the coupling conductance of coexpressed endogenous Cx43 in reference cell models. Nuclear magnetic resonance (NMR) protein s...

Journal: :Human molecular genetics 2008
Radoslaw Dobrowolski Philipp Sasse Jan W Schrickel Marcus Watkins Jung-Sun Kim Mindaugas Rackauskas Clemens Troatz Alexander Ghanem Klaus Tiemann Joachim Degen Feliksas F Bukauskas Roberto Civitelli Thorsten Lewalter Bernd K Fleischmann Klaus Willecke

Oculodentodigital dysplasia (ODDD) is a dominant negatively inherited disorder with variable but characteristic anomalies of the fingers and toes, eyes, face and teeth, which are caused by mutations in the connexin 43 (Cx43) gene. All mutations analyzed so far have a negative influence on the conductance through gap junctional channels and hemichannels, as well as trafficking of Cx43 protein in...

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