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10.1038/ncomms6727 http://scihub22266oqcxt.onion/10.1038/ncomms6727 C4267722!4267722!25473808     free     free     free Deprecated: Implicit conversion from float 227.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534 Deprecated: Implicit conversion from float 227.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534 Deprecated: Implicit conversion from float 227.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534 Deprecated: Implicit conversion from float 227.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534 Deprecated: Implicit conversion from float 227.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534 Deprecated: Implicit conversion from float 227.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534       Nat+Commun 2014 ; 5 (ä): 5727 Nephropedia Template TP {{tp|p=25473808|t=2014. Cryo-electron tomography reveals ciliary defects underlying human RSPH1 primary ciliary dyskinesia |pdf=|usr=}}{{25473808}} gab.com Text Cryo-electron tomography reveals ciliary defects underlying human RSPH1 primary ciliary dyskinesia JO: Nat Commun http://www.kidney.de/pget.php?&tw=1&pmid=25473808 #FOAvip Cilia play essential roles in normal human development and health; cilia dysfunction results in diseases such as primary ciliary dyskinesia (PCD). Despite their importance, the native structure of human cilia is unknown, and structural defects in the cilia of patients are often undetectable or remain elusive because of heterogeneity. Here, we develop an approach that enables visualization of human (patient) cilia at high-resolution using cryo-electron tomography of samples obtained non-invasively by nasal-scrape biopsy. We present the native 3D structures of normal and PCD-causing RSPH1-mutant human respiratory cilia in unprecedented detail; this allows comparisons of cilia structure across evolutionarily distant species and reveals the previously unknown primary defect and the heterogeneous secondary defects in RSPH1-mutant cilia. Our data provide evidence for structural and functional heterogeneity in radial spokes, suggest a mechanism for the milder RSPH1-PCD-phenotype, and demonstrate that cryo-electron tomography can be applied to human disease by directly imaging patient samples. Twit Text FOAVip Cryo-electron tomography reveals ciliary defects underlying human RSPH1 primary ciliary dyskinesia ????Nat Commun http://www.kidney.de/pget.php?&tw=1&pmid=25473808 #FOAvip Twit Text # Free Paper on # # # # # LINK http://www.kidney.de/pget.php?&tw=1&pmid=25473808 #FOAvip English Wikipedia <ref>{{Cite pmid|25473808}}</ref> Cryo-electron tomography reveals ciliary defects underlying human RSPH1 primary ciliary dyskinesia #MMPMID25473808 Lin J; Yin W; Smith MC; Song K; Leigh MW; Zariwala MA; Knowles MR; Ostrowski LE; Nicastro D Nat Commun 2014[]; 5 (ä): 5727 PMID25473808show ga Cilia play essential roles in normal human development and health; cilia dysfunction results in diseases such as primary ciliary dyskinesia (PCD). Despite their importance, the native structure of human cilia is unknown, and structural defects in the cilia of patients are often undetectable or remain elusive because of heterogeneity. Here, we develop an approach that enables visualization of human (patient) cilia at high-resolution using cryo-electron tomography of samples obtained non-invasively by nasal-scrape biopsy. We present the native 3D structures of normal and PCD-causing RSPH1-mutant human respiratory cilia in unprecedented detail; this allows comparisons of cilia structure across evolutionarily distant species and reveals the previously unknown primary defect and the heterogeneous secondary defects in RSPH1-mutant cilia. Our data provide evidence for structural and functional heterogeneity in radial spokes, suggest a mechanism for the milder RSPH1-PCD-phenotype, and demonstrate that cryo-electron tomography can be applied to human disease by directly imaging patient samples. äCryo-electron tomography reveals ciliary defects underlying human RSPH(epmc).pdf DeepDyve Pubget Overpricing