Obscurin is required for the lateral alignment of striated myofibrils in zebrafish

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dc.contributor.author Raeker, Maide Ö. en_US
dc.contributor.author Su, Fengyun en_US
dc.contributor.author Geisler, Sarah B. en_US
dc.contributor.author Borisov, Andrei B. en_US
dc.contributor.author Kontrogianni-Konstantopoulos, Aikaterini en_US
dc.contributor.author Lyons, Susan E. en_US
dc.contributor.author Russell, Mark W. en_US
dc.date.accessioned 2007-09-18T19:21:48Z
dc.date.available 2007-09-18T19:21:48Z
dc.date.issued 2006-08 en_US
dc.identifier.citation Raeker, Maide Ö.; Su, Fengyun; Geisler, Sarah B.; Borisov, Andrei B.; Kontrogianni-Konstantopoulos, Aikaterini; Lyons, Susan E.; Russell, Mark W. (2006). "Obscurin is required for the lateral alignment of striated myofibrils in zebrafish." Developmental Dynamics 235(8): 2018-2029. <http://hdl.handle.net/2027.42/55797> en_US
dc.identifier.issn 1058-8388 en_US
dc.identifier.issn 1097-0177 en_US
dc.identifier.uri http://hdl.handle.net/2027.42/55797
dc.identifier.uri http://www.ncbi.nlm.nih.gov/sites/entrez?cmd=retrieve&db=pubmed&list_uids=16779859&dopt=citation en_US
dc.description.abstract Obscurin/obscurin-MLCK is a giant sarcomere-associated protein with multiple isoforms whose interactions with titin and small ankyrin-1 suggest that it has an important role in myofibril assembly, structural support, and the sarcomeric alignment of the sarcoplasmic reticulum. In this study, we characterized the zebrafish orthologue of obscurin and examined its role in striated myofibril assembly. Zebrafish obscurin was expressed in the somites and central nervous system by 24 hours post-fertilization (hpf) and in the heart by 48 hpf. Depletion of obscurin using two independent morpholino antisense oligonucleotides resulted in diminished numbers and marked disarray of skeletal myofibrils, impaired lateral alignment of adjacent myofibrils, disorganization of the sarcoplasmic reticulum, somite segmentation defects, and abnormalities of cardiac structure and function. This is the first demonstration that obscurin is required for vertebrate cardiac and skeletal muscle development. The diminished capacity to generate and organize new myofibrils in response to obscurin depletion suggests that it may have a vital role in the causation of or adaptation to cardiac and skeletal myopathies. Developmental Dynamics 235:2018–2029, 2006. © 2006 Wiley-Liss, Inc. en_US
dc.format.extent 1135270 bytes
dc.format.extent 3118 bytes
dc.format.mimetype application/pdf
dc.format.mimetype text/plain
dc.publisher Wiley-Liss, Inc. en_US
dc.subject.other Life and Medical Sciences en_US
dc.subject.other Cell & Developmental Biology en_US
dc.title Obscurin is required for the lateral alignment of striated myofibrils in zebrafish en_US
dc.type Article en_US
dc.rights.robots IndexNoFollow en_US
dc.subject.hlbsecondlevel Pediatrics en_US
dc.subject.hlbtoplevel Health Sciences en_US
dc.description.peerreviewed Peer Reviewed en_US
dc.contributor.affiliationum Department of Pediatrics and Communicable Diseases, University of Michigan, Ann Arbor, Michigan en_US
dc.contributor.affiliationum Department of Internal Medicine, University of Michigan, Ann Arbor, Michigan en_US
dc.contributor.affiliationum Department of Pediatrics and Communicable Diseases, University of Michigan, Ann Arbor, Michigan en_US
dc.contributor.affiliationum Department of Pediatrics and Communicable Diseases, University of Michigan, Ann Arbor, Michigan en_US
dc.contributor.affiliationum Department of Internal Medicine, University of Michigan, Ann Arbor, Michigan en_US
dc.contributor.affiliationum Department of Pediatrics and Communicable Diseases, University of Michigan, Ann Arbor, Michigan ; Associate Professor of Pediatrics and Communicable Diseases, Division of Pediatric Cardiology, University of Michigan, L1242 Women's Hospital/Box 0204, 1500 E. Medical Center Dr., Ann Arbor, MI 48109 en_US
dc.contributor.affiliationother Department of Physiology, University of Maryland School of Medicine, Baltimore, Maryland en_US
dc.identifier.pmid 16779859 en_US
dc.description.bitstreamurl http://deepblue.lib.umich.edu/bitstream/2027.42/55797/1/20812_ftp.pdf en_US
dc.identifier.doi http://dx.doi.org/10.1002/dvdy.20812 en_US
dc.identifier.source Developmental Dynamics en_US
dc.owningcollname Interdisciplinary and Peer-Reviewed
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