Paralysis and delayed Z-disc formation in the Xenopus tropicalisunc45b mutant dicky tickerReportar como inadecuado




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BMC Developmental Biology

, 10:75

First Online: 16 July 2010Received: 09 April 2010Accepted: 16 July 2010

Abstract

BackgroundThe protein components of mature skeletal muscle have largely been characterized, but the mechanics and sequence of their assembly during normal development remain an active field of study. Chaperone proteins specific to sarcomeric myosins have been shown to be necessary in zebrafish and invertebrates for proper muscle assembly and function.

ResultsThe Xenopus tropicalis mutation dicky ticker results in disrupted skeletal muscle myofibrillogenesis, paralysis, and lack of heartbeat, and maps to a missense mutation in the muscle-specific chaperone unc45b. Unc45b is known to be required for folding the head domains of myosin heavy chains, and mutant embryos fail to incorporate muscle myosin into sarcomeres. Mutants also show delayed polymerization of α-actinin-rich Z-bodies into the Z-disks that flank the myosin-containing A-band.

ConclusionsThe dicky ticker phenotype confirms that a requirement for myosin-specific chaperones is conserved in tetrapod sarcomerogenesis, and also suggests a novel role for myosin chaperone function in Z-body maturation.

AbbreviationsAMOantisense morpholino oligonucleotide

ArgArginine

CysCysteine

MyHCmyosin heavy chain

SNPSingle Nucleotide Polymorphism

SSLPSimple Sequence Length Polymorphism

WISHwhole mount in situ hybridization.

Electronic supplementary materialThe online version of this article doi:10.1186-1471-213X-10-75 contains supplementary material, which is available to authorized users.

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Autor: Timothy J Geach - Lyle B Zimmerman

Fuente: https://link.springer.com/article/10.1186/1471-213X-10-75







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