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XB-ART-41772
BMC Dev Biol. July 16, 2010; 10 75.

Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker.



Abstract
The 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. The 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 alpha-actinin-rich Z-bodies into the Z-disks that flank the myosin-containing A-band. The 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.

PubMed ID: 20637071
PMC ID: PMC2919470
Article link: BMC Dev Biol.
Grant support: U117560482 Medical Research Council , MC_U117560482 Medical Research Council , MC_U117560482 Medical Research Council , U117560482 Medical Research Council , MRC_MC_U117560482 Medical Research Council , U117560482 Medical Research Council , MC_U117560482 Medical Research Council , U117560482 Medical Research Council

Genes referenced: actb actn1 actn2 myh6 sst ttn unc45b

Antibodies referenced: Actn2 Ab2 Fast Skeletal Muscle Ab1 Myh6 Ab1 Ttn Ab2
Morpholinos referenced: unc45b MO1 unc45b MO2

References:
Abu-Daya, 2009, Pubmed, Xenbase[+]


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