PUBLICATION
Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker
- Authors
- Geach, T.J., and Zimmerman, L.B.
- ID
- ZDB-PUB-100726-1
- Date
- 2010
- Source
- BMC Developmental Biology 10: 75 (Journal)
- Registered Authors
- Keywords
- none
- MeSH Terms
-
- Amino Acid Sequence
- Amino Acid Substitution
- Animals
- Genes, Lethal
- Molecular Chaperones/genetics*
- Molecular Chaperones/metabolism*
- Molecular Sequence Data
- Muscle Development
- Mutation, Missense*
- Myofibrils/metabolism
- Myosins/metabolism
- Sarcomeres/metabolism
- Sequence Alignment
- Xenopus/embryology*
- Xenopus/metabolism
- Zebrafish
- PubMed
- 20637071 Full text @ BMC Dev. Biol.
Citation
Geach, T.J., and Zimmerman, L.B. (2010) Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker. BMC Developmental Biology. 10:75.
Abstract
BACKGROUND: 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. RESULTS: 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. CONCLUSIONS: 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.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping