Cargando…
Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker
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...
Autores principales: | , |
---|---|
Formato: | Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2010
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2919470/ https://www.ncbi.nlm.nih.gov/pubmed/20637071 http://dx.doi.org/10.1186/1471-213X-10-75 |
_version_ | 1782185189502353408 |
---|---|
author | Geach, Timothy J Zimmerman, Lyle B |
author_facet | Geach, Timothy J Zimmerman, Lyle B |
author_sort | Geach, Timothy J |
collection | PubMed |
description | 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 α-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. |
format | Text |
id | pubmed-2919470 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2010 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-29194702010-08-11 Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker Geach, Timothy J Zimmerman, Lyle B BMC Dev Biol Research Article 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 α-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. BioMed Central 2010-07-16 /pmc/articles/PMC2919470/ /pubmed/20637071 http://dx.doi.org/10.1186/1471-213X-10-75 Text en Copyright ©2010 Geach and Zimmerman; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Research Article Geach, Timothy J Zimmerman, Lyle B Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker |
title | Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker |
title_full | Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker |
title_fullStr | Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker |
title_full_unstemmed | Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker |
title_short | Paralysis and delayed Z-disc formation in the Xenopus tropicalis unc45b mutant dicky ticker |
title_sort | paralysis and delayed z-disc formation in the xenopus tropicalis unc45b mutant dicky ticker |
topic | Research Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2919470/ https://www.ncbi.nlm.nih.gov/pubmed/20637071 http://dx.doi.org/10.1186/1471-213X-10-75 |
work_keys_str_mv | AT geachtimothyj paralysisanddelayedzdiscformationinthexenopustropicalisunc45bmutantdickyticker AT zimmermanlyleb paralysisanddelayedzdiscformationinthexenopustropicalisunc45bmutantdickyticker |