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| Status |
Public on Jul 30, 2006 |
| Title |
The zebrafish runzel mutant results in a novel muscular dystrophy phenotype due to a titin mutation |
| Organism |
Danio rerio |
| Experiment type |
Expression profiling by array
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| Summary |
The zebrafish runzel (ruz) carries a mutation in the titin gene resulting in muscle degeneration and embryonic lethality. At the onset of the visible phenotype (3-3.5 days post fertilization), RNA expression of the tail and trunk (primarily skeletal muscle) was compared between ruz mutants and wildtype siblings using the Affymetrix Zebrafish Gene Chip. Mutant RNA was matched with RNA of wildtype siblings from the same clutch. 3 separate clutches were used (n=3). RNA was labeled, sheered, and hybridized as in Shephard et al. (Shepard et al., 2005) and the data analyzed as in Choe et al. and Weber et al. (Choe et al., 2005; Weber et al., 2005). Results suggest a novel mechanism of titin muscular dystrophy pathogenesis including upregulation of heat shock proteins.
Keywords: disease state analysis
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| Overall design |
At the onset of the visible phenotype (3-3.5 days post fertilization), RNA expression of the tail and trunk (primarily skeletal muscle) was compared between ruz mutants and wildtype siblings using the Affymetrix Zebrafish Gene Chip. RNA of mutants was matched with RNA of wildtype siblings from the same clutch. 3 separate clutches were used (6 arrays total).
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| Contributor(s) |
Steffen LS, Guyon JR, Vogel ED, Weber GJ, Zhou Y, Zon LI, Kunkel LM |
| Citation missing |
Has this study been published? Please login to update or notify GEO. |
| Submission date |
Mar 30, 2006 |
| Last update date |
Jan 25, 2018 |
| Contact name |
Leta S Steffen |
| E-mail(s) |
leta_steffen@student.hms.harvard.edu
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| Organization name |
Harvard Medical School, Children's Hospital
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| Department |
Program in Genomics
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| Lab |
Louis M Kunkel
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| Street address |
320 Longwood Ave
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| City |
Boston |
| State/province |
MA |
| ZIP/Postal code |
02115 |
| Country |
USA |
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| Platforms (1) |
| GPL1319 |
[Zebrafish] Affymetrix Zebrafish Genome Array |
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| Samples (6)
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| Relations |
| BioProject |
PRJNA94433 |
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