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http://hdl.handle.net/1956/2087
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| Tittel: | Characterization of Nuclear Respiratory Factor 1 using transgenic zebrafish |
| Forfattere: | Kyrkjebø, Vibeke |
| Dato: | 1-Dec-2006 |
| Forlag: | The University of Bergen
Sars International Centre for Marine Molecular Biology |
| Sammendrag: | Nuclear respiratory factor 1 (NRF1) is a transcription factor essential for the
embryonic development of vertebrates. The zebrafish mutant nrf is characterized by
apoptotic photoreceptor death during embryogenesis followed by late larval lethality.
In zebrafish nrf1 is initially expressed throughout the CNS at a high level at days 1
and 2 and later fades to levels undetectable by in situ hybridization. This study shows
that the initial phase of expression is crucial for photoreceptor maintenance, but not
for their initial development, as homozygous mutant cells can form normal
photoreceptors in a wild type background. By utilizing reporter expression of a
marker insertion near the wild type allele of nrf1 combined with transgenic rescue, I
show that a single heat shock induced pulse of nrf1 expression at any time between 24
and 54 hours post fertilization is sufficient to rescue the mutant phenotype and delay
photoreceptor degeneration until larval stages. Moreover, no ectopic defects are
detected after ubiquitous expression of the gene, suggesting that Nrf1 serves no
detectable instructive role during embryogenesis. These results suggest that Nrf1
plays a permissive role in zebrafish photoreceptor maintenance and is crucial for the
formation and survival of the outer nuclear layer, but is not strictly necessary for the
initial development of individual photoreceptors. Gene expression comparison
analysis identifies several up and downregulated genes in the nrf mutant, suggesting
that the intraflagellar transport machinery of the photoreceptor connecting cilium
might be defect. |
| URI: | http://hdl.handle.net/1956/2087 |
| ISBN: | 82-308-0257-2 (print version) |
| Vises i samlingene: | Department of Molecular Biology
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