Project description:RNASET2-deficient leukoencephalophathy is a severe leukodystrophy affecting children with psychomotor impairements in their forst year of life. We generated the first zebrafish model for a human leukodystrophy by targetting the ortholog rnaset2 gene using mutagenesis. This zebrafish mutant recapitulated the human clinical manisfestations and developed white matter defects detectable by MRI. Additionally, this zebrafish mutant identified this disease as a lysosomal storage disorders, with RNA accumulating in neurons. To understand how accumulation of RNA in neurons trigger white matter lesions, we undertook an unbiased approach and perfored microarray analysis. We identified differentially expressed genes in mutants and the immune system as a key pathway disregulated in the zebrafish rnaset2 mutant.
Project description:To uncover the genes regulated by pharmacological activation of the Glucocorticoid Receptor, we performed microarray-based expression profiling of whole zebrafish embryos at 24 and 72 hours post fertilization (hpf) after 3-hour treatment with 25 µM of Beclomethasone, a potent glucocorticoid previously tested in zebrafish or with 0.1 % DMSO as control.
Project description:We performed a zebrafish forward genetic screen by Tol2 mediated gene-trap approach and uncovered one mutant stac (The number of the transgenic line: B55) that showed severe cell-death distributed in the various brain and trunk in the homozygote embryos. Analysis of stac homozygous embryos demonstrates typical apoptosis. So it is necessary to analyze whether the apoptosis and cell cycle regulated signaling transductions are changed in the mutant, in order to provide valuable clues to some other species. And the up-regulated and down-regulated genes in the mutant compared to the WT were examined by zebrafish cDNA microarray.
Project description:Transcriptional profiling of hdac1 mutant zebrafish in comparison to their sibling embryos. Embryos resulting from a cross between heterozygous hdac1 mutant zebrafish (hi1618/+) where cultured together then mutants separated from the siblings one the basis of phenotype and RNA extracted from the two groups at 27hpf was compared in a two-colour hybridisation.
Project description:The myelomonocyte fraction and whole kidney marrow were sorted from wild-type or mutant zebrafish kidney at 60 days post-fertilization (dpf).