Fig. 1
mne presents a myeloid phenotype at 48?h.p.f. and maps to zbtb11. (a) Brightfield: enlarged fourth ventricle (arrow-head), small dark eye and opacity due to CNS degeneration. Fluorescence: fewer mpx:EGFP-positive cells in mne compared to WT (enumerated in b). (b) Neutrophil deficiency, reflected by the abundance of mpx:EGFP-positive cells, is exacerbated by increasing temperature in the temperature-sensitive mutant mne. Percentage of neutrophils compared to WT is stated in the columns (data from one experiment (from left to right: n=11, 12, 19, 12, 10, 11) representative of four biologically independent replicates, two-tailed t-test; ****P?0.0001). (c) Decreased expression of multiple myeloid genes in mne. Whole-mount in situ hybridization of mne and WT siblings with neutrophil (mpx, lyz), myelomonocytic (lcp1, npsn1), and erythroid (hbbe3) markers at 48?h.p.f. (d) Summary of marsanne genome scan data defining a region narrowed by positional cloning to a 50?kb critical interval, which contained a single gene, zbtb11. Sequencing of mne zbtb11 identifies a single T>A transversion in exon 2 resulting in a Cys>Ser substitution at amino acid 116 (C116S); scale bars, 200??m (a,c). |
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Stage: | Long-pec |
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Stage: | Long-pec |