ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

Genome editing of gtpbp3 in zebrafish using CRISPR/Cas9 system. (A) Schematic representation of CRISPR/Cas9 target site at exon 2 of zebrafish gtpbp3 gene as used in this study. White boxes indicate coding region, black boxes indicate untranslated regions of exons and lines between exons indicate introns. The resultant truncated 98 aa non-functional protein caused by frame shifting deletion in gtpbp3 is shown in diagram. (B–D) Genotyping of gtpbp3^del14bp by Sanger sequence, the PAGE and Western blot analyses. (E) Lateral views of gtpbp3^+/+, gtpbp3^+/−, gtpbp3^−/− zebrafish at 5 dpf. (F) The ratios of genotypes/phenotype of offsprings (F2) in clutches from different F1 gtpbp3^+/- heterozygous crosses at 10 dpf (n = 42).

Cardiac defects in zebrafish. (A) Schematic diagram of zebrafish heart development at 1, 2 and 5 dpf. V, ventricle; A, atrium. (B) Tg (cmlc2:egfp) embryos at 2 dpf shows heart-restricted GFP expression in both chambers. (C) Whole-mount in situ hybridization against cmlc2. According to the looping state, measured by the angles between ventricle and atrium, hearts were divided into normal (α<90°), mild (90°<α<180°), and severe (α > 180°). (D) The proportions of each phenotypes in the embryos in mutant and WT zebrafish. (E) Time-lapse images, the ventricular long axis and ventricular short axis were labeled as V_L and V_S, respectively. End diastolic (red line) and systolic (green line) diameters of zebrafish ventricle at 5 dpf were measured using Olympus software CellSense. (F) The proportions of the short axis fractional shortening (FS_S), long axis fractional shortening (FS_L) and fractional area change (FAC) in mutant and WT zebrafish. the calculations were based on eight independent determinations. Graph details and symbols are explained in the legend to Figure 4.

Hypertrophic cardiomyopathy in zebrafish. (A) Hematoxylin and eosin stained (H&E) histological sections of hearts from gtpbp3^−/−, gtpbp3^+/− and gtpbp3^+/+ zebrafish at the age of one year. Scale bars: 10 μm. (B) The relative cross-sectional area of cardiomyocyte in gtpbp3^+/+ (n = 136), gtpbp3^+/- (n = 147), gtpbp3^−/− (n = 116) zebrafish, staining with H&E. (C) The cardiomyocytes from mutant and WT zebrafish were visualized via WGA staining. Scale bars: 5 μm. (D) Relative levels of nppa and nppb gene in hearts from mutant and WT zebrafish. (E) Ventricular heart muscle sections of transmission electron microscopy in mutant and WT zebrafish. Ultrathin sections were visualized with 15,000X magnifications. Altered myofibrils were indicated by white arrows and widened I-band in the sarcomere units were indicated by white arrowhead. Scale bars: 1 μm. (F) Quantification of the I-band length of gtpbp3^+/+ (n = 16), gtpbp3^+/- (n = 38) and gtpbp3^−/− (n = 37) zebrafish. The values for the mutants were expressed as percentages of the average values for the wild type.

Mitochondrial defects in the zebrafish heart. (A, B) Assessment of mitochondrial function in cardiomyocytes by enzyme histochemistry (EHC) staining for COX (A) and SDH (B) in the frozen-sections of ventricles in the gtbpbp3^−/−, gtpbp3^+/− and gtpbp3^+/+ zebrafish at one year old. Scale bars: 40 μm. (C) Mitochondrial networks from cardiomyocytes of transmission electron microscopy. Ultrathin sections were visualized with 30 000× magnifications.

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