ZFIN ID: ZDB-FIG-220404-10
Masek et al., 2022 - Loss of the Bardet-Biedl protein Bbs1 alters photoreceptor outer segment protein and lipid composition. Nature communications   13:1282 Full text @ Nat. Commun.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Antibody:
Fish:
Anatomical Term:
Stage: Days 7-13
PHENOTYPE:
Fish:
Observed In:
Stage: Days 7-13

Fig. 3 Morphological abnormalities and progressive retinal degeneration in mz<italic>bbs1</italic><sup>−/−</sup> mutants starting at 7 dpf.

a, c Representative images of semi-thin plastic sections stained with Richardson solution in wildtype (top) and mzbbs1−/− (bottom) retina at 7 dpf (a) and 10 dpf (c) showing slight morphological abnormalities at 7 dpf and more severe changes at 10 dpf with shorter and misshapen OSs. b, d DiO-staining highlighting OSs reveals progressive morphological alterations of the OSs between 7 dpf (b) and 10 dpf (d). e TEM reveals bulky OSs (*asterisk) with abnormal membrane disc stacking in mutant retina at 7 dpf. Disc membranes in mutants (middle & right image) are less compact in places or stacked vertically compared to controls (red arrowheads). Membrane-filled swirls are frequently observed in the RPE (yellow arrows) at that stage (middle & right image are two images of mzbbs1−/− retinae showing various alterations seen in the mutants). f At 8dpf, UV-Opsin (green) localizes normally to the OS in mzbbs1−/− mutants and controls, despite abnormal ultrastructure and shortened OS revealed by the membrane dye BODIPY (magenta). g Top panel shows bar plots of the maximum b-wave amplitude by electroretinography at 10 dpf. Reduced b-wave intensity at various light intensities demonstrates severely decreased response to light. Unpaired two-tailed multiple T-test; Sig: ns = FDR(q value) = 0.089, **** = FDR(q value) < 0.0001; Sample size (n = 13 WT, n = 8 Mut larvae), Error bars show standard deviation around the mean. The bottom panel shows the average response curve after a 100 ms light flash at log-2 intensity (Light On/Off line below the ERG curve) for mutant (grey) compared to control (black). Scale Bars: (a,c) 12 µm, (b, d, f) 50 µm, (e) 5 µm. Abbreviations: OS outer segment, ERG electroretinography, INL inner nuclear layer, M mitochondria, N nucleus, ONL outer nuclear layer, OPL outer plexiform layer, P pigment, TEM transmission electron microscopy.

Gene Expression Details No data available
Antibody Labeling Details
Antibody Assay Fish Conditions Stage Anatomy
Ab1-opn1sw1 IHC AB standard conditions Days 7-13 retinal outer plexiform layer
IHC bbs1ka742/ka742 (AB) standard conditions Days 7-13 retinal outer plexiform layer
IHC bbs1ka742/ka742 (AB) standard conditions Days 7-13 retinal outer plexiform layer
Phenotype Details
Fish Conditions Stage Phenotype
bbs1ka742/ka742 (AB) standard conditions Days 7-13 eye detection of light stimulus involved in visual perception decreased process quality, abnormal
Days 7-13 photoreceptor outer segment layer photoreceptor disc membrane disorganized, abnormal
Days 7-13 photoreceptor outer segment layer shape, abnormal
Days 7-13 photoreceptor outer segment layer shortened, abnormal
Days 7-13 retinal pigmented epithelium inclusion body increased amount, abnormal
Acknowledgments:
This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ Nat. Commun.