ZFIN ID: ZDB-FIG-220520-8
Duclaux-Loras et al., 2022 - UNC45A deficiency causes microvillus inclusion disease-like phenotype by impairing myosin VB-dependent apical trafficking. The Journal of Clinical Investigation   132(10) Full text @ Journal of Clin. Invest.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Antibodies:
Fish:
Anatomical Terms:
Stage: Day 5
PHENOTYPE:
Fish:
Observed In:
Stage: Day 5

Figure 7 MVID features in enterocytes of <italic toggle='yes'>unc45a</italic> zebrafish mutants.

(AC) Confocal microscopy analysis of the intestinal bulb of WT and unc45a–/– mutant larvae stained for villin and pERM (A), F-actin (phalloidin) (B), and Rab11 (C) at 5 dpf. Scale bars: 20 μm. Boxes showing microvillus-like inclusions are enlarged ×4. (D and E) TEMs of thin sections of intestinal bulb of 5 dpf WT and unc45a–/– mutant larvae showing defects in the organization of the brush border. Scale bars: 2 μm (D); 0.5 μm (E). Quantitative analysis showing decrease in microvillus length and density in unc45a–/– enterocytes as compared with WT enterocytes. Data are represented as mean + SD. ****P < 0.001, t test. N nucleus; MV microvilli.

Gene Expression Details No data available
Antibody Labeling Details
Antibody Assay Fish Conditions Stage Anatomy
Ab1-villin IHC WT standard conditions Day 5 enterocyte apical plasma membrane
IHC unc45atr12/tr12 standard conditions Day 5 enterocyte apical plasma membrane
Ab2-rab11 IHC WT standard conditions Day 5 enterocyte vesicle
IHC unc45atr12/tr12 standard conditions Day 5 intestinal epithelial cell basolateral plasma membrane
Phenotype Details
Fish Conditions Stage Phenotype
unc45atr12/tr12 standard conditions Day 5 enterocyte apical plasma membrane Ab1-villin labeling spatial pattern, abnormal
Day 5 enterocyte apical plasma membrane invaginated, abnormal
Day 5 enterocyte microvillus decreased amount, abnormal
Day 5 enterocyte microvillus decreased length, abnormal
Day 5 intestinal epithelial cell basolateral plasma membrane ab2-rab11 labeling mislocalised, 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 @ Journal of Clin. Invest.