FIGURE

Fig. 3

ID

ZDB-FIG-170824-2

Publication

Schmidts et al., 2015 - TCTEX1D2 mutations underlie Jeune asphyxiating thoracic dystrophy with impaired retrograde intraflagellar transport

Other Figures

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Fig. 3

Knockdown of tctex1d2 in zebrafish leads to a typical ciliopathy phenotype.

Whole-mount light microscopy showing control morpholino (mo)-injected embryos (a–d) and tctex1d2 morphants at 4 days post fertilization (e–h). Compared with controls, knockdown of tctex1d2 results in ventrally curved body axis (a,e), small eyes (b,f), pronephric cysts (c,g) and otolith defects (d,h). Alcian blue staining of cartilage identifies craniofacial cartilage defects in tctexd2 morphants (m,n) compared with controls (i,j). Immunofluorescence analysis after staining of cilia at 24 h.p.f. with anti-acetylated tubulin antibody reveals shorter cilia in the pronephric duct of tctex1d2 morphants (o, magnified in p) compared with control embryos (k, magnified in l); however, this difference was no longer evident at 48 h.p.f. (data not shown). Scale bars, 100 μm (a–j,m,n) or 50 μm (k,l,o,p).

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	WT + MO1-dynlt2b + MO4-tp53
Knockdown Reagents:	MO1-dynlt2b MO4-tp53
Observed In:	embryonic cranial skeleton morphogenesis eye otolith pronephric duct cilium pronephros trunk whole organism
Stage Range:	Prim-5 to Day 4

Phenotype Detail

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.