FIGURE

Fig. 4.

ID

ZDB-FIG-230119-39

Publication

Wang et al., 2022 - Variable phenotypes and penetrance between and within different zebrafish ciliary transition zone mutants

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Fig. 4.

Immunohistochemical (IHC) and transcriptional analysis of rod and cone photoreceptors for embryonic and adult retina sections of WT and cilia-related mutants. (A) IHC staining of retinal cryosections of WT, and ift88, ift172, b9d2, mks1, tmem67, rpgrip1l, tctn1, tmem216, cc2d2a, cep290, nphp1 and nphp4 mutant embryos at 5 dpf. Dorsal views are shown. Images of IHC staining of cone photoreceptors with Zpr1 (green; inner segment), PNA (red; outer segment) and DAPI (blue; nuclei) in column 1. Higher-magnification images are in column 2. Images of IHC staining of rod photoreceptors with Zpr3 (red; rhodopsin – outer segment if trafficked properly) and nuclei with DAPI (blue) are in column 3. Higher-magnification images are in column 4. Scale bars: 50 µm (columns 1 and 3); 25 µm (columns 2 and 4). (B) The table depicting retinal defects observed in mutant embryos. ift172^−/− and ift88^−/− results are highlighted by a red box. (C) K-means clustering of log2 normalized counts of the 65 retinal genes. The experiment workflow for how we defined these 65 genes is shown in Fig. S9A. Colored sidebars represent different clustering groups (yellow, blue, red and orange). (D) Representative cryosectioned 3-month-old adult retinae of WT, and viable b9d2, cep290, tmem67, rpgrip1l, tctn1, nphp1 and nphp4 mutants stained with Zpr1/PNA (green/red; column 1), Zpr3 (red; column 2), Pcna (proliferating cells; red; column 3) and L-plastin/4C4 (green; pan-leukocyte marker/red; microglia marker; column 4). Scale bars: 100 µM.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	b9d2^uab17/uab17 (AB) cc2d2a^uab16/uab16 (AB) cep290^uab18/uab18 (AB) ift88^tz288/tz288 ift172^uab11/uab11 (AB) mks1^uab12/uab12 (AB) nphp1^uab20/uab20 (AB) nphp4^uab21/uab21 (AB) rpgrip1l^uab15/uab15 (AB) tctn1^uab19/uab19 (AB) tmem67^uab14/uab14 (AB) tmem216^uab13/uab13 (AB)
Observed In:	retina retina central region retina inflammatory response retinal cone cell retinal cone cell cone photoreceptor outer segment retinal cone cell photoreceptor inner segment retinal cone cell photoreceptor outer segment retinal outer nuclear layer cell population proliferation retinal rod cell retinal rod cell photoreceptor inner segment
Stage Range:	Day 5 to Adult

Phenotype Detail

Acknowledgments

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