FIGURE

Fig. 7

ID

ZDB-FIG-220914-112

Publication

Zhang et al., 2022 - GCAF(TMEM251) regulates lysosome biogenesis by activating the mannose-6-phosphate pathway

Other Figures

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Fig. 7

a Sequence alignments of human GCAF short isoform and zebrafish GCAF. b Zebrafish GCAF rescued the GNPTAB processing defects in GCAF KO HEK293T cells (n = 3 independent replicates). c Zebrafish GCAF rescued the lysosome function in GCAF KO HEK293T cells (n = 3 independent replicates). d Morphology of the F0 GCAF and GNPTAB deficient fish at 5 and 7 dpf. Arrowheads point to heart edema. e Quantification of morphological phenotypes observed in d. Mean of 3 independent replicates is shown. Error bars represent standard deviation. *p ≤ 0.05, **p ≤ 0.01, ***p ≤ 0.001. See source data file for exact P values. f χ² test to compare control, sgGCAF, and sgGNPTAB embryos. ****p ≤ 0.0001. The numbers in x-axis represent the fishes included in the quantification. g Ventral view of alcian blue stained zebrafish larvae at 4 dpf. ch: ceratohyal, m: Meckel’s cartilage; cb: ceratobranchials. h Alizarin red staining of zebrafish embryos at 7 dpf. Arrowheads: ear stones. Arrows: vertebrate columns.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	WT + CRISPR1-gnptab + CRISPR2-gnptab + CRISPR3-gnptab + CRISPR4-gnptab WT + CRISPR1-tmem251 + CRISPR2-tmem251 + CRISPR3-tmem251 + CRISPR4-tmem251
Knockdown Reagents:	CRISPR1-gnptab CRISPR1-tmem251 CRISPR2-gnptab CRISPR2-tmem251 CRISPR3-gnptab CRISPR3-tmem251 CRISPR4-gnptab CRISPR4-tmem251
Observed In:	cranial cartilage embryonic cranial skeleton morphogenesis pericardium post-vent region
Stage Range:	Day 4 to Days 7-13

Phenotype Detail

Acknowledgments

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