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. 2018 Jul 12;29(8):2110–2122. doi: 10.1681/ASN.2017121338

Figure 1.

Figure 1.

Transient suppression and CRISPR/Cas9 genome editing of anillin (anln) demonstrates loss-of-function phenotypes for two human FSGS mutations. (A) Zebrafish larvae injected with 3 ng morpholino (MO) alone or with 150 pg ANLN mRNA were scored live at 4 days postfertilization (dpf) for edema as a proxy for glomerular filtration defects: class 1 (mild), mild ocular and pericardial edema; class 2 (severe), pronounced ocular, pericardial, and yolk sac edema (B). Statistical calculations were performed using a chi-squared test (n=42–56 per batch, repeated). p.G618C, p.Gly618Cys; p.R185K, p.Arg185Lys (likely benign variant because of common incidence in healthy control populations, allele frequency 0.6480; dbSNP ID: rs572020591); p.R431C, p.Arg431Cys; WT, wild type. (B) Representative live lateral images of zebrafish larvae at 4 dpf show different phenotypic classes. (C) CRISPR/Cas9 F0 mutants mimic anln morphant phenotypes. Qualitative scoring of zebrafish larvae at 4 dpf on the basis of objective phenotypic criteria (B). Statistical differences were compared using a chi-squared test (n=41–75 per batch, repeated). sgRNA, single-guide RNA.