Deletion of sav1 causes larval lethality and abnormal gallbladder development. (A) Wild-type and mutant alleles of zebrafish sav1. sav1 mutants contain an 11?bp insertion (red), causing a frameshift and early stop codon (red box). (B) Morphology of embryos at 8?dpf. Dashed red lines on higher magnification images outline the gallbladder. Pan, pancreas; Gb, gallbladder. (C) Quantification of gallbladder area shown in B; gallbladder location is indicated by red circles. Unpaired t-test with equal s.d., P<0.0001. n=38, sav1+/+ or +/?; n=12, sav1?/?. Data are meanąs.d. (D) BODIPY FL C5 gallbladder uptake and ratiometric analysis at 8?dpf. Sample number (n) indicated within the graph. (E,E?) Hematoxylin and Eosin stained paraffin sections of normal gallbladder and liver in wild-type and sav1?/? mutants. Gb, gallbladder. Scale bars: 100 ?m. (F,F?) Adjacent sections stained for annexin A4 confirm abnormal biliary morphology. Gb, gallbladder; IBD, intrahepatic bile duct. n=5 for both wild-type and sav1?/? paraffin histology.
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