Fig. 3.

Loss of Lss activity phenotype is epistatic to msmo1^nu81 mutation. (A) The lss^nu60 allele. PAM, protospacer adjacent motif (underlined in red). (B) Survival analysis of clutches of lss^nu60/+ in-crosses, from 9 dpf to 23 dpf, reveals that most lss^nu60 mutants die by 12 dpf. [9 dpf wild type (wt; black) n=21, heterozygotes (het; pale gray) n=30, knockout mutants (KO; dark gray) n=15, P=0.4412; 10 dpf wt n=15, het n=27, KO n=16, P=0.8563; 11 dpf wt n=20, het n=44, KO n=14, P=0.3320; 12 dpf wt n=24, het n=48, KO n=10, *P=0.0277; 13 dpf wt n=33, het n=51, KO n=10, **P=0.0026; 23 dpf wt n=8, het n=27, KO n=0, ***P=0.0009.] (C) Survival analysis of clutches of msmo1^nu81/+;lss^nu60/+ in-crosses reveals that loss of Lss activity increases survivability of msmo1^nu81 mutants at 10 dpf. [wt observed (O) n=11, expected (E) n=11; msmo1^nu81 O n=4, E n=11; msmo1^nu81;lss^nu60/+ O n=9, E n=22; msmo1^nu81;lss^nu60 O n=10, E n=11; lss^nu60 O n=12, E n=11; msmo1^nu81/+;lss^nu60 O n=30, E n=22; msmo1^nu81/+;lss^nu60/+ O n=40, E n=44; msmo1^nu81/+ O n=18, E n=22; lss^nu60/+ O n=18, E n=22; *P=0.0431.] Two-tailed P-values were calculated using chi-squared test. (D,E) Suppression of early lethality of lss^nu60 mutants by liver-specific expression of lss. (D) Adult Tg(fabp10:lss:pA)^nu101;lss^nu60 mutants phenocopy kol^nu7. Tg(fabp10:lss:pA)^nu101 n=12, Tg(fabp10:lss:pA)^nu101;lss^nu60 n=12. (E) Whole-mount skeletal preparations reveal gross malformations throughout Tg(fabp10:lss:pA)^nu101;lss^nu60 craniofacial and axial skeleton, similar to those observed in kol^nu7. Tg(fabp10:lss:pA)^nu101 n=3, Tg(fabp10:lss:pA)^nu101;lss^nu60 n=5.