Phenotypic analysis of zebrafish larvae carrying KDM5C variants treated with CU-CPT 4a. (A, F) Representative head images of CTRL, WT, c.3019del (A) or c.782-2A>T (F), and corresponding CU-CPT 4a-treated groups at 72 hpf. (B, G) Quantification of head area for CTRL, WT, c.3019del or c.782-2A>T, and their respective CU-CPT 4a-treated groups (n =10 per group). (C, H) Representative whole-body images of CTRL, WT, c.3019del (C) or c.782-2A>T (H), and corresponding CU-CPT 4a-treated groups. (D, I) Quantification of body length for the same groups (n =10 per group). (E, J) Quantification of eye area for the same groups (n =10 per group). (K, M) Representative alcian blue staining image of CTRL, WT, c.3019del (K) or c.782-2A>T (M), corresponding CU-CPT 4a-treated groups at 120 hpf. (L, N) Quantification of alcian blue staining area for CTRL, WT, c.3019del or c.782-2A>T, and their respective CU-CPT 4a-treated groups (n =3 per group). ANOVA was used to compare data among groups. Data are expressed as mean ± SD. Statistical significance was determined as follows: ns, no significant difference; *P < 0.05; **P < 0.01; ***P < 0.001; ****P < 0.0001.
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