To confirm the role of CEP120 in vivo, we next performed knockdown of the zebrafish (ZF) homolog (CABZ01084007.1–201, ENSDART00000122378) using an antisense morpholino oligonucleotide targeting the inton1–exon2 splice site resulted in a typical ciliopathy phenotype with ventrally curved body axis, hydrocephalus, otolith defects, heart edema and smaller eyes at 3 days post-fertilization compared with control morpholino-injected embryos (Fig. 4A–G; Supplementary Material, Fig. S6A). This evidence concerns the gene CEP120 and ciliopathy.