(A) tfap2a^-/-;tfap2c^-/- mutants present the first morphological phenotypes at the 15 somite stage. (B) By 28 hpf the morphological phenotype leads to an overall dorsalised form, bifurcation of the forming eye, heart oedema, and complete lack of neural crest cells. All other genotypes appear normal. (C) At 48 hpf the previously described reduction of melanocytes can be noted in tfap2a^-/-;tfap2c^+/+ embryos and a modest reduction of melanocytes can be identified in the dorsal tail (red arrow heads) in tfap2a^-/-;tfap2c^+/- mutants. (D) Quantification of melanocytes in the three corresponding genotypes at 36 hpf. (E) Chart indicating the number of differentially expressed gene 3’ ends identified with an adjusted p-value of <0.05 for each pairwise comparison of genotypes tfap2a^-/-;tfap2c^-/-, tfap2a^-/-;tfap2c^+/-, tfap2a^-/-;tfap2c^+/+ and tfap2a^+/+;tfap2c^-/- to tfap2a^+/+;tfap2c^+/+ siblings at 4 somites, 15 somites and 24 hpf (F) An UpSet[53] diagram to compare multiple pairwise DE gene lists derived from the tfap2a^-/-;tfap2c^-/- vs wild-type siblings (adj. p-value <0.05) for the 4 somite, 15 somite and 24 hpf stages and the list of neural crest-enriched genes derived from sorted neural crest cells at 22–23 hpf. The horizontal black bars represent the size of the gene lists. Individual subsets are marked with a black dot and overlaps with a connecting line. The number of genes in each subset is shown above each vertical bar. The vertical bars are numbered consecutively along the x-axis. GO/ZFA enrichment was carried out on the subset of the 4 and 15 somite stages (blue box), the subsets indicated with the orange boxes and on all genes contained in the neural crest FACS enrichment and in at least one of the three different double knockout time points (magenta box). The developmental time course nature of the data allows for the grouping of the subsets into timing based on neural crest development starting with early neural crest-specific gene expression and then moving towards early-mid, mid, mid-later and later. The complete list of the 26 genes in group 13 can be found in S2 Table.

(A) Transcripts of members of the Hippo signalling pathway fat2, lats2 and yap1 were less abundant in tfap2a;tfap2c mutants when compared to wild-type siblings. A schematic showing their role in signal transduction and transcription inside a cell. (B) CRISPR/Cas9 mutations were made in the first exon of yap1 leading to the two alleles described. The exon-intron structure of the yap1 transcript is shown in gold. The exact deletions are displayed below. (C) Embryos from a single clutch were split and raised at 24C and 31.5C. Bars indicate the absolute number of fish forming a swim bladder at 5 dpf for each yap1^sa25458 genotype (D) Normalised counts of 3’ tag sequencing data at 24 hpf comparing yap1^sa25458 mutants to wild-type siblings. All four genes, yap1, gch2, wu:fc46h12 and padi2, have an adj. p-value <0.05. (E) Maternal zygotic yap1 mutants present a strong reduction in melanocyte numbers at 36 hpf at both dorsal (arrow head) and ventral tail regions (arrow). (F) Quantification of melanocytes with the quantities on the left and then broken down into the regions of the head, yolk, ventral tail and dorsal tail. Each dot represents a region in a single larva, siblings in blue and MZyap1^sa25458 in red. A statistical significance of <0.05 is indicated with “*”.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

(A-H) Whole mount in situ analysis of wu:fc46h12 and gch2 as a pigment cell comparison. sox10^baz/+ heterozygotes embryos as sibling controls (A-B) and mutant sox10^baz1embryos at 24 hpf (C-D). At 48 hpf in situs were carried out on albino embryos to serve as wild-type controls (E-F) with arrows indicating the heart and arrow heads the dorsal aorta. A blow up of this region can be found in E’-F’. (G-H) Expression of gch2 and wu:fc46h12 at 48 hpf in sox10^baz1 mutants. I-J Wild-type and MZwu:fc46h12^sa30587 embryos at 36 hpf with oedema around the forming heart (J). (K) Wild-type sibling and mutant akr1b1^sa30579at 4 dpf with mutant larvae presenting a reduction of yellow colour produced by xanthophores. Magnifications indicated with a black box. (L) Wild-type sibling and mutant cax1^sa10712 larvae at 5 dpf. Close ups indicated by black boxes around the head show dull yellow colour and abnormal cell morphology in mutants (arrow head). (M) MZcax1^sa10712phenotype at 19 somite stage.

(A) Homozygous yap1 mutants are viable but present with a variation in size. (B) Quantification of size at two months of age with the corresponding genotypes for both yap1 alleles. A statistically significant difference with p-val <0.05 is indicated by “*”.