ZFIN ID: ZDB-FIG-121212-42
Blaker-Lee et al., 2012 - Zebrafish homologs of 16p11.2, a genomic region associated with brain disorders, are active during brain development, and include two deletion dosage sensor genes. Disease models & mechanisms   5(6):834-851 Full text @ Dis. Model. Mech.
ADDITIONAL FIGURES
PHENOTYPE:
Fish:
Knockdown Reagents:
Observed In:
Stage Range: Prim-5 to Long-pec

Fig. 3

Phenotypes obtained after LOF of zebrafish 16p11.2 homologs. Embryonic phenotypes observed after LOF (Fig. 2) are catalogued, and structures or assays are indicated at the top of each column. All assays were performed in comparison with embryos injected with control MO. Quantification of phenotypes is given in supplementary material Table S3, and rescue conditions are shown in Fig. 4 and Table 3. aGenes assayed are indicated in the left column and gene identifiers are included in supplementary material Table S1. MOs targeting these genes are included in supplementary material Table S2. bMorphological analyses addressed head morphology (brain ventricle shape and eye formation), tail shape and length, and muscle segment shape (chevron vs U-shape). cTwo types of movement were tested: spontaneous movement at 24 hpf and touch response at 48 hpf. The ino80e LOF embryos respond with one flip of the tail or not at all. mapk3 LOF embryos have a jerky response and the ypel3 LOF embryos move in small, jerky circles. mvp LOF embryos range from not responding to spinning in response to touch. Otherwise, touch response was weak, sluggish or not responsive. dInitial axon tracts form by 36 hpf and were assayed by immunostaining for acetylated tubulin. Axon tracts were not assayed in mvp LOF embryos owing to lack of rescue, in prrt2 LOF owing to lack of observable phenotype at these time points, and in tbx24 LOF owing to lack of head expression. ePigmentation was observed at 48 hpf in LOF embryos and images are included in supplementary material Fig. S3. fPersistence of early phenotypic abnormalities was monitored up to 5 dpf. 1The mvp MO used for the phenotype reported is indicated with matching superscript and listed first in Table 1 and supplementary material Table S2. Red boxes: abnormal phenotype in >70% embryos; speckled red boxes: abnormal phenotype in >70% of embryos, but the phenotype was mild. ND, not determined.

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
WT + MO1-aldoaa standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
Long-pec whole organism pigmented, abnormal
WT + MO1-asphd1 standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
WT + MO1-coro1a standard conditions Prim-5 eye morphology, abnormal
Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 whole organism movement quality, abnormal
Prim-25 central nervous system projection neuron axonogenesis disrupted, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
Long-pec whole organism pigmented, abnormal
WT + MO1-fam57ba standard conditions Prim-5 eye morphology, abnormal
Prim-5 somite U-shaped, abnormal
Prim-5 whole organism movement quality, abnormal
Prim-25 central nervous system projection neuron axonogenesis disrupted, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
Long-pec whole organism pigmented, abnormal
WT + MO1-gdpd3a standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Prim-5 whole organism movement quality, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
Long-pec whole organism pigmented, abnormal
WT + MO1-hirip3 standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Prim-5 whole organism movement quality, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
Long-pec whole organism pigmented, abnormal
WT + MO1-ino80e standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
Long-pec whole organism pigmented, abnormal
WT + MO1-kif22 standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Prim-25 central nervous system projection neuron axonogenesis disrupted, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
WT + MO1-maza standard conditions Prim-5 eye morphology, abnormal
Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
WT + MO1-pagr1 standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
WT + MO1-taok2a standard conditions Prim-5 somite U-shaped, abnormal
WT + MO1-taok2b standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
WT + MO1-tbx6 standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
WT + MO1-ypel3 standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
WT + MO2-kctd13 standard conditions Prim-25 central nervous system projection neuron axonogenesis disrupted, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
WT + MO2-sez6l2 standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
WT + MO3-mvp standard conditions Prim-5 eye morphology, abnormal
Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
WT + MO5-mapk3 standard conditions Prim-5 eye morphology, abnormal
Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Prim-25 central nervous system projection neuron axonogenesis disrupted, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
Long-pec whole organism pigmented, abnormal
WT + MO5-ppp4ca standard conditions Prim-5 post-vent region decreased length, abnormal
Prim-5 post-vent region shape, abnormal
Prim-5 somite U-shaped, abnormal
Prim-25 central nervous system projection neuron axonogenesis disrupted, abnormal
Long-pec thigmotaxis decreased process quality, abnormal
Acknowledgments:
ZFIN wishes to thank the journal Disease models & mechanisms for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ Dis. Model. Mech.