FIGURE SUMMARY
Title

Role of the ADCY9 gene in cardiac abnormalities of the Rubinstein-Taybi syndrome

Authors
Wu, Y., Xia, Y., Li, P., Qu, H.Q., Liu, Y., Yang, Y., Lin, J., Zheng, M., Tian, L., Wu, Z., Huang, S., Qin, X., Zhou, X., Chen, S., Liu, Y., Wang, Y., Li, X., Zeng, H., Hakonarson, H., Zhuang, J.
Source
Full text @ Orphanet J Rare Dis
Fig. 1

Phenotypes of adcy9 zebrafish morphants. (a-f) Gross morphology at 3-dpf. Compared with control MO, knock down adcy9 present pericardial oedema (b, d, f, red arrow) (Heart beat is visible in the control fish, but is abnormal in adcy9-e3i3-MO injected fish (Supplementary Movie 1, Supplementary Movie 2). The bar graph in Panel G shows the percentage of embryos with development defects. h A time-course plot of percent survival in control vs. adcy9 morphants for 5 days

PHENOTYPE:
Fish:
Knockdown Reagent:
Observed In:
Stage Range: 1-cell to Day 5
Fig. 2

Effects of adcy9 knock down on macrophage migration. a-f Representative bright field and fluorescent images of TG (zlyz: EGFP) larvae at 6 days postfertilization (dpf). Control MO injected fish show the normal distribution of labeled cells (b and c). Compared with control fish, embryos injected with adcy9-e3i3-MO present potent macrophage migration in heart (e and f, red circle area). g-k Quantification of the macrophage number at heart shows a 13.3-fold increased in adcy9 morphants. Columns, mean; bars, SEM (n = 10; Student’s t test; ***P < 0.0001;). dpf, days post fertilization. l Endogenous efnb2a, COX2, mmp9, ptp-rb and pik3r2 in control and adcy9 morphants assessed by qRT-PCR (n = 6–10 individual embryos). ns, not significant

EXPRESSION / LABELING:
Genes:
Fish:
Knockdown Reagent:
Anatomical Term:
Stage: Protruding-mouth
PHENOTYPE:
Fish:
Knockdown Reagent:
Observed In:
Stage Range: Protruding-mouth to Day 6
Fig. 3

Morpholino knock down of adcy9 induces potent apoptosis in heart. Control-MO injected embryos and embryos injected with adcy9-e3i3-MO were stained with acridine orange (AO) at 3-dpf. Heart apoptotic cells are visible as bright green spots or red spots (f-h), and less bright homogenous green staining is unspecific background staining. a-d Control-MO injected zebrafish exhibited few or no apoptotic cells in heart. In contrast, significantly increased staining was observed throughout the heart in zebrafish injected with 4 ng adcy9-e3i3-MO (F-H). The red boxed regions are shown at higher magnification in the right panels. i Quantification of apoptosis particle number at heart shows a 66.1-fold increased in adcy9 morphants. Error bars, s.e.m.; ***P < 0.0001(n = 10; Student’s t test;); a-h: lateral view, anterior, left. Dpf, days post fertilization. j Endogenous tnfsf10l3, hdr, tnfrsfa, caspase-8, tp53, pmaip1, bbc3, badb, baxa, baxb, caspase-3a and caspase-a in control and adcy9 morphants assessed by qRT-PCR (n = 6–10 individual embryos). ns, not significant

EXPRESSION / LABELING:
Genes:
Fish:
Knockdown Reagent:
Anatomical Term:
Stage: Protruding-mouth
PHENOTYPE:
Fish:
Knockdown Reagent:
Observed In:
Stage: Protruding-mouth
text only

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Acknowledgments
This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ Orphanet J Rare Dis
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