- Title
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DNAH6 and Its Interactions with PCD Genes in Heterotaxy and Primary Ciliary Dyskinesia
- Authors
- Li, Y., Yagi, H., Onuoha, E.O., Damerla, R.R., Francis, R., Furutani, Y., Tariq, M., King, S.M., Hendricks, G., Cui, C., Saydmohammed, M., Lee, D.M., Zahid, M., Sami, I., Leatherbury, L., Pazour, G.J., Ware, S.M., Nakanishi, T., Goldmuntz, E., Tsang, M., Lo, C.W.
- Source
- Full text @ PLoS Genet.
dnah6 morpholino knockdown in zebrafish embryo cause laterality defects. (A,B) dnah6 MO injected embryos exhibited curly tail and cardiac edema phenotype at 48 hours post fertilization (hpf). (C,D) dnah6 MO injected embryos at 48hpf exhibited heart (C) and gut (D) looping defects, including failure to loop (middle panels) and reversal looping (right panels). (E) In situ hybridization analysis revealed abnormal right sided (R), bilateral (B), and absent (Ab) spaw expression after dnah6 MO knockdown. (F,G) Heart/gut looping defects were observed in dnah6 MO-injected embryos, including normal (N), right sided (R), and straight (St) heart/gut looping phenotypes. (H). KV cilia were shorter (middle) in dnah6 AUG-MO but not dnah6 spl-MO injected embryos (p-value = 0.0017). (I). High-speed videomicroscopy showed reduction in KV motile cilia of dnah6 MO injected embryos (p = 1.6x10-4 for 7.5 ng and p = 1.1x10-5 for 10 ng dnah6 MO; two-tailed Student’s t-test). (J) Little bead movement (color tracing) was observed in dnah6 MO injected embryos. (K) KV flow was absent or reduced with dnah6 knockdown (Chi-square test, p-value = 0.0192). PHENOTYPE:
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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. PHENOTYPE:
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Analysis of CCNO, FOXJ1 and MCIDAS transcripts with DNAH6 knockdown. (A, B) qPCR analysis showed CCNO, FOXJ1 and MCIDAS transcripts were not affected in human and mouse respiratory epithelia after Dnah6 knockdown as compared to control. (C) In situ hybridization showed foxj1 expression level and pattern did not change in zebrafish embryos after dnah6 antisense morpholino gene knockdown. |
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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. PHENOTYPE:
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dnah6 in situ hybridization analysis of early zebrafish embryos. In situ hybridization analysis showed dnah6 transcripts are ubiquitously expressed as maternally derived transcript in the early zebrafish embryo. EXPRESSION / LABELING:
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