Loss of zygotic NUP107 protein causes missing of pharyngeal skeleton and other tissue defects with impaired nuclear pore function in zebrafish embryos

Zheng, X., Yang, S., Han, Y., Zhao, X., Zhao, L., Tian, T., Tong, J., Xu, P., Xiong, C., and Meng, A.
The Journal of biological chemistry   287(45): 38254-38264 (Journal)
Registered Authors
Han, Yanchao, Meng, Anming, Tian, Tian, Tong, Jingyuan, Xiong, Cong, Xu, Pengfei, Zhao, Long
cartilage, embryo, mutant, nuclear pore, zebrafish, Nup107, nucleoporin
MeSH Terms
  • Animals
  • Animals, Genetically Modified
  • Apoptosis/genetics
  • Chondrogenesis/genetics
  • Embryo, Nonmammalian/embryology
  • Embryo, Nonmammalian/metabolism*
  • Eye Abnormalities/embryology
  • Eye Abnormalities/genetics
  • Female
  • Gene Expression Regulation, Developmental
  • Gene Knockdown Techniques
  • Green Fluorescent Proteins/genetics
  • Green Fluorescent Proteins/metabolism
  • Immunohistochemistry
  • In Situ Hybridization
  • Intestines/abnormalities
  • Intestines/metabolism
  • Male
  • Microscopy, Confocal
  • Mutation
  • Nuclear Pore/metabolism*
  • Nuclear Pore/physiology
  • Nuclear Pore Complex Proteins/genetics*
  • Nuclear Pore Complex Proteins/metabolism
  • Pharynx/abnormalities
  • Pharynx/metabolism*
  • RNA Transport/genetics
  • RNA, Messenger/genetics
  • RNA, Messenger/metabolism
  • Reverse Transcriptase Polymerase Chain Reaction
  • Zebrafish/embryology
  • Zebrafish/genetics*
  • Zebrafish/metabolism
  • Zebrafish Proteins/genetics*
  • Zebrafish Proteins/metabolism
  • Zygote/metabolism
22965233 Full text @ J. Biol. Chem.

The Nup107-160 multiprotein subcomplex is essential for the assembly of nuclear pore complexes. The developmental functions of individual constituents of this subcomplex in vertebrates remain elusive. In particular, it is unknown whether Nup107 plays an important role in development of vertebrate embryos. Zebrafish nup107 is maternally expressed and its zygotic expression becomes prominent in the head region and the intestine from 24 hours postfertilization (hpf) onward. In this study, we generate a zebrafish mutant line, nup107tsu068Gt, in which the nup107 locus is disrupted by an insertion of Tol2 transposon element in the first intron and as a result it fails to produce normal transcripts. Homozygous nup107tsu068Gt mutant embryos exhibit tissue-specific defects after 3 days postfertilization (dpf), including loss of the pharyngeal skeletons, degeneration of the intestine, absence of the swim bladder, and smaller eyes. These mutants die at 5-6 days. Extensive apoptosis occurs in the affected tissues, which is partially dependent on p53 apoptotic pathways. In cells of the defective tissues, FG-repeat nucleoporins are disturbed and nuclear pore number is reduced, leading to impaired translocation of mRNAs from the nucleus to the cytoplasm. Our findings shed new light on developmental function of Nup107 in vertebrates.

Genes / Markers
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Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Engineered Foreign Genes