PUBLICATION

WDR55 Is a Nucleolar Modulator of Ribosomal RNA Synthesis, Cell Cycle Progression, and Teleost Organ Development

Authors
Iwanami, N., Higuchi, T., Sasano, Y., Fujiwara, T., Hoa, V.Q., Okada, M., Talukder, S.R., Kunimatsu, S., Li, J., Saito, F., Bhattacharya, C., Matin, A., Sasaki, T., Shimizu, N., Mitani, H., Himmelbauer, H., Momoi, A., Kondoh, H., Furutani-Seiki, M., and Takahama, Y.
ID
ZDB-PUB-080908-13
Date
2008
Source
PLoS Genetics   4(8): e1000171 (Journal)
Registered Authors
Furutani-Seiki, Makoto, Kondoh, Hisato
Keywords
Thymus, Embryos, Ribosomal RNA, Zebrafish, Cell cycle and cell division, Nucleolus, Point mutation, Complementary DNA
MeSH Terms
  • Amino Acid Sequence
  • Nuclear Proteins/genetics
  • Nuclear Proteins/metabolism*
  • RNA Processing, Post-Transcriptional
  • Fish Proteins/genetics
  • Fish Proteins/metabolism*
  • Cell Cycle*
  • Zebrafish/genetics
  • Zebrafish/growth & development
  • Zebrafish/metabolism
  • Animals
  • Oryzias/genetics
  • Oryzias/growth & development*
  • Oryzias/metabolism*
  • Phenotype
  • Sequence Alignment
  • RNA, Ribosomal/biosynthesis*
  • RNA, Ribosomal/genetics
  • NIH 3T3 Cells
  • Cell Nucleolus/genetics
  • Cell Nucleolus/metabolism
  • Molecular Sequence Data
  • Mice
  • Thymus Gland/growth & development*
  • Thymus Gland/metabolism
  • Mutation, Missense
  • Mice, Knockout
(all 27)
PubMed
18769712 Full text @ PLoS Genet.
Abstract
The thymus is a vertebrate-specific organ where T lymphocytes are generated. Genetic programs that lead to thymus development are incompletely understood. We previously screened ethylnitrosourea-induced medaka mutants for recessive defects in thymus development. Here we report that one of those mutants is caused by a missense mutation in a gene encoding the previously uncharacterized protein WDR55 carrying the tryptophan-aspartate-repeat motif. We find that WDR55 is a novel nucleolar protein involved in the production of ribosomal RNA (rRNA). Defects in WDR55 cause aberrant accumulation of rRNA intermediates and cell cycle arrest. A mutation in WDR55 in zebrafish also leads to analogous defects in thymus development, whereas WDR55-null mice are lethal before implantation. These results indicate that WDR55 is a nuclear modulator of rRNA synthesis, cell cycle progression, and embryonic organogenesis including teleost thymus development.
Genes / Markers
Figures
Figure Gallery (1 images)
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Expression
Phenotype
Fish Conditions Stage Phenotype Figure
wdr55hi2786bTg/hi2786bTgstandard conditionsDay 6
wdr55hi2786bTg/hi2786bTgstandard conditionsDay 6
wdr55hi2786bTg/hi2786bTgstandard conditionsDay 6
wdr55hi2786bTg/hi2786bTgstandard conditionsDay 6
wdr55hi2786bTg/hi2786bTgstandard conditionsDays 7-13
1 - 5 of 5
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Mutations / Transgenics
Allele Construct Type Affected Genomic Region
hi2786bTgTransgenic Insertion
1 - 1 of 1
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Human Disease / Model
No data available
Sequence Targeting Reagents
No data available
Fish
Fish
wdr55hi2786bTg/hi2786bTg
wdr55hi2786bTg/+
WT
1 - 3 of 3
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Antibodies
No data available
Orthology
No data available
Engineered Foreign Genes
No data available
Mapping
No data available
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Citation
Iwanami, N., Higuchi, T., Sasano, Y., Fujiwara, T., Hoa, V.Q., Okada, M., Talukder, S.R., Kunimatsu, S., Li, J., Saito, F., Bhattacharya, C., Matin, A., Sasaki, T., Shimizu, N., Mitani, H., Himmelbauer, H., Momoi, A., Kondoh, H., Furutani-Seiki, M., and Takahama, Y. (2008) WDR55 Is a Nucleolar Modulator of Ribosomal RNA Synthesis, Cell Cycle Progression, and Teleost Organ Development. PLoS Genetics. 4(8):e1000171.