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
-
- Zebrafish/genetics
- Zebrafish/growth & development
- Zebrafish/metabolism
- Fish Proteins/genetics
- Fish Proteins/metabolism*
- Mutation, Missense
- Amino Acid Sequence
- Mice
- Cell Cycle*
- RNA, Ribosomal/biosynthesis*
- RNA, Ribosomal/genetics
- Molecular Sequence Data
- Phenotype
- Thymus Gland/growth & development*
- Thymus Gland/metabolism
- RNA Processing, Post-Transcriptional
- Cell Nucleolus/genetics
- Cell Nucleolus/metabolism
- Animals
- Sequence Alignment
- Nuclear Proteins/genetics
- Nuclear Proteins/metabolism*
- Mice, Knockout
- Oryzias/genetics
- Oryzias/growth & development*
- Oryzias/metabolism*
- NIH 3T3 Cells
- PubMed
- 18769712 Full text @ PLoS Genet.
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.
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
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping