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
- 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
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping