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PLoS Genet. 2008 Aug 29;4(8):e1000171. doi: 10.1371/journal.pgen.1000171.

WDR55 is a nucleolar modulator of ribosomal RNA synthesis, cell cycle progression, and teleost organ development.

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1
Division of Experimental Immunology, Institute for Genome Research, University of Tokushima, Tokushima, Japan. iwanami@genome.tokushima-u.ac.jp

Erratum in

  • PLoS Genet. 2008 Sep;4(9). doi: 10.1371/annotation/6683988c-4647-4550-88e3-e8ba052f7916.

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.

PMID:
18769712
PMCID:
PMC2515640
DOI:
10.1371/journal.pgen.1000171
[Indexed for MEDLINE]
Free PMC Article
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