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

Norimasa Iwanami, Tomokazu Higuchi, Yumi Sasano, Toshinobu Fujiwara, Vu Q. Hoa, Minoru Okada, Sadiqur R. Talukder, Sanae Kunimatsu, Jie Li, Fumi Saito, Chitralekha Bhattacharya, Angabin Matin, Takashi Sasaki, Nobuyoshi Shimizu, Hiroshi Mitani, Heinz Himmelbauer, Akihiro Momoi, Hisato Kondoh, Makoto Furutani-Seiki, Yousuke Takahama

研究成果: Article査読

18 被引用数 (Scopus)

抄録

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.

本文言語English
論文番号e1000171
ジャーナルPLoS Genetics
4
8
DOI
出版ステータスPublished - 2008 8

ASJC Scopus subject areas

  • Ecology, Evolution, Behavior and Systematics
  • Molecular Biology
  • Genetics
  • Genetics(clinical)
  • Cancer Research

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