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

Research output: Contribution to journalArticlepeer-review

18 Citations (Scopus)

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.
Original languageEnglish
Article numbere1000171
Number of pages13
JournalPlos Genetics
Volume4
Issue number8
DOIs
Publication statusPublished - 29 Aug 2008

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