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

Reference:

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.

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Official URL:

http://dx.doi.org/10.1371/journal.pgen.1000171

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.

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