WDR55 is a nucleolar modulator of ribosomal RNA synthesis, cell cycle progression, and teleost organ development.
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 muta...
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oai:doaj.org-article:723628f5f9384418af7fe9381a1c29572021-12-02T20:03:12ZWDR55 is a nucleolar modulator of ribosomal RNA synthesis, cell cycle progression, and teleost organ development.1553-73901553-740410.1371/journal.pgen.1000171https://doaj.org/article/723628f5f9384418af7fe9381a1c29572008-08-01T00:00:00Zhttps://www.ncbi.nlm.nih.gov/pmc/articles/pmid/18769712/pdf/?tool=EBIhttps://doaj.org/toc/1553-7390https://doaj.org/toc/1553-7404The 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.Norimasa IwanamiTomokazu HiguchiYumi SasanoToshinobu FujiwaraVu Q HoaMinoru OkadaSadiqur R TalukderSanae KunimatsuJie LiFumi SaitoChitralekha BhattacharyaAngabin MatinTakashi SasakiNobuyoshi ShimizuHiroshi MitaniHeinz HimmelbauerAkihiro MomoiHisato KondohMakoto Furutani-SeikiYousuke TakahamaPublic Library of Science (PLoS)articleGeneticsQH426-470ENPLoS Genetics, Vol 4, Iss 8, p e1000171 (2008) |
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Genetics QH426-470 |
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Genetics QH426-470 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 WDR55 is a nucleolar modulator of ribosomal RNA synthesis, cell cycle progression, and teleost organ development. |
description |
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. |
format |
article |
author |
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 |
author_facet |
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 |
author_sort |
Norimasa Iwanami |
title |
WDR55 is a nucleolar modulator of ribosomal RNA synthesis, cell cycle progression, and teleost organ development. |
title_short |
WDR55 is a nucleolar modulator of ribosomal RNA synthesis, cell cycle progression, and teleost organ development. |
title_full |
WDR55 is a nucleolar modulator of ribosomal RNA synthesis, cell cycle progression, and teleost organ development. |
title_fullStr |
WDR55 is a nucleolar modulator of ribosomal RNA synthesis, cell cycle progression, and teleost organ development. |
title_full_unstemmed |
WDR55 is a nucleolar modulator of ribosomal RNA synthesis, cell cycle progression, and teleost organ development. |
title_sort |
wdr55 is a nucleolar modulator of ribosomal rna synthesis, cell cycle progression, and teleost organ development. |
publisher |
Public Library of Science (PLoS) |
publishDate |
2008 |
url |
https://doaj.org/article/723628f5f9384418af7fe9381a1c2957 |
work_keys_str_mv |
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1718375667013255168 |