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A histone H3 lysine 36 trimethyltransferase links Nkx2-5 to Wolf–Hirschhorn syndrome

Author

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  • Keisuke Nimura

    (Osaka University Graduate School of Medicine, 2-2 Yamada-oka, Suita, Osaka 565-0871, Japan)

  • Kiyoe Ura

    (Osaka University Graduate School of Medicine, 2-2 Yamada-oka, Suita, Osaka 565-0871, Japan)

  • Hidetaka Shiratori

    (Developmental Genetics Group, Graduate School of Frontier Biosciences, and,)

  • Masato Ikawa

    (Genome Information Research Center, Osaka University, 1-3 Yamada-oka, Suita, Osaka 565-0871, Japan)

  • Masaru Okabe

    (Genome Information Research Center, Osaka University, 1-3 Yamada-oka, Suita, Osaka 565-0871, Japan)

  • Robert J. Schwartz

    (Institute of Bioscience and Technology, Center for Molecular Development and Disease, 2121 West Holcombe Boulevard, Houston, Texas 77030, USA)

  • Yasufumi Kaneda

    (Osaka University Graduate School of Medicine, 2-2 Yamada-oka, Suita, Osaka 565-0871, Japan)

Abstract

Transcription factor Nkx2-5 and Wolf-Hirschhorn syndrome Histone H3 trimethylation at lysine 36 (H3K36me3) is associated with actively transcribed regions and has been proposed to provide landmarks for ongoing transcription. Here, an H3K36me3-specific histone methyltransferase Wolf–Hirschhorn syndrome candidate 1 (WHSC1) is shown to function in transcriptional regulation together with developmental transcription factors such as Nkx2-5. Whsc1-deficient mice display growth and developmental defects similar to those seen in WHS patients, and the defects are more severe when Nkx2-5 is also deficient. WHSC1 seems to function together with developmental transcription factors to prevent transcriptional pathophysiologies.

Suggested Citation

  • Keisuke Nimura & Kiyoe Ura & Hidetaka Shiratori & Masato Ikawa & Masaru Okabe & Robert J. Schwartz & Yasufumi Kaneda, 2009. "A histone H3 lysine 36 trimethyltransferase links Nkx2-5 to Wolf–Hirschhorn syndrome," Nature, Nature, vol. 460(7252), pages 287-291, July.
    • Handle: RePEc:nat:nature:v:460:y:2009:i:7252:d:10.1038_nature08086
    DOI: 10.1038/nature08086
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    Cited by:

    1. Ko Sato & Amarjeet Kumar & Keisuke Hamada & Chikako Okada & Asako Oguni & Ayumi Machiyama & Shun Sakuraba & Tomohiro Nishizawa & Osamu Nureki & Hidetoshi Kono & Kazuhiro Ogata & Toru Sengoku, 2021. "Structural basis of the regulation of the normal and oncogenic methylation of nucleosomal histone H3 Lys36 by NSD2," Nature Communications, Nature, vol. 12(1), pages 1-10, December.

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