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Hedgehog signalling in the mouse requires intraflagellar transport proteins

Author

Listed:
  • Danwei Huangfu

    (Sloan-Kettering Institute
    Cornell University)

  • Aimin Liu

    (Sloan-Kettering Institute
    Cornell University)

  • Andrew S. Rakeman

    (Sloan-Kettering Institute
    Molecular Cell and Developmental Biology Program
    Cornell University)

  • Noel S. Murcia

    (Case Western Reserve University)

  • Lee Niswander

    (Sloan-Kettering Institute
    Cornell University)

  • Kathryn V. Anderson

    (Sloan-Kettering Institute)

Abstract

Intraflagellar transport (IFT) proteins were first identified as essential factors for the growth and maintenance of flagella in the single-celled alga Chlamydomonas reinhardtii1. In a screen for embryonic patterning mutations induced by ethylnitrosourea, here we identify two mouse mutants, wimple (wim) and flexo (fxo), that lack ventral neural cell types and show other phenotypes characteristic of defects in Sonic hedgehog signalling. Both mutations disrupt IFT proteins: the wim mutation is an allele of the previously uncharacterized mouse homologue of IFT172; and fxo is a new hypomorphic allele of polaris, the mouse homologue of IFT88. Genetic analysis shows that Wim, Polaris and the IFT motor protein Kif3a are required for Hedgehog signalling at a step downstream of Patched1 (the Hedgehog receptor) and upstream of direct targets of Hedgehog signalling. Our data show that IFT machinery has an essential and vertebrate-specific role in Hedgehog signal transduction.

Suggested Citation

  • Danwei Huangfu & Aimin Liu & Andrew S. Rakeman & Noel S. Murcia & Lee Niswander & Kathryn V. Anderson, 2003. "Hedgehog signalling in the mouse requires intraflagellar transport proteins," Nature, Nature, vol. 426(6962), pages 83-87, November.
    • Handle: RePEc:nat:nature:v:426:y:2003:i:6962:d:10.1038_nature02061
    DOI: 10.1038/nature02061
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    Cited by:

    1. Kodaji Ha & Nadine Mundt-Machado & Paola Bisignano & Aide Pinedo & David R. Raleigh & Gabriel Loeb & Jeremy F. Reiter & Erhu Cao & Markus Delling, 2024. "Cilia-enriched oxysterol 7β,27-DHC is required for polycystin ion channel activation," Nature Communications, Nature, vol. 15(1), pages 1-13, December.
    2. Xiaoliang Liu & Patricia T. Yam & Sabrina Schlienger & Eva Cai & Jingyi Zhang & Wei-Ju Chen & Oscar Torres Gutierrez & Vanesa Jimenez Amilburu & Vasanth Ramamurthy & Alice Y. Ting & Tess C. Branon & M, 2024. "Numb positively regulates Hedgehog signaling at the ciliary pocket," Nature Communications, Nature, vol. 15(1), pages 1-20, December.

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