Analysis of point mutations in mouse Sufu, which mediates Hedgehog signal transduction, in tumor formation

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Hedgehog (Hh) signal transduction is known to have important roles in cancer formation. To understand the in vivo function of Sufu, which is a negative mediator of Hh signaling, we made use of Sufu mutant mice, which we established by the RIKEN ENU-based gene-driven mutagenesis system. The results showed that Sufu was necessary to regulate subcellular localization as well as stabilization of the Gli3^<REP> transcription factor. These results are not consistent with the previous publications, indicating novel mechanism of Hh signal transduction.