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Genetic Analysis of the Two Zebrafish Patched Homologues Identifies Novel Roles for the Hedgehog Signaling Pathway by Marco J Koudijs; Marjo J den Broeder; Evelyn Groot; Fredericus J M van Eeden is a Biochemistry, Genetics and Molecular Biology article available to read on EtoBox.

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## Background Aberrant activation of the Hedgehog (Hh) signaling pathway in different organisms has shown the importance of this family of morphogens during development. Genetic screens in zebrafish have assigned specific roles for Hh in proliferation, differentiation and patterning, but mainly as a result of a loss of its activity. We attempted to fully activate the Hh pathway by removing both receptors for the Hh proteins, called Patched1 and 2, which are functioning as negative regulators in this pathway. ## Results Here we describe a splice-donor mutation in Ptc1, called __ptc1__^__hu1602__^, which in a homozygous state results in a subtle eye and somite phenotype. Since we recently positionally cloned a __ptc2__ mutant, a __ptc1;ptc2__ double mutant was generated, showing severely increased levels of __ptc1__, __gli1__ and __nkx2.2a__, confirming an aberrant activation of Hh signaling. As a consequence, a number of phenotypes were observed that have not been reported previously using __Shh__ mRNA overexpression. Somites of __ptc1;ptc2__ double mutants do not express anteroposterior polarity markers, however initial segmentation of the somites itself is not affected. This is th

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Author

Marco J Koudijs; Marjo J den Broeder; Evelyn Groot; Fredericus J M van Eeden

Publisher

BioMed Central; Springer (Biomed Central Ltd.); London: BioMed Central, 2001-; Springer Science and Business Media LLC; Society for Mining, Metallurgy and Exploration Inc. (ISSN 1471-213X)

Published

2008

Language

EN

Field

Biochemistry, Genetics and Molecular Biology (Life Sciences)