The Notch-regulated ankyrin repeat protein is required for proper anterior-posterior somite patterning in mice.

Document Type

Article

Publication Date

4-1-2012

Institution/Department

Center for Molecular Medicine; Maine Medical Center Research Institute

Journal Title

Genesis (New York, N.Y. : 2000)

MeSH Headings

Animals, Ankyrin Repeat, Body Patterning, Female, Gene Expression Regulation, Developmental, Gene Targeting, Immunohistochemistry, In Situ Hybridization, Intracellular Signaling Peptides and Proteins, Male, Mesoderm, Mice, Mice, Knockout, Mutation, Phenotype, Proteins, Receptors, Notch, Signal Transduction, Somites

Abstract

The Notch-regulated ankyrin repeat protein (Nrarp) is a component of a negative feedback system that attenuates Notch pathway-mediated signaling. In vertebrates, the timing and spacing of formation of the mesodermal somites are controlled by a molecular oscillator termed the segmentation clock. Somites are also patterned along the rostral-caudal axis of the embryo. Here, we demonstrate that Nrarp-deficient embryos and mice exhibit genetic background-dependent defects of the axial skeleton. While progression of the segmentation clock occurred in Nrarp-deficient embryos, they exhibited altered rostrocaudal patterning of the somites. In Nrarp mutant embryos, the posterior somite compartment was expanded. These studies confirm an anticipated, but previously undocumented role for the Nrarp gene in vertebrate somite patterning and provide an example of the strong influence that genetic background plays on the phenotypes exhibited by mutant mice.

ISSN

1526-968X

First Page

366

Last Page

374

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