A conserved domain of Cfap298 governs left-right symmetry breaking in vertebrates.

ABSTRACT: Cfap298 is a highly conserved gene required for ciliary motility and dynein arm assembly, with known roles in left-right (LR) patterning in zebrafish and links to human ciliopathies. Here, we describe a Cfap298 mutant allele, Cfap298ΔΔS, which selectively disrupts LR axis establishment in mice. Mutant embryos display organ laterality defects and abnormal Nodal, Pitx2 and Lefty1 expression, consistent with an early disruption in LR symmetry breaking. LR asymmetry is established by leftward fluid flow in the node, generated by planar-polarized cilia. Although cfap298 mutations are reported to affect planar polarity, we did not observe changes in cilia position, length or CELSR1 localization within the node, suggesting that Cfap298ΔΔS functions at the level of cilia motility. Accordingly, cil

SUBMITTER: Cortez M 

PROVIDER: S-EPMC12633728 | biostudies-literature | 2025 Oct

REPOSITORIES: biostudies-literature