Data from: Cohesin protein Smc3 influences kinocilial structure and function

Cohesinopathies and ciliopathies are congenital disorders affecting
overlapping body systems. The extent to which these syndromes may be
linked remains largely untested. Recently, reduced expression of a cohesin
core subunit, Smc3, was found to result in abnormal otolith development in
zebrafish embryos. This finding suggests that Smc3 may contribute to
kinociliary development and function, which would represent a novel role
for Smc3. Using hair cells found in neuromasts of the posterior lateral
line, we found that Smc3 knockdown resulted in reduced kinociliary length.
To address the role of Smc3 in kinocilial function, we monitored neomycin
resistance of neuromasts (associated with several cilial gene mutants) and
FM1-43X uptake in hair cells (associated with mechanotransduction). We
found that Smc3 knockdown indeed led to neomycin resistance of the
posterior lateral line neuromasts, suggesting impaired kinocilium
function. However, neuromast hair cells did not have defects in FM1-43X
uptake. We further demonstrated that hair cell number is reduced within
neuromasts. This study suggests a significant influence of cohesin subunit
Smc3 in ciliary structure and function and provides a preliminary link
between cohesinopathy and ciliopathy etiologies.