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DYX1C1,  a susceptibility gene for dyslexia, encodes a tetratricopeptide repeat domain containing protein that has been implicated in neuronal migration in rodent models. The developmental role of this gene remains unexplored. To understand the biological function(s) of zebrafish  dyx1c1  during embryonic development, we cloned the zebrafish  dyx1c1  and used morpholino-based knockdown strategy. Quantitative real-time PCR analysis revealed the presence of  dyx1c1  transcripts in embryos, early larval stages and in a wide range of adult tissues. Using mRNA in situ hybridization, we show here that  dyx1c1  is expressed in many ciliated tissues in zebrafish. Inhibition of  dyx1c1  produced pleiotropic phenotypes characteristically associated with cilia defects such as body curvature, hydrocephalus, situs inversus and kidney cysts. We also demonstrate that in  dyx1c1  morphants, cilia length is reduced in several organs including Kupffer’s vesicle, pronephros, spinal canal and olfactory placode. Furthermore, electron microscopic analysis of cilia in  dyx1c1  morphants revealed loss of both outer (ODA) and inner dynein arms (IDA) that have been shown to be required for cilia motility. Considering all these results, we propose an essential role for  dyx1c1  in cilia growth and function.

The Zebrafish Orthologue of the Dyslexia Candidate Gene DYX1C1 Is Essential for Cilia Growth and Function