Abstract
Forward and reverse genetic screens represent powerful methods to uncover new components for any biological process, but suffer from gene cloning or throughput bottleneck issues, respectively. Here, we employ an innovative approach to gene discovery: we screened a C. elegans whole-genome sequenced multi-mutation library for our phenotype of interest, namely defective ciliated sensory neuron development, and performed the Sequence Kernel Association Test (SKAT) to rapidly identify genes exhibiting this phenotype. Our approach unveiled a novel gene, bgnt-1.1, that influences cilia length. Notably, the human orthologue B3GNT1/B4GAT1 is associated with Walker-Warburg syndrome, a suspected ciliary disorder.
Copyright
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