Historically, research in our laboratory has focused on centrioles and centrosomes, but given the recent explosion of interest in cilia and the potential advantages of Drosophila, we are now trying to study cilia assembly in living flies. Much of the expertise and many of the reagents we have acquired over the years studying centrioles and centrosomes will be directly applicable to these studies.

Several genetic human syndromes (such as Bardet-Biedel, Joubert and Meckel-Gruber syndromes) have been linked to cilia malfunction, and many of the genes associated with these syndromes encode ciliary proteins. Several of these proteins form complexes, but it is unclear how these proteins work together to form a functional cilium. Homologues of many of these proteins exist in flies, and we have recently developed an assay to follow cilium assembly live in the well-studied Sensory Organ Precursor (SOP) cells of the pupal notum. SOPs go through a stereo-typical pattern of divisions to generate a cluster of four cells—one ciliated sensory neuron and three non-ciliated support cells—so we know which cell will form a primary cilium and when it will do so (Roque et al., Plos Genetics, 2018). We can now follow the centrioles in these cells as they proceed through their last division, disassemble their PCM, migrate to the cell cortex, and nucleate cilium assembly. We are currently using CRISPR technology to make mutants in all the Drosophila genes that are homologues of the human ciliopathy genes (Pratt et al., JCS, 2016), and we will use our live-cell assay to study centriole, centrosome and cilium behaviour in these mutants.