Nearly all cell types in mammals contain cilia, small rod-like or more elaborate structures that extend from the cell surface. Cilia house signaling proteins that allow the cell to sample their environment and respond appropriately. Mutations in ciliary genes alter the functions of a broad range of cell and tissue types, including sensory and central neurons, and underlie a collection of heterogeneous human disorders called ciliopathies. Here, I highlight the critical contributions of nearly three centuries of research in diverse organisms to our current knowledge of cilia function in sensory signaling and human disease.