An organism's awareness of its surroundings is dependent on sensory function. As antennas to our external environment, cilia are involved in fundamental biological processes such as olfaction, photoreception, and touch. The olfactory system has adapted this organelle for its unique sensory function and optimized it for detection of external stimuli. The elongated and tapering structure of olfactory cilia and their organization into an overlapping meshwork bathed by the nasal mucosa is optimized to enhance odor absorption and detection. As many as 15-30 nonmotile, sensory cilia on dendritic endings of single olfactory sensory neurons (OSNs) compartmentalize signaling molecules necessary for odor detection allowing for efficient and spatially confined responses to sensory stimuli. Although the loss of olfactory cilia or deletion of selected components of the olfactory signaling cascade leads to anosmia, the mechanisms of ciliogenesis and the selected enrichment of signaling molecules remain poorly understood. Much of our current knowledge is the result of elegant electron microscopy studies describing the structure and organization of the olfactory epithelium and cilia. New genetic and cell biological approaches, which compliment these early studies, show promise in elucidating the mechanisms of olfactory cilia assembly, maintenance, and compartmentalization. Importantly, emerging evidence suggests that olfactory dysfunction represents a previously unrecognized clinical manifestation of multiple ciliary disorders. Future work investigating the mechanisms of olfactory dysfunction combining both clinical studies with basic science research will provide us important new information regarding the pathogenesis of human sensory perception diseases.