The last two decades have witnessed the invention and development of super-resolution microscopy (SRM) that breaks the diffraction limit of light and pushes the fluorescence microscopy resolution to several nanometers. While SRM is widely used in biological studies, such as resolving subdiffraction structures, molecular mapping, tracking single molecules, probing protein-protein interactions, and observing organelle dynamics, its direct application in translational medicine, such as disease diagnosis, is still preliminary. Despite their small size, cilia play a crucial role as organelles in cell signaling and motility, with defects in cilia leading to ciliopathy. Similar to other miniature organelles and macromolecular complexes, cilia are ideal for super-resolution imaging. In this review, we will 1) introduce cilia and ciliopathy, 2) show how SRM extends our knowledge of cilia, and 3) focus on how SRM improves the diagnosis of motile ciliopathies.

The last two decades have witnessed the invention and development of super-resolution microscopy (SRM) that breaks the diffraction limit of light and pushes the fluorescence microscopy resolution to several nanometers. While SRM is widely used in biological studies, such as resolving subdiffraction structures, molecular mapping, tracking single molecules, probing protein-protein interactions, and observing organelle dynamics, its direct application in translational medicine, such as disease diagnosis, is still preliminary. Despite their small size, cilia play a crucial role as organelles in cell signaling and motility, with defects in cilia leading to ciliopathy. Similar to other miniature organelles and macromolecular complexes, cilia are ideal for super-resolution imaging. In this review, we will 1) introduce cilia and ciliopathy, 2) show how SRM extends our knowledge of cilia, and 3) focus on how SRM improves the diagnosis of motile ciliopathies.