Establishment of the Early Cilia Preassembly Protein Complex
Establishment of the early cilia preassembly protein PNAS PLUS complex during motile ciliogenesis Amjad Horania,1, Alessandro Ustioneb, Tao Huangc, Amy L. Firthd, Jiehong Panc, Sean P. Gunstenc, Jeffrey A. Haspelc, David W. Pistonb, and Steven L. Brodyc aDepartment of Pediatrics, Washington University School of Medicine, St. Louis, MO 63110; bDepartment of Cell Biology and Physiology, Washington University School of Medicine, St. Louis, MO 63110; cDepartment of Medicine, Washington University School of Medicine, St. Louis, MO 63110; and dDepartment of Medicine, University of Southern California, Keck School of Medicine, Los Angeles, CA 90033 Edited by Kathryn V. Anderson, Sloan Kettering Institute, New York, NY, and approved December 27, 2017 (received for review September 9, 2017) Motile cilia are characterized by dynein motor units, which preas- function of these proteins is unknown; however, missing dynein semble in the cytoplasm before trafficking into the cilia. Proteins motor complexes in the cilia of mutants and cytoplasmic locali- required for dynein preassembly were discovered by finding human zation (or absence in the cilia proteome) suggest a role in the mutations that result in absent ciliary motors, but little is known preassembly of dynein motor complexes. Studies in C. reinhardtii about their expression, function, or interactions. By monitoring show motor components in the cell body before transport to ciliogenesis in primary airway epithelial cells and MCIDAS-regulated flagella (22–25). However, the expression, interactions, and induced pluripotent stem cells, we uncovered two phases of expres- functions of preassembly proteins, as well as the steps required sion of preassembly proteins. An early phase, composed of HEATR2, for preassembly, are undefined.
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