Cilia,as microtubule-based subcellular structure,widely present on the surface of nearly all ver-tebrate cells and play crucial roles in various biological processes such as cell motility and signal transduction.A series of genetic diseases caused by abnormalities in the structure or function of cilia are collectively referred to as ciliopathies.Due to the high evolutionary conservation of cilia and the similarity between zebrafish and humans in multiple specialized ciliary types,zebrafish has emerged as an ideal model organism for studying ciliary functions and ciliopathies.This paper summarizes the structural features of zebrafish cilia,sorts out the advances in common-ly used related novel techniques,and describes the important findings and contributions of the zebrafish model in the study of human ciliopathy.Studies have shown that zebrafish,with its unique biological development character-istics and highly similar ciliary structure to that of humans,has greatly contributed to the research progress in cilia-related fields and provided important clues for a deeper understanding of the pathogenesis of ciliopathies.