Skeletal genetic diseases are characterized by disorders in bone development and growth throughout the body. Such diseases often present clinically with pathological manifestations such as head and limb deformities and scoliosis, which can seriously affect patient quality of life. The Nosology of genetic skeletal disorders: 2023 revision, recognizes 41 major categories, involving 552 genes; however, the pathogenic mechanisms of around half of all bone genetic diseases remain unclear. As a new type of model animal, zebrafish have a highly conserved skeletal development process and regulatory mechanism compared with mammals. They also have the advantages of small size, strong reproductive ability, short reproductive cycle, and transparent embryos, potentially making them suitable for studying the pathogenesis of human skeletal genetic diseases. This review focuses on progress in the application of zebrafish models in research related to human skeletal genetic diseases. We carried out an extensive literature review and selected nine major categories of skeletal genetic disorders for detailed discussion, including fibroblast growth factor receptor 3 chondrodysplasias, type 2 collagen disorders, and type 11 collagen disorders. This article summarizes the disease overview, zebrafish model construction, and their research significance, with the aim of providing a reference for in-depth research on the pathogenesis of human skeletal genetic diseases.