Skeletal genetic diseases are a type of genetic disease of the skeletal system, characterized by disorders in the development and growth of bones throughout the body. Clinically, they often present as abnormal pathological manifestations such as head and limb deformities, scoliosis, etc., which seriously affect the quality of life of patients. According to the Nosology of genetic skeletal disorders: 2023 revision, they can be divided into 41 major categories, involving 552 genes. The pathogenic mechanism of about half of bone genetic diseases has not been fully elucidated. As a new type of model animal, zebrafish has a highly conserved skeletal development process and regulatory mechanism compared to mammals. Zebrafish have the characteristics of small size, strong reproductive ability, short reproductive cycle, and transparent embryos, which provides the possibility for further using zebrafish models to study the pathogenesis of human skeletal genetic diseases. This review focuses on the application progress of zebrafish models in research related to human skeletal genetic diseases. Through an extensive literature review, we have selected nine major categories of skeletal genetic disorders for detailed discussion, including FGFR3 chondrodysplasias, type 2 collagen disorders, and type 11 collagen disorders, etc. This article summarizes the disease overview, zebrafish model construction, and their research significance, aiming to provide a reference for in-depth research on the pathogenesis of human skeletal genetic diseases.