FGF signaling pathways play essential roles in both endochondral and intramembranous bone development and in human genetic diseases affecting bone development. FGF signaling is crucial for early limb development and throughout skeletal development. This review examines the role of FGF signaling in bone development and human genetic diseases that affect bone development, as well as the interactions between FGF signaling and other major signaling pathways regulating chondrogenesis and osteogenesis. Skeletal elements are formed through two distinct processes: endochondral ossification, which forms long bones, facial bones, vertebrae, and lateral medial clavicles, and intramembranous ossification, which forms flat bones such as the cranium and medial clavicles. Endochondral ossification involves a cartilaginous template that regulates skeletal growth and patterning, while intramembranous ossification forms bone directly from mesenchymal cells. FGF signaling is essential for skeletal development, with mutations in FGF receptors causing various skeletal dysplasias and craniosynostosis syndromes. For example, mutations in FGFR3 cause Achondroplasia, the most common form of dwarfism, while mutations in FGFR2 are associated with several craniosynostosis syndromes. These mutations often result in constitutive or hyperactivation of the receptor, leading to abnormal bone development. FGF signaling pathways interact with other signaling pathways such as the Hedgehog, BMP, and PTHrP pathways to regulate chondrogenesis and osteogenesis. These pathways work together to coordinate the complex processes of bone formation and development. FGF signaling is also involved in the regulation of cell proliferation, differentiation, and apoptosis in the developing skeleton. In intramembranous bone formation, FGF signaling is important for the development of calvarial bones and the regulation of suture closure. FGF2 and FGF18 are particularly important in this process, with FGF2 promoting osteogenic cell proliferation and FGF18 playing a role in osteoblast differentiation. FGF signaling also influences the balance between undifferentiated and differentiated osteogenic cells, ensuring proper cranial vault morphogenesis. Overall, FGF signaling pathways are critical for the proper development of both endochondral and intramembranous bone, and their dysregulation can lead to a variety of skeletal disorders. Understanding the role of FGF signaling in bone development is essential for the treatment of genetic bone diseases.FGF signaling pathways play essential roles in both endochondral and intramembranous bone development and in human genetic diseases affecting bone development. FGF signaling is crucial for early limb development and throughout skeletal development. This review examines the role of FGF signaling in bone development and human genetic diseases that affect bone development, as well as the interactions between FGF signaling and other major signaling pathways regulating chondrogenesis and osteogenesis. Skeletal elements are formed through two distinct processes: endochondral ossification, which forms long bones, facial bones, vertebrae, and lateral medial clavicles, and intramembranous ossification, which forms flat bones such as the cranium and medial clavicles. Endochondral ossification involves a cartilaginous template that regulates skeletal growth and patterning, while intramembranous ossification forms bone directly from mesenchymal cells. FGF signaling is essential for skeletal development, with mutations in FGF receptors causing various skeletal dysplasias and craniosynostosis syndromes. For example, mutations in FGFR3 cause Achondroplasia, the most common form of dwarfism, while mutations in FGFR2 are associated with several craniosynostosis syndromes. These mutations often result in constitutive or hyperactivation of the receptor, leading to abnormal bone development. FGF signaling pathways interact with other signaling pathways such as the Hedgehog, BMP, and PTHrP pathways to regulate chondrogenesis and osteogenesis. These pathways work together to coordinate the complex processes of bone formation and development. FGF signaling is also involved in the regulation of cell proliferation, differentiation, and apoptosis in the developing skeleton. In intramembranous bone formation, FGF signaling is important for the development of calvarial bones and the regulation of suture closure. FGF2 and FGF18 are particularly important in this process, with FGF2 promoting osteogenic cell proliferation and FGF18 playing a role in osteoblast differentiation. FGF signaling also influences the balance between undifferentiated and differentiated osteogenic cells, ensuring proper cranial vault morphogenesis. Overall, FGF signaling pathways are critical for the proper development of both endochondral and intramembranous bone, and their dysregulation can lead to a variety of skeletal disorders. Understanding the role of FGF signaling in bone development is essential for the treatment of genetic bone diseases.