In summary, this study demonstrates, for the first time, that PTHrP is a significant endogenous pathogenic factor in FD; it participates in initiating the cAMP/PKA/CREB signaling pathway and promotes the overactive Wnt/β-Catenin signaling pathway, leading to abnormal osteogenesis and proliferation in FD BMSCs. Here, PTHLH is linked to Fabry disease.