The combined anabolic and antiresorptive effect of 4PBA and ALN, respectively, as well as the response to single treatments (4PBA-only and ALN-only) were evaluated in both dominant and recessive forms of OI by exploiting larvae and adult Chihuahua (Chi/+)7 and p3h1−/− knockout9 zebrafish models, respectively. This evidence concerns the gene P3H1 and osteogenesis imperfecta.