Cronin O, Subedi D, Forsyth L, et al. - Since mutations in SQSTM1 are strongly correlated with Paget disease of bone (PDB), however little is known about the clinical features of those with early disease, researchers analyzed radionuclide bone scans, biochemical markers of bone turnover, and clinical characteristics in SQSTM1 mutation carriers who participated in the Zoledronic acid in the Prevention of Paget's disease (ZiPP) study. Participants in the study were 222 individuals [54.9% were female, mean ± SE age of 50.1 ± 0.6 years]. The ZiPP study established the significance of SQSTM1 mutations as a genetic risk factor for PDB, and showed that in the target population a programme of genetic testing coupled with therapeutic intervention is both appropriate and feasible. Further follow‐up of this cohort should provide valuable details about the natural history of early PDB and its treatment response.