The article published by Mbalaviele and colleagues (PLoS One, 2012, 7:e35979) on the development of a mouse model of neonatal-onset multisystem inflammatory disease (NOMID) is among the top 25% most cited PLOS ONE articles as of July 3, 2017. NOMID, which is caused by NLRP3 activating-mutations, is associated with excessive IL-1β secretion and skeletal deformities, including bony outgrowths and disorganized growth plate. IL-1 blocking drugs are efficacious in resolving NOMID-associated inflammatory symptoms, but not bony outgrowths in patients. In a follow-up study (Scientific Reports, 2017, July 7;7(1)), Mbalaviele and collaborators prevented skeletal manifestations in NOMID mice by deleting IL-1 receptor during embryogenesis. These findings have clinical implications as they suggest that early diagnosis and therapeutic intervention with IL-1 drugs prior to the onset of skeletal lesions may prevent the development of these devastating complications in humans.