The variant in Beukes hip dysplasia reduces UFSP2’s in vitro catalytic activity, although its effects on the levels of UFMylated proteins in cells is unknown.12 Understanding the pathophysiology of these UFSP2-related diseases will require a more comprehensive assessment of how each variant alters the UFMylated proteome in relevant tissues, and how these changes impact the function of UFMylated proteins. Here, UFSP2 is linked to Hip dysplasia.