The finding of markedly increased ratio of RANKL/OPG transcripts in osteoblasts from affected bone in patients with MAP2K1‐positive melorheostosis raises the possibility of using RANKL inhibitors in these patients,21 although it is possible that the increased ratio of RANKL/OPG transcripts is actually a compensatory mechanism for the dense bone. Here, MAP2K1 is linked to melorheostosis.