As previously described, histological review of skin samples from patients with MAP2K1‐positive melorheostosis showed thickened vascular wall and increased density of superficial vasculature in skin overlying affected bone in comparison to skin from the contralateral extremity.16 This pattern was not noted in skin samples from patients with MAP2K1‐negative melorheostosis. Here, MAP2K1 is linked to melorheostosis.