BACKGROUND : Melorheostosis is a bone hyperostosis disorder characterized by flowing bone thought to occur in a sclerodermal distribution. These bony lesions often arise in soft tissues adjacent to joints. Pain arises from joint limitations and impingements, but diffuse pain can also occur. METHODS : We present a case of a patient with symptomatic compression of the common peroneal nerve by an extraosseous hyperostotic tissue island in a patient with melorheostosis. CONCLUSIONS : Melorheostosis is a rare bone overgrowth syndrome that can lead to joint mobility limitations and pain. In specific locations, it can also lead to tethering and symptomatic compression of the peripheral nerves.