We present a case report of melorheostosis with the novel radiographic finding of underlying cortical resorption. A number of radiographic patterns of melorheostosis have been described; however, the combination of new bone formation and resorption of the original cortex appears unique. Although the presence of underlying lysis has been postulated in published studies, direct radiographic evidence of bony resorption in melorheostosis has not been reported. These findings can be subtle and might go unnoticed using standard imaging. An in-depth review of the radiographic features is presented, including multimodality imaging with magnetic resonance imaging and computed tomography.