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Melorheostosis is an uncommon mesenchymal dysplasia that rarely affects the axial skeleton.We describe two atypical cases of melorheostosis with classical imaging findings - the first one involving the cervico-dorsal spine with encroachment of left vertebral artery canal causing attenuation of the left vertebral artery and the second one of mixed sclerosing bony dysplasia (monomelic involvement coexisting with osteopoikilosis).