Sclerosing bone disorders encompass a range of genetic and acquired diseases. The potential for bone metastases is often a significant concern, especially when multiple discrete lesions are present. Several non-malignant disorders can also produce similar patterns of bone abnormalities. Among these is osteomesopyknosis, a rare condition characterized by multiple sclerotic lesions in the axial skeleton. Only a few cases of this presumably genetic disease have been documented. In this report, we present a new case and review previously published cases to enhance understanding and facilitate recognition of this disorder within the broader category of sclerosing bone diseases.