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We report a 15-year-old boy who suffered from calvarial sclerosing osteomyelitis and presented with painful head swelling. X-rays of the skull revealed areas of irregular radiolucency. MR imaging and CT showed a well-demarcated intradiploic lesion with thickening of the skull extending from the frontal to the parietal calvarium with a low signal on T1-weighted images, strong but heterogeneous enhancement after gadolinium application and a mixed signal on T2-weighted images. Computer-navigated neurosurgery was planned, and the craniotomy defect was reconstructed by a preformed titanium implant. Sclerosing osteomyelitis of the calvarium has to be included in the differential diagnosis of osteolytic and sclerosing lesions of the skull coinciding with persistent swelling of the head.