Abstract: Hu Xingrong, Zhao Huiyan, Deng Yanbin, Cui Xiannian, Huang Zhihua, Xiang Haibo, Zhang Lijun Department of Medical Imaging, The Central Hospital of Enshi Tujia and Miao Autonomous Prefecture, Affiliated to Hubei University for Nationalities, Enshi 445000, Hubei, China （Hu XR, Zhao HY, Deng YB, Huang ZH, Xiang HB, Zhang LJ）; Clinical Laboratory Center, The Central Hospital of Enshi Tujia and Miao Autonomous Prefecture, Affiliated to Hubei University for Nationalities, Enshi 445000, Hubei, China （Cui XN） Corresponding author: Hu Xingrong, Email: xrh_cct@126.com 【Abstract】 A 50-year-old woman presented with intermittent dull pain in the forehead and mild dizziness occasionally after her forehead was subjected to a mild bump accidentally 20 days prior to the presentation, and was diagnosed with angioneurotic headache in a local hospital. After the treatment with oral sibelium tablets, the condition wasn′t relieved obviously. Computed tomography （CT） scan showed multiple localized bone destruction and low-density area in the frontal and bilateral parietal bones with adjacent soft tissue swelling. Magnetic resonance imaging （MRI） revealed equal T1 signals and slightly long T2 signals for multiple nodules in the frontal and bilateral parietal bones, high signals on diffusion-weighted imaging （DWI）, obvious enhancement on contrast-enhanced MRI, and linear enhancement in adjacent meninges. Whole-body bone scintigraphy showed multiple increased radionuclide uptake in the skull. Laboratory examination demonstrated that specific antibodies to Treponema pallidum （Tp） were positive, and the serum rapid plasma reagin （RPR） titer was 1∶128. Cerebrospinal fluid （CSF） examination showed normal CSF pressure, nucleated cell counts （8 × 106/L） and glucose level （4.0 mmol/L）, slightly high chloride flux （129.1 mmol/L）, high protein level （0.9 g/L）, high CSF-RPR titer of 1∶16 and presence of specific antibodies to Tp. Histopathological examination revealed hyperemia of adjacent tissues in the cranial osteolytic area, hyperplasia of interstitial fibrous tissue, endothelial cell swelling, and infiltration of inflammatory cells mainly containing plasma cells. The treatment regimen for neurosyphilis was given, and headache was relieved after 1 week of treatment, basically disappeared after 2 weeks, and completely disappeared after 4 weeks, and no similar headache occurred thereafter. Finally, the patient was diagnosed with acquired syphilitic skull osteomyelitis complicated by syphilitic meningitis.