Occipital condyle syndrome (OCS) is a rare cause of headache. This study herein reports a case in which a unique headache and tongue deviation appear as symptoms of the first presentation of a malignant tumor. A healthy 67-year-old male presented with a unilateral shooting pain in the occipital region, accompanied by slurred speech and difficulty swallowing. Neurological examinations later revealed atrophy and mild fasciculation of the tongue. The clinical symptoms and MRI results suggested

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... Screening for tumor markers showed an elevated CEA. The chest CT revealed a lobulated soft-tissue mass in the lower left lobe, and a CTguided biopsy confirmed the diagnosis of adenocarcinoma. A whole body bone scan found multiple foci. The adenocarcinoma was graded pT2bN3M1b, stage IV. The headache improved with a prescription of prednisone, 60 mg to be taken daily. With three months of treatment, clinical examinations showed that the patient was free of pain and that there had been no progression of the atrophy or deviation of the tongue. The possible etiology of OCS includes a primary tumor or metastatic lesion that directly invades the base of the skull. Determining the underlying causes of OCS can be challenging, but MR imaging is currently the diagnostic tool of choice. An awareness of the features of OCS in healthy adults may be able to lead to earlier diagnosis of the underlying etiology and efficient relief of the symptoms.