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Background: Tumor markers are widely applied in clinical practice,however,few serum markers have been found for intracranial tumors.Herein,we firstly report an intracranial epidermoid cyst case with extremely high level of serum CA 199.Furthermore,the relationship between CA 199 level and intracranial epidermoid cyst was closely followed for a long period.Case presentation: We report a case of 41-year-old man with a history of 2-months headache and a sudden exacerbation for 3 days.Radiology examination suggested multiple lesions spread along ventricular system.Laboratory tests showed exceeding increase of serum CA 199.The patient underwent craniotomy and continuous lumber drainage.Post-operative pathology proved a ruptured intracranial epidermoid cyst.MRI scans and serum CA 199 were closely followed up for three years.