This report describes an unusual case of biventricular involvement caused by an epidermoid cyst in a 60-year-old man who presented with symptoms such as headache, dizziness, nausea, vomiting and gait ataxia. Investigations for infectious diseases and metastatic neoplasms were negative. Magnetic resonance imaging (MRI) of the brain revealed an expansive intracranial lesion with a cystic appearance in the left lateral ventricle, as well as similar material in the fourth ventricle associated with hydrocephalus. Surgical removal of the fourth ventricle mass restored normal cerebrospinal fluid (CSF) drainage, resulting in significant resolution of the hydrocephalus and improvement of symptoms.
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