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Abstract Background. Dissemination of ovarian cancer occurs mainly within the peritoneal cavity and central nervous system involvement (CNS) is encountered rarely. This report describes an unusual case with iatrogenic leptomeningeal metastasis from ovarian carcinoma. Case. 36 year-old lady diagnosed with ovarian carcinoma develops neurologic symptoms 10 months following primary treatment. Medical history reveals a venticuloperitoneal shunt placement 16 years ago for non-communicating hydrocephalus. She undergoes surgery for obstruction of the VP catheter and pathologic evaluation reveals metastatic carcinoma consistent with the primary diagnosis. With subsequent systemic chemotherapy, the patient remains alive for 2 years. Conclusion. This report describes a unique patient with ovarian cancer, who developed iatrogenic leptomeningeal involvement of the lateral ventricule through an indwelling ventriculoperitoneal shunt.