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Department of Neurosurgery, Ankara Numune Hospital, Ankara, Turkey One to two of all Echinococcus granulosus infections involve the brain, and the finding of a hydatid cyst in the aqueduct of Sylvius is extremely rare. We describe the case of a 10-year-old girl with a symptomatic hydatid cyst in the aqueduct of Sylvius. The patient exhibited signs of elevated intracranial pressure and also had left hemiparesis, ataxia and cerebellar signs. Neuroradiological imaging revealed obstructive hydrocephalus and a cystic lesion in the aqueduct of Sylvius. Magnetic resonance imaging (MRI) was used to evaluate the patient pre- and postoperatively, and surgery was carried out via a suboccipital approach using Dowling's technique. Multiple cysts were removed intact without rupture, and the patient survived and recovered fully. This is the first case with a lesion at this location to be documented by MRI, and only the second of its kind in the literature. MRI is important in terms of diagnosing and planning surgical treatment for these cysts. Total removal without rupture should be the surgical goal in all cases. Keywords : Aqueduct of Sylvius, cerebral hydatid cyst, magnetic resonance imaging