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Trakya Üniversitesi Tıp Fakültesi Nöroşirürji Anabilim Dalı, Edirne Schisencephaly is a developmental anomaly characterized by a defect of neuronal migration and cortical lamellation of the brain before the sixth week of the embryonic period. A 3-year-old patient with spastic hemiparesis due to a large right parietal schisencephaly extending to the lateral ventricle, and associated hydrocephalus is reported. The patient underwent cystoventricular anastomosis and ventriculocystoperitoneal shunt procedures. Significant improvement in spastic hemiparesis was observed in the postoperative period. Schisencephalic cavities open to the ventricles may lead to cystic dilatation, hydrocephalus, and subsequent mass effect, compromising cerebrospinal fluid circulation. Anahtar Kelimeler : Developmental disorder, hydrocephalus, schisencephaly, spasticity