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Aicardi's syndrome is a severe congenital syndrome characterized by infantile spasm, corpus callosum agenesis, ocular abnormalities, especially chorioretinal lacuna, and motor mental retardation. The diagnosis of the disease is made by clinical findings. Electroencephalography, magnetic resonance imaging findings and ophthalmologic examination are helpful in the diagnosis. The first problem in patients with Aicardi syndrome is seizure and motormental retardation. In this study, two siblings who were born as a result of consanguineous marriages and who were diagnosed Aicardi syndrome with mental-motor retardation, ocular abnormalities, corpus callosum hypoplasia and interventricular cyst were presented. [ABSTRACT FROM AUTHOR]