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Agenesis or hypogenesis of Corpus callosum with interhemispheric cyst is a rare entity. Origin of interhemispheric cyst is controversial. These cysts may be Arachnoid cyst, neuroepithelial cyst, or extension of lateral or third ventricles. Here we describe a case of Corpus callosal hypogenesis with an interhemispheric cyst. Associated aqueductal stenosis and schizencephaly is also present. Interhemispheric cyst is communicating with lateral ventricle and the cyst has MRI signal intensitydoi:10.5455/2320-6012.ijrms20150447 fatcat:v64lbu7yqfchfjddg5pjlvnp5u