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This article discusses a rare case of bilateral open lipped schizencephaly in a 23-year-old pregnant woman. The ultrasound and MRI findings revealed the absence of the cavum septum pellucidum, dilatation of the lateral ventricle, abnormal Sylvian fissure operculisation, and the presence of CSF-filled bilateral clefts extending from the subarachnoid spaces to the cerebral cortex. Due to the poor neurological outcome, the pregnancy was terminated. Genetic workup was done, and the results are pending. The article highlights the association of this disorder with genetic mutations. [Extracted from the article]