Lissencephaly–pachygyria complex or spectrum is a rare malformation of cortical development characterised by neuronal migration defect and abnormal formation of cerebral gyri or convolutions . The main features of lissencephaly are abnormally thick cortex along with absent (agyria) or abnormally developed (pachygyria) cerebral convolutions making the brain appear smooth. We present a case of nine-year-old girl with lissencephaly-pachygyria complex with hypoxic ischemic encephalopathy. She presented with severe intellectual disability, seizures, autistic features with marked sensory issues, self-stimulating behaviors, hyperactivity and aggression. We describe the clinical manifestations and highlight the role of non-pharmacological interventions that include family psycho-education, sensory and occupational therapy, functional behavior analysis and therapy, and addressing caregiver burn-out.