Tuberous sclerosis is a neurocutaneous autosomal-dominant disorder, presents with highly variable clinical manifestation and affects multiple organ system. Its manifestation in neonate is poorly described. We report a case of intractable neonatal seizure with multiple hypopigmented patch. Neuroimaging of brain shows classical cortical tubers and subependymal nodule suggestive of tuberous sclerosis in newborn. Hence tuberous sclerosis should be considered as an etiology of neonatal seizure and neuroimaging should be performed when other common causes of neonatal seizure have been excluded.
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