Background: Skin dimples at specific sites of the body at birth are a clue to underlying congenital malformation and intimate the need for extensive evaluation. A midline dimple in the spinal region may be a sign of occult spinal dysraphism in a neonate. Methods: A toddler presented with clinical symptoms and signs of acute paraparesis. The child also had thoracic dermal sinus on clinical examination. The child was evaluated with hematological and radiological investigations to diagnose the cause of acute paraparesis. Results: Hematological investigations were normal. Magnetic resonance imaging of the spine showed an intramedullary epidermoid cyst at the D1 vertebral level to D4 vertebral level with dorsal dermal sinus at the D3 vertebral level. Conclusion: We are reporting this case to insist on clinical examination of all neonates for skin dimples and detailed evaluation of neonates with suspicious skin dimples to prevent future neurological disability. A clinically significant skin dimple implies impending neurological problems in a patient.
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