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Spinal intramedullary hamartoma with acute presentation in a 13-month old infant: case report. True hamartomas of the spinal cord are very rare, and although several have been reported in the literature, there are few detailed radiological and pathological descriptions of the condition. There is also considerable overlap with other entities, the most common being spinal cord teratomas. The authors report the case of a 13-month-old child with a supragluteal sacral dimple who presented with acute neurological deterioration. MRI of the spine revealed a big intramedullary lesion with heterogeneous signal intensity. A near-total resection was performed, and histopathological examination demonstrated findings consistent with a spinal cord hamartoma. The authors believe that careful preoperative evaluation and rigorous pathological examination are mandatory to establish diagnosis and direct further management of cases in which such a lesion is suspected.