We herein report the case of a 21-day-old boy in which the detection of an intramedullary spinal cord abscess led to the diagnosis of Currarino syndrome (CS). He had a complete phenotype of CS, including sacral agenesis, an anorectal malformation, a presacral mass, and spinal cord malformations. In addition, he had an intramedullary spinal cord abscess. Intramedullary spinal cord abscess is rare in CS and is thought to require immediate intervention. Therefore, we additionally reviewed the

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... able literature and discussed the therapeutic approach for CS with an intramedullary cord abscess.