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We report a case of a one-year-old boy with a congenital dermal sinus situated at the lateral buttock. The exact pathway of the sinus was shown by magnetic resonance imaging, contrast medium examination and computed tomographic examination. The sinus was not connected to the spinal canal and the sinus ended near the anterior surface of the sacral bone in a blind manner. Complete surgical resection of the sinus was achieved after these imaging examinations.