The hydromyelia is one kind of four types of syringomyelia, chronic progressive and degenerative disorder, which develops pathological cavity or syrinx within spinal cord. It is rare and comprises dilated spinal syrinx. Its clinical manifestations include muscle weakness without pain in arm and hand, dissociated segmental dysesthesia, and loss of pain and temperature sensation. It is also frequently associated with hydrocephalus. We report a case of hydromyelia with erythematous depressed patch on low back in term neonate.