Visceral hyperalgesia refers to increased pain sensation in response to gastrointestinal sensory stimulus. In neonates with neurological impairments, gabapentin has been successfully used as a treatment for visceral hyperalgesia in neonates. The authors describe a preterm infant with myelomeningocele and persistent neuropathic pain that manifested as irritability, hypertonicity, poor weight gain, and feeding intolerance. After exclusion of other etiologies, the diagnosis of visceral hyperalgesia was suspected and the infant was treated with gabapentin. Following appropriate titration to effect and close monitoring of side effects of gabapentin, he subsequently demonstrated improved tone, decreased irritability with feedings, and appropriate weight gain. In addition, the authors provide a review of the available literature of gabapentin use in neonates and offer suggestions on when to consider starting gabapentin in a neonate with neurological impairment and chronic unexplained gastrointestinal manifestations.