The Pediatric Pain Questionnaire and relevant medical records were used to investigate the frequency, intensity, location, and duration of pain experienced by children with spina bifida and to examine the concordance between child self-report and parent proxy report of pain. There were 68 children with spina bifida (30 males, 38 females) between the ages of 8 and 19 years (mean age 12y 8m). Fifty-nine children (87%) were diagnosed with myelomeningocele, six (9%) with lipomyelomeningocele, and three (4%) with lipomeningocele (4%). Forty-nine children (72%) had shunted hydrocephalus. Twenty-six children (38%) ambulated full-time. Fifty-six per cent of children reported experiencing pain once a week or more often. For these children, pain occurred most frequently in the head, back, abdomen, neck, shoulders, legs, and hands. Of the 49 children with shunted hydrocephalus, 43 (88%) reported headaches; 15 of 19 children (79%) without hydrocephalus reported headaches. Children who reported more intense pain also reported more frequent pain and more pain locations. Parents were most reliable at reporting locations of their children's severe pain. Parents of younger children significantly underestimated their children's current pain and worst pain in the past 7 days. Lesion level and ambulatory status were not associated with any of the pain variables. It was concluded that children with spina bifida frequently report clinically significant, yet under-recognized and untreated pain.