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This study aimed to identify the sexual and reproductive health knowledge, attitudes, and behaviour of young people, aged between 14 and 23 years, with spina bifida who had attended the Spina Bifida Clinic at a tertiary centre in Melbourne, Australia within the past 10 years. Subjects participated in a structured interview and their parents completed a written questionnaire. Fifty-one (55%) of 93 eligible young people and 69 of 97 parents (71%) participated. Most young people were satisfied with the amount of general sex education they had received. However, 95% stated they had inadequate knowledge about sexual and reproductive health relating to spina bifida and 59% of parents considered they had inadequate knowledge. Thirty-nine percent of young people and 30% of parents had discussed sexuality issues with a doctor. However, 93% of young people and 100% of parents said that they would definitely talk about these issues if the discussions were initiated by their doctor. A significant degree of sexual intimacy was reported, with 60% reporting an intimate relationship, and 25% (10 females, three males) reporting sexual intercourse. Thirty-seven percent of females had experienced unwanted sexual attention, and 30% reported unwanted sexual touching. This study suggests that health professionals should pay greater attention to sexual and reproductive health issues in young people with spina bifida.