Background: Intrauterine parvovirus B19 infection is a well-known cause for hydrops fetalis and fetal demise. We present a case of isolated fetal obstructive hydrocephalus caused by an intrauterine parvovirus B19 infection. Case: A 23-year-old woman, gravida 3 para 2, was referred for fetal obstructive hydrocephalus. Although there were no signs of fetal anemia, increased maternal parvovirus B19-specific immunoglobulin G and immunoglobulin M raised the suspicion of fetal infection. This was confirmed by polymerase chain reaction in the amniotic fluid. Elective cesarean delivery was performed at 34 weeks of gestation, followed by craniotomy and placement of shunt. At the age of 20 months, the child's development was in accordance with his age, with the exception of gross motor function, which was moderately delayed. Conclusion: Evaluation of fetal obstructive hydrocephalus should include maternal serological tests for parvovirus B19.