Parental concern can play an important role in overall management of children with serious chronic illness. We quantitatively assessed the concerns of parents of children with hydrocephalus, using the Hydrocephalus Concerns Questionnaire for parents (HCQ-P). Over a 12-month study period, parents of 332 children with hydrocephalus (mean age 11.7 years, SD 3.8) attending a routinely scheduled out-patient clinic at the Hospital for Sick Children, Toronto, completed the HCQ-P. HCQ-P scores were widely distributed, with mean maternal (N = 283) and paternal (N = 137) scores of 0.53 (SD 0.30) and 0.51 (SD 0.29), respectively (0 = least concerned, 1 = most concerned). Based on a multivariable regression model (adjusted R (2) = 0.49), important associations with greater maternal concern were: Worse child cognitive health (p < 0.001) and worse child social-emotional health (p < 0.001). Identified important associations with greater paternal concern based on a multivariable model (adjusted R (2) = 0.45) were: Worse child cognitive health (p = 0.03), worse child social-emotional health (p = 0.03), frequent child seizures (p < 0.001), younger child age at first surgery (p = 0.03) and having had an endoscopic procedure for the child's hydrocephalus treatment (p = 0.04). Based on the adjusted multivariable models, less than 5% of parents were considered to have extreme levels of concern (either very high or very low). This study confirms that parental concern is highly variable in this population. Much of the parental concern can be appropriately explained by child health factors.