With the objective of identifying whether hypospadias in infants is associated with maternal use of oral contraceptives before pregnancy or in early pregnancy, 846 case-control pairs were collected from eight different malformation monitoring programs around the world and mothers were interviewed using structured questionnaires administered after the birth of the infants. There was no difference in the preconceptional use of oral contraceptives between cases and controls, neither with respect to the number of years of oral contraceptive usage nor the time between stopping oral contraceptives and the present pregnancy. To this material was added data on oral contraceptive usage in early pregnancy from two other sources: an ongoing case-control study in Spain (725 infants with hypospadias) and a population-based study in Sweden (631 infants with hypospadias). There was no statistically significant difference in oral contraceptive exposure in early pregnancy between cases and controls. There is no demonstrable association between oral contraceptive use and infant hypospadias.