In a population-based case-control study, we examined relations between maternal and paternal occupations and the risk of infant craniosynostosis. Cases were 212 children born to Colorado residents and diagnosed during 1986-1989 with radiographically confirmed synostosis of unknown etiology. Controls were 291 children randomly selected from state birth records and frequency matched to cases on month and year of birth. Trained staff conducted telephone interviews of mothers of case and control children. Information was obtained about each job held by either parent during the pregnancy, and any job that the father held during the 3 months before the last menstrual period before conception. Jobs were coded using 1980 Census occupation and industry codes. We found no strong associations for maternal occupations. Of paternal occupations, two groups were associated with moderately increased odds ratios after adjustment for maternal smoking and altitude: agriculture and forestry (odds ratio = 2.3; 95% confidence interval = 1.0-5.2), and mechanics and repairmen (odds ratio = 2.7; 95% confidence interval = 1.2-6.1). For both, the odds ratios were higher for males.