BackgroundPaternal occupational exposures to potential health hazards are likely to affect congenital malformations through the spermatogenesis cycle.PurposeThe aim of this case–control study was to assess the relationship between the risk of musculoskeletal congenital malformations in offspring and paternal workplace exposure to potential health hazards during the preconception period.MethodThe study comprised 105 patients (cases) with a musculoskeletal congenital malformation(s) and 135 controls matched for age and demographic characteristics. Both parents of each case and control were interviewed in the hospital by a trained physician. They also completed a questionnaire focusing on the preconception period and on the 3-month period immediately before and after the pregnancy conception date, respectively, of the child under study.ResultsThe odds of having a child with a congenital malformation was higher (P < 0.05) if the father was occupationally exposed to pesticides, solvents, or welding fumes during the preconception period.ConclusionControl of workplace exposures and adherence to threshold limit values of these potential health hazards should be adopted to minimize the risk of fathers having offspring with a congenital malformation.