This study examined depression, self-esteem, and mastery in the family caretakers of a group of males with Duchenne muscular dystrophy in comparison to a control group. A questionnaire based on the National Population Health Survey from Statistics Canada, a survey to collect information on the health of the Canadian population and related sociodemographic information, was conducted by telephone with 42 parents. The results were compared with the national data from the National Population Health Survey (1994 and 1999), matched for province of residence, number of children in the household, age, and marital status of the respondents. Parents of children with Duchenne muscular dystrophy had a higher probability of going through a major depressive episode and had significantly lower self-esteem and mastery scores than the national control group. None of the variables investigated (age, intelligence quotient, and ambulatory status of child or sex, age, and marital status of parent) could predict the depressive episode, with two exceptions. Parents without a partner had lower scores on the mastery scale, and parents of males older than 13 years of age were more likely to experience distress that interfered with life. It is incumbent on those caring for patients with Duchenne muscular dystrophy to counsel families regarding their potential to suffer a major depressive episode and to advise on appropriate therapy.