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A case-control study of Wolf -Hirschhorn syndrome

Sharon Elizabeth Goodrich, University of Massachusetts Amherst


This exploratory case-control study used questionnaire data to determine risk factors in parents related to having a child with Wolf-Hirschhorn syndrome (WHS). The study included information on 181 case mothers, 174 case fathers, 135 control mothers, and 128 control fathers. When demographic, lifestyle, and reproductive risk factors were considered, case mothers were less likely than control mothers to consume alcohol a few times a month or more in the year preconception (crude POR = 0.53, 95% CI: 0.29–0.96; adjusted POR = 0.57, 95% CI: 0.31–1.06). Also, case parents were more likely than control parents to have this pregnancy reported as unplanned (crude POR = 1.75, 95% CI: 1.10–2.79; adjusted POR = 1.47, 95% CI: 0.88–2.45), and case mothers were less likely than control mothers to have ever taken birth control pills preconception (crude POR = 0.58, 95% CI: 0.34–0.99; adjusted POR = 0.66, 95% CI: 0.38–1.15). When preconception exposure to x-rays was considered, it was found that case fathers were more likely than control fathers to have had at least one non-dental head x-ray ever preconception (adjusted POR = 2.06, 95% CI: 1.11–3.84). When number of x-rays was considered by site, case fathers were more likely than control fathers to have had three or more non-dental head x-rays preconception (adjusted POR = 5.54, 95% CI: 1.21–22.45), and they were also more likely to have had three or more abdominal x-rays preconception (adjusted POR = 8.24, 95% CI: 1.02–66.32). Case mothers were less likely than control mothers to have had preconception dental x-rays once every couple of years (adjusted POR = 0.32, 95% CI: 0.14–0.73) or once per year or more (adjusted POR = 0.31, 95% CI: 0.14–0.71). In this study of WHS only 43% of eligible case families referred a friend or neighbor family for use as a control. Subsequent analyses revealed significant differences between case families that did and did not have a matched control family in the study. The main variables affecting this were education at conception, race, and months married at conception. Case families with mothers who had graduated college at conception, white mothers, white fathers, and parents married 25 months or more at conception were more likely to have a friend or neighbor control family in the study.

Subject Area

Public health

Recommended Citation

Goodrich, Sharon Elizabeth, "A case-control study of Wolf -Hirschhorn syndrome" (2002). Doctoral Dissertations Available from Proquest. AAI3068560.