Factors associated with preterm delivery in mothers of children with Beckwith-Wiedemann syndrome: A case cohort study from the BWS registry

Pregnancy that results in a child with Beckwith-Wiedemann syndrome (BWS) is associated with preterm delivery. Based on previous case series, we hypothesized that preterm delivery of a child with BWS was due to known risk factors for preterm delivery such as polyhydramnios and gestational hypertension. A case cohort study using the BWS Registry at Washington University School of Medicine was undertaken. Cases were pregnancies that resulted in the birth of a child with BWS, controls were pregnancies resulting in the birth of siblings without BWS. Univariate analyses of maternal complications and logistic regression to predict preterm delivery were used. Children with BWS (n = 304) were delivered preterm at a significantly higher rate than their siblings (n = 269) odds ratio 19.1 (95% CI 9.1-40.2). Polyhydramnios, gestational hypertension, and vaginal bleeding also occurred at high rates in the BWS group with odds ratios of 31.6 (95% CI 12.6-79.1), 2.4 (95% CI 1.4-4.1), and 3.9 (95% CI 2.3-6.4), respectively. In a multivariate logistic regression model within the BWS group, polyhydramnios, vaginal bleeding, and gestational hypertension were significant predictors of preterm delivery, odds ratios of 2.9 (95% CI 1.6-5.4), 2.6 (95% CI 1.3-5.0), and 5.3 (2.3-12.0), respectively. However, a significant proportion, 6.5% (18 of 277), of patients in the BWS group delivered preterm without known risk factors. Preterm delivery of a child with BWS is associated with an increased frequency of polyhydramnios, gestational hypertension, and vaginal bleeding in the mother. However, preterm delivery also occurs in the absence of these risk factors.