Nephrologie Pediatrique, Hôpital Robert Debré AP-HP, Paris, France.
Pediatr Res. 2010 Mar;67(3):300-3. doi: 10.1203/PDR.0b013e3181ca038d.
Bartter syndrome is an autosomic recessive disease characterized by severe polyuria and sodium renal loss. The responsible genes encode proteins involved in electrolyte tubular reabsorption. Prenatal manifestations, mainly recurrent polyhydramnios because of fetal polyuria, lead to premature delivery. After birth, polyuria leads to life-threatening dehydration. Prenatal genetic diagnosis needs an index case. The aim of this study was to analyze amniotic fluid biochemistry for the prediction of Bartter syndrome. We retrospectively studied 16 amniotic fluids of Bartter syndrome-affected fetuses diagnosed after birth, only six of them being genetically proven. We assayed total proteins, alpha-fetoprotein, and electrolytes and defined a Bartter index corresponding to the multiplication of total protein and of alpha-fetoprotein. Results were compared with two control groups matched for gestational age-non-Bartter polyhydramnios (n = 30) and nonpolyhydramnios (n = 60). In Bartter syndrome, we observed significant differences (p < 0.0001) for protein amniotic fluid levels when compared with the two control groups (1.55 g/L, 3.9 g/L, and 5.2 g/L, respectively) and low Bartter index (0.16, 0.82, and 1.0, respectively). No statistical difference was observed for electrolytes. In conclusion, Bartter syndrome can be prenatally suspected on amniotic fluid biochemistry (sensitivity 93% and specificity 100%), allowing appropriate management before and after birth.
巴特综合征是一种常染色体隐性遗传病,其特征为严重的多尿和钠肾脏丢失。相关基因编码参与电解质管状重吸收的蛋白。产前表现主要为胎儿多尿导致的复发性羊水过多,从而引起早产。出生后,多尿会导致危及生命的脱水。产前基因诊断需要一个索引病例。本研究旨在通过分析羊水生化指标来预测巴特综合征。我们回顾性研究了 16 例出生后被诊断为巴特综合征的羊水,其中仅 6 例经基因证实。我们检测了总蛋白、甲胎蛋白和电解质,并定义了巴特指数,即总蛋白和甲胎蛋白的乘积。结果与两个对照组进行了比较,分别为胎龄匹配的非巴特氏羊水过多组(n = 30)和非羊水过多组(n = 60)。在巴特综合征中,与两个对照组相比,羊水的蛋白水平存在显著差异(p < 0.0001)(分别为 1.55 g/L、3.9 g/L 和 5.2 g/L),巴特指数也较低(分别为 0.16、0.82 和 1.0)。电解质无统计学差异。总之,羊水生化指标可用于产前怀疑巴特综合征(灵敏度为 93%,特异性为 100%),以便在出生前后进行适当的管理。