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基于不同严重程度的下尿路梗阻对婴儿神经发育结局的影响。

Neurodevelopmental Outcome in Infants with Lower Urinary Tract Obstruction Based on Different Degrees of Severity.

机构信息

Department of Developmental Pediatrics, Baylor College of Medicine, Houston, Texas, USA.

Department of Obstetrics and Gynecology, Baylor College of Medicine, Houston, Texas, USA.

出版信息

Fetal Diagn Ther. 2020;47(8):587-596. doi: 10.1159/000504977. Epub 2020 Mar 6.

Abstract

OBJECTIVE

To determine postnatal neurodevelopmental (ND) outcomes for children with congenital lower urinary tract obstruction (LUTO) based on disease severity.

METHODS

Twenty male infants with LUTO were classified prenatally as Stage 1 (normal amniotic fluid and renal function; n = 5), Stage 2 (signs of obstruction with preserved renal function; n = 13), and Stage 3 (signs of severe renal damage; n = 2). ND status was assessed using the Developmental Profile-3 test in 5 developmental domains (physical, adaptive behavior, social-emotional, cognitive, and communication). Each domain was considered to be delayed if standard scores were 2 or more SD below the mean. ND outcomes were compared between cases with an expected normal renal function (LUTO Stage 1) and those with impaired renal function (LUTO Stages 2 and 3). Results from cases with Stage 2 were also compared to those from Stage 3. ORs were calculated to predict risk for adverse ND outcome for each domain considering prenatal and postnatal factors.

RESULTS

Gestational age (GA) at the diagnosis of LUTO was similar between both groups (Stage 1: 24.85 ± 7.87 vs. Stages 2 and 3: 21.4 ± 4.31 weeks; p = 0.24). Twelve of 15 cases with Stage 2 or 3 underwent vesicoamniotic shunt placement compared to none of Stage 1 fetuses (p < 0.01). No differences in GA at delivery were detected between the groups (37.9 ± 1.6 vs. 35.1 ± 3.6 weeks; p = 0.1). One of the infants in the Stage 2 and 3 groups received a kidney transplant during follow-up. One case (20%) from Stage 1 group required dialysis during the first 6 months of life, and 1 case from Stage 2 to 3 group required it during the first 6 months (p = 1.0), whereas 3 additional cases needed dialysis from 6 to 24 months (p = 0.6). Mean age at Developmental Profile 3 (DP-3) testing was 20.3 ± 12.3 months (Stage 1: 11.2 ± 8.6 vs. Stages 2 and 3: 23.4 ± 13.4 months; p = 0.07). Fifteen of the 20 patients (75%) had no ND delays. Of the 5 patients with ND delays, 4 received dialysis. No differences in ND outcomes between infants with LUTO Stage 1 and those with Stages 2 and 3 were detected except for a trend toward better physical development in Stage 1 (102.6 ± 11.6 vs. 80.7 ± 34.9; p = 0.05). Infants diagnosed with LUTO Stage 3 showed significantly lower adaptive scores than those diagnosed with Stage 2 (Stage 2: 101.9 ± 22.3 vs. Stage 3: 41.5 ± 30.4; p = 0.04) and a nonsignificant trend for lower results in physical (85.8 ± 33.0 vs. 47.5 ± 38.9; p = 0.1) and socio-emotional (94.7 ±17.9 vs. 73.5 ± 13.4; p = 0.1) domains. Infants who received dialysis showed 15-fold increased risk (95% CI 0.89-251) for delayed socio-emotional development (p = 0.06). Diagnosis of fetal renal failure increased the risk for delays in the adaptive domain 30-fold (95% CI 1.29-93.1; p = 0.03). Infants with abnormal renal function had 19 times (95% CI 1.95-292) increased risk for delays in the physical domain (p = 0.03).

CONCLUSIONS

While most patients with LUTO do not exhibiting ND delays, our results support the importance of ND monitoring, especially in severe forms of LUTO, as increased severity of this condition may be associated with poorer ND outcomes.

摘要

目的

根据疾病严重程度,确定先天性下尿路梗阻(LUTO)患儿的产后神经发育(ND)结局。

方法

20 名男性 LUTO 婴儿在产前被分为 Stage 1(羊水和肾功能正常;n=5)、Stage 2(有梗阻迹象但肾功能正常;n=13)和 Stage 3(严重肾功能损害;n=2)。使用发育概况-3 测试在 5 个发育领域(身体、适应行为、社会情感、认知和沟通)评估 ND 状态。如果标准分数低于平均值 2 个标准差,则认为每个领域都存在延迟。将预期肾功能正常的 LUTO 病例(LUTO Stage 1)与肾功能受损的病例(LUTO Stages 2 和 3)进行 ND 结局比较。还比较了 Stage 2 与 Stage 3 的结果。考虑到产前和产后因素,计算了每个领域发生不良 ND 结局的风险的优势比(OR)。

结果

两组 LUTO 诊断时的胎龄(GA)相似(Stage 1:24.85±7.87 与 Stages 2 和 3:21.4±4.31 周;p=0.24)。15 例 Stage 2 或 3 中有 12 例行膀胱羊膜分流术,而无 Stage 1 胎儿(p<0.01)。两组间分娩时的 GA 无差异(37.9±1.6 与 35.1±3.6 周;p=0.1)。在随访期间,Stage 2 和 3 组中有 1 例婴儿接受了肾移植。1 例 Stage 1 组的婴儿(20%)在生命的前 6 个月需要透析,1 例 Stage 2 到 3 组的婴儿在生命的前 6 个月需要透析(p=1.0),而另外 3 例需要在 6 到 24 个月期间透析(p=0.6)。接受发育概况-3(DP-3)测试的平均年龄为 20.3±12.3 个月(Stage 1:11.2±8.6 与 Stages 2 和 3:23.4±13.4 个月;p=0.07)。20 例患者中,15 例(75%)无 ND 延迟。5 例 ND 延迟的患者中,4 例接受了透析。除了 Stage 1 组在身体发育方面的趋势较好(102.6±11.6 与 80.7±34.9;p=0.05)外,LUTO Stage 1 与 Stages 2 和 3 组之间的 ND 结局无差异。与诊断为 Stage 2 的婴儿相比,诊断为 Stage 3 的婴儿的适应性评分显著降低(Stage 2:101.9±22.3 与 Stage 3:41.5±30.4;p=0.04),身体(85.8±33.0 与 47.5±38.9;p=0.1)和社会情感(94.7±17.9 与 73.5±13.4;p=0.1)领域也有非显著的较低趋势。接受透析的婴儿社会情感发育延迟的风险增加 15 倍(95%CI 0.89-251;p=0.06)。胎儿肾功能衰竭的诊断使适应域延迟的风险增加 30 倍(95%CI 1.29-93.1;p=0.03)。肾功能异常的婴儿身体域延迟的风险增加 19 倍(95%CI 1.95-292;p=0.03)。

结论

虽然大多数 LUTO 患者没有 ND 延迟,但我们的结果支持 ND 监测的重要性,尤其是在严重形式的 LUTO 中,因为这种疾病的严重程度增加可能与较差的 ND 结局相关。

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