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后颅窝疝反转在神经管缺陷产前修复后的意义。

The significance of hindbrain herniation reversal following prenatal repair of neural tube defects.

机构信息

1Midwest Fetal Care Center, Children's Minnesota, Minneapolis, Minnesota.

Departments of2Obstetrics, Gynecology, and Reproductive Sciences.

出版信息

J Neurosurg Pediatr. 2023 Mar 24;32(1):106-114. doi: 10.3171/2023.2.PEDS22457. Print 2023 Jul 1.

DOI:10.3171/2023.2.PEDS22457
PMID:36964730
Abstract

OBJECTIVE

The aim of this study was to determine whether reversal of hindbrain herniation (HBH) on MRI following prenatal repair of neural tube defects (NTDs) is associated with reduced rates of ventriculoperitoneal (VP) shunt placement or endoscopic third ventriculostomy (ETV) within the 1st year of life.

METHODS

This is a secondary analysis of prospectively collected data from all patients who had prenatal open repair of a fetal NTD at a single tertiary care center between 2012 and 2020. Patients were offered surgery according to inclusion criteria from the Management of Myelomeningocele Study (MOMS). Patients were excluded if they were lost to follow-up, did not undergo postnatal MRI, or underwent postnatal MRI without a report assessing hindbrain status. Patients with HBH reversal were compared with those without HBH reversal. The primary outcome assessed was surgical CSF diversion (i.e., VP shunt or ETV) within the first 12 months of life. Secondary outcomes included CSF leakage, repair dehiscence, CSF diversion prior to discharge from the neonatal intensive care unit (NICU), and composite neonatal morbidity. Demographic, prenatal sonographic, and operative characteristics as well as outcomes were assessed using standard univariate statistical methods. Multivariate logistic regression models were fit to assess for independent contributions to the primary and secondary outcomes.

RESULTS

Following exclusions, 78 patients were available for analysis. Of these patients, 38 (48.7%) had HBH reversal and 40 (51.3%) had persistent HBH on postnatal MRI. Baseline demographic and preoperative ultrasound characteristics were similar between groups. The primary outcome of CSF diversion within the 1st year of life was similar between the two groups (42.1% vs 57.5%, p = 0.17). All secondary outcomes were also similar between groups. Patients who had occurrence of the primary outcome had greater presurgical lateral ventricle width than those who did not (16.1 vs 12.1 mm, p = 0.02) when HBH was reversed, but not when HBH was persistent (12.5 vs 10.7 mm, p = 0.49). In multivariate analysis, presurgical lateral ventricle width was associated with increased rates of CSF diversion before 12 months of life (adjusted OR 1.18, 95% CI 1.03-1.35) and CSF diversion prior to NICU discharge (adjusted OR 1.18, 95% CI 1.02-1.37).

CONCLUSIONS

HBH reversal was not associated with decreased rates of CSF diversion in this cohort. Predictive accuracy of the anticipated benefits of prenatal NTD repair may not be augmented by the observation of HBH reversal on MRI.

摘要

目的

本研究旨在确定胎儿神经管缺陷(NTD)产前修复后磁共振成像(MRI)上小脑蚓部疝(HBH)逆转是否与出生后第 1 年内脑室腹腔(VP)分流或内镜第三脑室造瘘术(ETV)的发生率降低有关。

方法

这是对 2012 年至 2020 年期间在一家三级护理中心接受胎儿 NTD 产前开放性修复的所有患者的前瞻性收集数据进行的二次分析。根据管理脊髓脊膜膨出研究(MOMS)的纳入标准为患者提供手术。如果患者失访、未进行产后 MRI 检查或进行产后 MRI 检查但没有报告评估小脑蚓部状态,则将其排除在外。将 HBH 逆转患者与无 HBH 逆转患者进行比较。评估的主要结果是出生后 12 个月内进行的 CSF 分流手术(即 VP 分流或 ETV)。次要结果包括脑脊液漏、修复裂开、新生儿重症监护病房(NICU)出院前 CSF 分流以及新生儿复合发病率。使用标准的单变量统计方法评估人口统计学、产前超声和手术特征以及结果。使用多元逻辑回归模型评估对主要和次要结果的独立贡献。

结果

排除后,78 名患者可进行分析。这些患者中,38 名(48.7%)HBH 逆转,40 名(51.3%)在出生后 MRI 上持续存在 HBH。两组间基线人口统计学和术前超声特征相似。两组第 1 年 CSF 分流的主要结局相似(42.1%对 57.5%,p=0.17)。两组间所有次要结局也相似。当 HBH 逆转时,发生主要结局的患者术前侧脑室宽度大于未发生主要结局的患者(16.1 对 12.1mm,p=0.02),但当 HBH 持续存在时则不然(12.5 对 10.7mm,p=0.49)。多元分析显示,术前侧脑室宽度与出生后 12 个月内 CSF 分流率增加相关(调整后的比值比 1.18,95%CI 1.03-1.35)和 NICU 出院前 CSF 分流相关(调整后的比值比 1.18,95%CI 1.02-1.37)。

结论

本队列中 HBH 逆转与 CSF 分流率降低无关。MRI 上观察到 HBH 逆转可能不会增加产前 NTD 修复预期益处的预测准确性。

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